Rutter's Child and Adolescent Psychiatry Book 2

fussing, crying lasting more than 3 hours a day, on more than 3 days in 1 week” and continuing for more than 3 weeks, between the ages of 1 and 4 months. Colic is common, reported as occurring in 10–20% of babies in large community surveys (Crowcroft & Stachan, 1997). There may be little justification for using the term “colic” for a phenomenon that might be more parsimoniously viewed as persistent unexplained infant crying (St. James-Roberts, 2004). There are a number of theories of etiology, focusing on gastrointestinal dysfunction, central nervous system immaturity, and parental stress and anxiety. However, there is very little conclusive evidence to support these theories (Stifter & Wiggins, 2004), or to validate treatments that follow from them (Garrison & Christakis, 2000). Irrespective of cause, colic is a significant cause of parental stress and low confidence. A recent working party (Barr, St. James-Roberts, & Keefe, 2001) concluded that colic could most usefully be seen as a problem resulting from early individual differences in reactivity and the development of regulation. Although colic is seen by parents and professionals as a physical problem, rarely is there any known underlying condition, such as gastrointestinal disorder. Failure to Thrive Historically, there has been little agreement on a precise definition of the term “failure to thrive” (FTT). It has been defined in several ways, including by weight centile, by weightfor-height and by growth faltering over time (Benoit, 2000). Traditionally, FTT was divided into two subgroups: organic and non-organic, the latter term carrying connotations of psychosocial causation, such as parental neglect, abuse or poor management of feeding. However, more recently the assumption of a clear-cut distinction between organic and nonorganic FTT has been called into question (Boddy & Skuse, 1994; Chatoor & Ganiban, 2004). Absence of a physical explanation does not mean that poor parenting is necessarily the cause. However, attributing growth delay to parental neglect may be unhelpful (Skuse, 1985), and in some cases may have harmful consequences. Furthermore, many children with nonorganic FTT turn out later to have undiagnosed illnesses or impairments in oral–motor skills. One population survey (Reilly, Skuse, Wolke, & Stevenson, 1999) found oral–motor dysfunction in 35% of children with non-organic FTT, suggesting that assumptions about parental causes may often be unwarranted. Finally, as discussed in relation to other early behavior problems, FTT is now thought likely to have multifactorial causes, where factors such as temperament, developmental delay and poor feeding skills interact with parental stress and poor management of feeding. In a UK population survey of growth delay (Skuse, Reilly, & Wolke, 1994), a prevalence rate of 3–4% was found. However, it was also noted that there was considerable variation between ethnic groups, with higher rates among South Asian and lower rates in African groups. We should be cautious about assuming that these are related to cultural differences in early feeding patterns; it is also possible that norms are inadequate for defining growth faltering in minority groups. Food Refusal Food refusal is a general term for a range of feeding problems, broader than DSM “Feeding disorder of infancy and early childhood,” as it focuses on child behavior rather than requiring growth failure. It includes children of normal weight who consistently or selectively refuse to eat. Selective refusal may be unpredictable, or may be specific to certain foods or situations (Chatoor & Ganniban, 2004; Douglas, 2002). In toddlers, it may be linked to other behavioral problems, or may be limited to feeding contexts. It may be related to long-standing early feeding problems or arise anew in the “terrible twos.” Again, causes are likely to be multifactorial, including combinations of temperament, family stress and poor parenting skills, as for other oppositional problems. In many cases, there may be more specific contributing factors related to food, including feeding experiences, parental anxiety about food and body shape or prior health problems that might initiate the feeding problem (e.g., pain or anxiety about vomiting or reflux), which may then be unwittingly reinforced by parental reactions (Douglas, 2002; Stein & Barnes, 2002). Treatment General Principles for Selecting Treatments From earlier discussions of multiple interacting factors that contribute to behavioral problems in the 0–5 period, it will be clear that intervention requires assessment of the presenting problems in the context of family and caregiver influences, as well as the child’s development and physical health. Thus, factors such as fussy temperament, parental inconsistency, stress of poverty, early illness and oral–motor delay may all combine to produce toddler eating problems. Knowing the causes of a problem can be helpful for understanding and selecting treatment; however, at the same time it is vital that clinicians use, wherever possible, interventions that have a strong evidence-base, and that they keep up to date with this knowledge as new trials and systematic reviews appear. Thus, knowing that oral–motor dysfunction is contributing to a longstanding eating problem in a toddler born very prematurely is helpful in making sense of a problem, and for reducing parental blame and guilt; however, arguably it is not useful information for treatment if there is no evidence of effectiveness of available speech therapy for oral–motor delay. However, there is better evidence about the effectiveness of parenting interventions for food refusal and other mealtime behavior problems (Kerwin, 1999; Turner, Sanders, & Wall, 1994). Parent management difficulties are only one of several causal factors contributing to early eating, sleep, oppositional or attentional problems, but parenting intervention might nevertheless be the treatment of choice. In this age group, it is developmentally appropriate and has the strongest evidence-base from randomized trials. This is the case even for preschool ADHD, where there is a clear genetic contribution to etiology. Given these considerations, this section focuses on common principles behind evidence-based approaches, particularly BEHAVIORAL PROBLEMS 887 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 887

parent management of toddler behavior problems, as well as including specific guidance for clinicians. We concentrate on interventions with an evidence-base from high-quality systematic reviews, such as those published by the Cochrane Collaboration, and randomized controlled trials (RCTs). Some of these cover a wide age range but, where possible, we have sought effectiveness data applicable to the 0–5 age group. Readers are referred to other chapters for fuller details on particular problems and interventions. Overview of Evidence We begin with a brief overview of evidence on intervention effectiveness. A systematic review of interventions for early conduct problems age 3–8 (Barlow & Stewart-Brown, 2000) concluded that behaviorally based parent-training is effective. Several recent trials that comprise entirely, or high proportions of, preschoolers reach similar conclusions (Gardner, Burton, & Klimes, 2006; Hutchings, Bywater, Daley et al., 2007; Sanders, Markie-Dadds, Tully, & Bor, 2000; Scott, Spender, Doolan, Jacobs, & Aspland, 2001; Webster-Stratton, 1998a), suggesting that interventions are translatable across countries and varied service settings. Some programs appear to be equally effective across ethnic groups; a secondary analysis of 630 preschoolers in trials of the Webster-Stratton preventive parenting program found no differences by ethnicity on any parent or child outcome measures, or on parent engagement and satisfaction (Reid, Webster-Stratton, & Beauchaine, 2001). For sleeping problems, systematic reviews suggest that behavioral parent management of settling and night-waking problems is effective (Mindell, 1999; Ramchandani, Wiggs, Webb, & Stores, 2000). For toddler and preschool eating problems, evidence from two very small RCTs and several case series leads to the tentative conclusion that parent management approaches may be effective (Turner, Sanders, & Wall, 1994). Kerwin’s (1999) review reached a similar conclusion for severe infant feeding problems. The picture is much less clear for early emotional disorders, as very few treatment trials include children under 5. Medication is controversial in this age group, and practice varies between USA and Europe. For ADHD, stimulant medication is not licensed for preschoolers because of worries about safety, and parenting interventions have been found to be as effective in this age group as stimulant drugs are in older children (Sonuga-Barke, Daley, Thompson, LaverBradbury, & Weeks, 2001). Nevertheless, one trial claimed methylphenidate may be effective in preschoolers (Greenhill, Kollins, Abikoff et al., 2006), although it should be noted that the trial only included children for whom parent-training approaches were not effective, and there was quite a high rate of side effects. Tricyclic antidepressants are not recommended in prepubertal children (Hazell, O’Connell, Heathcote, & Henry, 2002), and there are considerable doubts about the effectiveness of selective serotonin reuptake inhibitors (SSRIs) in children of all ages (Whittington, Kendall, Fonagy et al., 2004). Interventions for Disruptive Behavior Problems We begin with principles of parenting interventions for early oppositional and attentional problems, while noting that these broad principles are also applicable to toddler eating and sleeping problems, particularly given the frequent comorbidity and common causes for these problems. Specific interventions for feeding problems follow. (For fuller details on parenting interventions see chapter 64. For useful detailed guides to carrying out parenting interventions in young children see Sutton, 1999; Webster-Stratton, 1992; Webster-Stratton & Herbert, 1994.) Intervention begins with broad assessment of the child’s health, development and temperament, the overall family and caregiving setting, and a detailed evaluation of the frequency, duration and situational determinants of the presenting problems. In particular, assessment should focus on parenting strategies and other situational factors that may be triggering or maintaining the problem on a day-to-day basis. Equally important is examination of parenting strengths and competencies that can be built on in treatment. What are the positive features of the relationship between child and parents? Under what conditions does the problem fail to occur, or how have parents succeeded in preventing or alleviating it? Parenting strategies are best assessed through a combination of parent diary records, direct observation of parent–child interaction and careful interviewing. Observational methods are particularly useful for preschoolers (see chapter 19), who tend to be less reactive to being observed. There is increasing evidence that learning and putting into practice new parenting skills, carefully tailored to the child’s needs, is a critical intervening mechanism (Rutter, 2005) underlying successful interventions for early conduct problems (Forgatch & DeGarmo, 1999; Gardner, Burton, & Klimes, 2006; Gardner, Shaw, Dishion et al., 2007; Hutchings, Lane, & Gardner, 2004). Hence, this parenting assessment information is central to planning the main active ingredients of the intervention. Providing systematic sensitive feedback to parents about the findings of assessment may be helpful for several reasons (Dishion & Kavanagh, 2003; Shaw, Dishion, Gardner et al., 2006), including to motivate parents’ engagement with challenging interventions; to reach a shared formulation and plan for treatment (rather than one that is imposed by the clinician); and to aid later skills coaching. Although enhancing parenting skills is a key part of intervention for behavior problems, it is important to help the parents understand that they are not necessarily the main cause of the problem, rather that they are the best solution. Helping parents understand how the child’s temperament and development (e.g., language or cognitive impairment; see chapters 47 and 49) contribute to parenting and conflict can be very helpful in formulating a problem, and preparing the ground for skills-based work. A preschool ADHD intervention provides an excellent example of this approach (Sonuga-Barke, Daley, Thompson et al., 2001; Thompson, Weeks, & LaverBradbury, 1999). The first part of the intervention involves helping parents to understand and empathize with the individual temperamental characteristics of the child with ADHD, CHAPTER 53 888 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 888

and considering how some of the parents’ expectations and skills may need to be adapted to fit better with the child. Following this, parents work on setting new expectations and limits, and practice strategies to manage the child and change symptomatic behavior. As the majority of children with ADHD show comorbid conduct problems, such approaches are also applicable to changing oppositional behavior (Sonuga-Barke, Daley, Thompson et al., 2001). Following assessment, intervention focuses on applying cognitive–behavioral parenting principles in an individualized manner to the child and family. An example of a wellspecified effective parenting intervention is Webster-Stratton’s (1998a,b; Webster-Stratton & Herbert, 1994) “Incredible Years” program (see chapter 64), which employs a collaborative approach, building on parents’ strengths and expertise. Principles covered include parent–child play, praise, incentives, limit-setting, problem-solving and discipline. Video clips are used to encourage problem-solving around strategies to manage their child, and to illustrate their varying effects on child behavior. Role plays and homework are used to find solutions and practice parenting skills. In each session, homework successes and problems are discussed, and used as further problem-solving examples. Although this program is designed to be delivered to groups of parents, these principles are equally important in individual work with families of young children. Similar effective programs, aimed particularly at 2- to 5-year-olds, and designed to be carried out in a home or clinic setting with individual families, include Sanders, MarkieDadds, Tully et al.s’ (2000) Triple-P program, and Forgatch and DeGarmo’s (1999) Parent Management Training. When advising parents about disruptive behavior, it is helpful to encourage them to observe and record their child’s behavior, the situations that trigger it, and their reactions and attempts at managing the behavior. It is important to help parents build a warm cooperative relationship with the child, especially if this appears to be lacking, by advising a regular daily joint play session, say 5–10 min, using an age-appropriate calm activity. Co-operative play should be encouraged and rewarded. Meanwhile, it is important for parents to explain simple ground rules to the child (behavior that is expected or prohibited, in key situations), and then to reward the child using praise, and token rewards (e.g., sticker or star charts) for behaving well, especially in situations that commonly lead to troublesome behavior. Planning ahead for how to deal with the most difficult situations is an important element. It is possible to plan to ignore milder problem behaviors, and use mild consequences for more severe problems (e.g., aggression, repeated defiance about important issues), such as time out (for 2–3 min at age 2–5) or brief immediate withdrawal of enjoyable activities. For milder problems, a Cochrane review suggests that self-help materials can improve disruptive problem behavior (Montgomery, Bjornstad, & Dennis, 2006). For example, books explaining evidence-based principles for managing early behavior problems (e.g., Sanders, 2004; Webster-Stratton, 1992) can be useful for parents, or for clinicians to work through with parents. Non-attendance is a common problem in treatment services, and it behoves clinicians to find ways to overcome barriers to engagement, especially for distressed and disadvantaged families. Webster-Stratton (1998a,b) achieves this by offering flexible timing of intervention, transport and child care, which are particularly important for families with preschoolers. Other strategies include home-visiting, or using community locations, such as primary healthcare clinics (Stewart-Brown, Patterson, Mockford et al., 2004; Turner & Sanders, 2006). Others have developed specific strategies to enhance parent motivation (Dishion & Kavanagh, 2003; Dishion, Shaw, Connell et al. (in press)). It is undoubtedly challenging to be flexible to parents’ needs when working in a clinic setting; without attention to these issues, attendance rates may remain low and the most troubled children may be unable to access services. Interventions for Emotional Problems There is a good deal of evidence from systematic reviews and randomized trials (Ollendick & King, 1998) to support the use of cognitive–behavioral interventions for anxiety and depression in school-age children, but few treatment trials include children under 5 years. However, there is some evidence from single-case studies and small trials (Ollendick & King, 1998) that the same techniques may be applied to 3- to 5-year-olds, provided the intervention is carefully matched to the children’s developmental level. There is also important evidence emerging about cognitive–behavioral interventions from studies aimed primarily at helping preschool conduct problems, namely Webster-Stratton and Hammond’s (1997) child-focused “Dinosaur” program. They find that cognitive– behavioral techniques, delivered using developmentally appropriate games and puppets, to groups of preschoolers, can reduce comorbid emotional symptoms as well as disruptive problems. Parental involvement in cognitive–behavioral interventions is useful in older children, especially where parents are very anxious, and may well prove to be helpful with younger children, pending further empirical validation (Barrett, Dadds, & Rapee, 1996). Chapter 63 describes detailed principles of cognitive–behavioral interventions for anxiety. These intervention principles can be adapted for the developmental level of preschoolers (Hirshfeld-Becker & Biederman, 2002); for example, using pictures to help with discussions about emotions, and by a greater focus on behavioral strategies, such as graded exposure and modeling to deal with phobias. Interventions for Feeding Problems Colic A systematic review by Garrison and Christakis (2000) found 22 randomized trials of interventions for colic. Most trials were of poor quality, and the reviewers concluded that there was little clear evidence for efficacy of the various interventions tested. Medication did not appear to be effective; nor did infant carrying (although this has been found to be effective for other, less persistent forms of crying). There was slightly more promising support for helping parents to manage crying by BEHAVIORAL PROBLEMS 889 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 889

reducing stimulation or improving communication skills, and some support for dietary interventions, including four small trials of hypoallergenic diets. Lactase was not effective. Because of methodological weaknesses, these results need to be treated with a great deal of caution, as putting infants on restricted diets, although potentially beneficial, could cause harm to the infant and unnecessary stress to the family. A recent trial of 600 neonates tested whether persistent crying could be prevented with behavioral interventions (St. James-Roberts & Gillham, 2001). Only modest effects on sleeping and crying were found, representing an increase of 10% in the number of babies sleeping through the night. Given the evidence from many trials suggesting that most interventions are ineffective for colic, including parental management strategies (e.g., carrying) and dietary change, advice needs to be based on reassurance and information, coupled with acknowledgment and management of parents’ understandably high levels of stress. Parents can be advised that there are no significant long-term consequences of early colic, that it normally peaks around 6 weeks and stops at 3–4 months (Stifter & Wiggins, 2004; St. James-Roberts, 2004), and that the child is not ill, nor necessarily in pain. Failure to Thrive Intervention should aim to assess the contribution of parent management and child feeding skills to mealtime and food intake problems, in order to reduce tense cycles of coercive interaction that often prevail (Skuse, 1985). Direct observations of mealtime interaction (see chapter 19), ideally at home, may be particularly useful for analyzing factors that contribute to maintaining conflict over food. There is some evidence for effectiveness of parent-focused interventions from a small number of trials. Wright, Callum, Birks, and Jarvis’ (1998) RCT of a primary care-based, health visitor-led multidisciplinary intervention for under-twos, involving dietary advice and some parent-management work, found positive effects on growth 1 year later compared to usual health visitor care. Black, Dubowitz, Hutcheson et al. (1995) found more mixed effects for a combined intervention involving clinic-based dietary advice and home-based parenting skills. For under-twos with nonorganic FTT from low-income families, there were no effects on growth; rather, both intervention and control group children improved over the trial period. However, beneficial intervention effects were found in the quality of the home environment and on child language development, suggesting that such interventions may alleviate some of the concomitant psychosocial problems found with FTT. Food Refusal Careful assessment is needed to establish whether food refusal is linked to weight loss, and to reveal the situations in which it occurs, and in toddlers whether it is linked to other oppositional behaviors. In the latter case, feeding problems need to be assessed and managed in the context of other toddler noncompliant behaviors. If assessment reveals that parents are very anxious about food intake, then this may be a factor contributing to mealtime conflict (Stein & Barnes, 2002). Where appropriate, reassuring parents about physical health and growth of the child is important, coupled with advice on strategies for setting reasonable limits on the child and for reducing mealtime conflict (Douglas, 2002; Turner, Sanders, & Wall, 1994). There is modest evidence of effectiveness of interventions for food refusal. No Cochrane or other rigorous systematic review was found. We can draw tentative conclusions from a narrative review by Kerwin (1999), which found two very small RCTs (Turner, Sanders, & Wall, 1994) and 27 single-case designs with quantitative outcome measurement, suggesting that behavioral parent management interventions may be effective. Searching did not reveal further RCTs since 1999. When dealing with food refusal and other problem behavior at mealtime in 2- to 5-year-olds, clinicians need to adapt the broad principles outlined for disruptive behavior. Additionally, it is important with mealtime problems to plan how to help eating to become a positive experience and how to reduce stress and conflict over food, particularly by taking a graduated approach to changing the specific problem. Douglas (2002) provided very helpful advice to clinicians. For example, to help food refusal and fussy eating, parents should cease force-feeding and angry responses, and instead take small steps to introduce new habits. It is important to switch to providing praise and attention for eating, and to reduce parents’ natural response, which is to attend to a child when they refuse to eat. Where child anxiety appears to be factor, modeling pleasure in eating and even playing with food can be helpful in establishing enjoyment and reducing anxiety around meals. This may be particularly difficult for parents who feel very anxious around food and its mess, and they may need a good deal of support and encouragement to change (see chapter 27). Conclusions It will be apparent that the state of knowledge about preschool behavior problems is quite uneven. A good deal is known about preschool disruptive behavior, and about effective preventive and treatment interventions for this age group. This is particularly true for oppositional problems, and somewhat less so for ADHD. There is a growing literature on how interventions for early disruptive behavior that have been tested in specialized settings (i.e., “efficacy” trials) can be translated to other settings and cultures, and how they might be disseminated on a wider scale. In contrast, little is known about preschool emotional problems, with poor agreement on classification and little evidence about causes and treatment. To some extent, the same applies to childhood emotional disorders in general. This area should be a priority for future basic and treatment research, especially if it can be confirmed that these problems are stable and significant from an early age. Feeding problems have a longer history of treatment research, much of it from pediatric multidisciplinary teams, but work in this area is still hampered by lack of an agreed classification. Furthermore, understanding diagnosis and causes is complicated by great clinical variability in children who may have CHAPTER 53 890 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 890

accompanying disabilities and developmental delays, combined with psychogenic feeding problems. At the younger end of the 0–5 range, in the specialty known as infant psychiatry, diagnosis is much more controversial and there is a need to establish validity or otherwise of categories and, eventually, effectiveness of treatments that follow from diagnosis. To further understand causal mechanisms, and to delineate the active ingredients of interventions in this age group, work is needed on intervention mechanisms, including gene–environment interactions (Caspi, McClay, Moffitt et al., 2002), to address questions about whether interventions operate in different or similar ways in early childhood compared to older ages. References Achenbach, T. M. (1992). Manual for the Child Behavior Checklist 2/3 and 1992 profile. Burlington, VT: University of Vermont Department of Psychiatry. American Psychiatric Association (APA). (2000). 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894 Sleep problems occur frequently in children and adolescents – particularly among youth with behavioral and/or emotional disorders. Moreover, difficulties related to sleep often overlap with psychiatric problems, making sleep an important domain of symptoms relevant to the diagnosis and treatment of psychopathology. For example, sleep difficulties are a common symptom of depression and improving sleep can contribute positively to the management of mood disorders (Dahl, 1996a,b). A second example is illustrated by the way sleep deprivation can create irritability and difficulties with focused attention that mimic or exacerbate symptoms of attention deficit/hyperactivity disorder (ADHD) and improvements in sleep can have a positive impact on attentional functioning (Chervin, Archbold, Dillon et al., 2002; Dahl, Pelham, & Wierson, 1991). Thus, not only is a knowledge about the assessment, diagnosis and treatment of sleep problems in youth an important clinical domain, but acquiring a solid knowledge of sleep – including its development and disorders – provides a critical foundation of expertise relevant to understanding (and effectively intervening in) a broad range of clinical problems in child and adolescent psychiatry. At a pragmatic level, pediatric sleep problems in children range from highly specific disorders such as narcolepsy where the pathophysiology involves alterations in specific neural systems underpinning sleep–wake regulation to simple behavioral difficulties such as bedtime resistance and late erratic schedules (for estimates of prevalence rates see Table 54.1). However, it is important to recognize that even simple and seemingly minor behavioral sleep problems can become a source of major distress and conflict in children and their families, and these can have serious health consequences. There is increasing evidence that sleep has an important role in basic aspects of learning and memory consolidation as well as impacting on the regulation of behavior and emotions. Therefore, inadequate or disturbed sleep in childhood and adolescence may have long-term consequences on learning and the development of self-regulation that have major clinical implications for child and adolescent psychiatry – crucial questions at the forefront of current research. For the clinician wanting to provide effective prevention, diagnosis and treatment of sleep problems or appropriate referral for the evaluation of more serious disorders, knowledge and clinical skills are needed in four areas: 1 Understanding normal sleep physiology and the normal development of sleep patterns in children and adolescents. 2 Knowledge of common sleep disorders, including aspects of the etiology, pathophysiology and differential diagnoses for common symptoms. 3 Clinical skills and knowledge relevant to assessing sleep habits and symptoms and diagnosing sleep disorders. 4 Knowledge of the relevant treatment principles – particularly behavioral interventions. Accordingly, in this chapter we provide an overview of sleep physiology, organization and function, including emerging findings on the role of sleep in learning and regulatory control in the developing brain, and discuss five key examples of sleep disorders in youth, including assessment, diagnosis and approach to treatment. Physiology, Organization and the Function of Sleep during Human Development Beginning in infancy, the brain cycles through stages of neural activity/behavioral states which correspond to periods of wakefulness and different stages of sleep. Sleep is divided into two major categories: rapid eye movement (REM) sleep and non-REM (NREM) sleep. REM sleep is characterized by high levels of desynchronized cortical electroencephalogram (EEG) activity (mixed frequencies, relatively low voltage), absence of muscle tone, irregular heart rate and respiratory patterns, and episodic bursts of phasic eye movements. NREM sleep is characterized by low-frequency, high-voltage EEG activity, low muscle tone and absence of eye movements. NREM sleep is subdivided into four stages: stage 1 occurs at transitions of sleep and wakefulness; stage 2 is characterized by frequent bursts of rhythmic EEG activity, called sleep spindles, and highvoltage slow spikes, called K-complexes; stages 3 and 4 (also called slow wave sleep [SWS] or delta sleep), represent the deepest stages of sleep and are comprised largely of highvoltage EEG activity in the slowest (delta) frequency range. Two independent regulatory processes appear to interact in regulating the timing, intensity and duration of sleep: a homeostatic sleep process and a circadian sleep process (Borbély, 1982; Borbély & Achermann, 2005). The first, often called Sleep Disorders 54 Ronald E. Dahl and Allison G. Harvey 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 894 Rutter’s Child and Adolescent Psychiatry, 5th Edition, Edited by M. Rutter, D. V. M. Bishop D. S. Pine, S. Scott, J. Stevenson, E. Taylor and A. Thapar © 2008 Blackwell Publishing Limited. ISBN: 978-1-405-14549-7

“Process S,” represents a sleep–wake dependent homeostatic component of sleep that increases as a function of previous wakefulness and gradually decreases over the course of a sleep period. Process S has been associated with basic restorative processes of the brain. Intriguing from a developmental neuroscience perspective is the recent evidence that sleep homeostasis is involved in learning and neural plasticity – the progressive strengthening of neural circuits that occurs through use during wakefulness (Tononi & Cirelli, 2003, 2006). It appears that the progressive increase in synaptic strength from daytime activity must be “downscaled” during sleep to maintain synaptic homeostasis. This homeostatic aspect of sleep also appears to correlate with the deep stages 3 and 4 (slowwave) sleep, which is particularly strong early in development. The second process, often called “Process C,” is the circadian or “biological clock” component. The neurobehavioral underpinnings of this circadian system have been well delineated, including the identification of specific neuroanatomical and molecular components (Carskadon, Acebo, & Jenni, 2004). From a developmental perspective, both the homeostatic and the circadian processes undergo significant maturational changes (Jenni & LeBourgeois, 2006). Some of these developmental changes have important clinical implications. For example, the circadian phase marker, melatonin, correlates with pubertal stage such that more mature adolescents show a delay in the melatonin excretion pattern, even after controlling for psychosocial influences on sleep–wake patterns in adolescence (Carskadon, 2002). This contributes to the tendency to stay up later and sleep in later among adolescents (see p. 898). Furthermore, evidence is accumulating that the dynamics of sleep homeostatic processes slow down over the course of childhood (i.e., sleep pressure accumulates more slowly with increasing age), enabling children to be awake for consolidated periods during the day (Jenni & Carskadon, 2005; Jenni & LeBourgeois, 2006). Recent progress in understanding the neural mechanisms involved in key aspects of the basic regulation of sleep and wakefulness (Saper, Cano, & Scammell, 2005; Saper, Scammell, & Lu, 2005) has focused on complex interactions across a number of different cell groups involved in cortical arousal, with a key part being played by sleep-inducing structures in the ventrolateral preoptic nucleus in the hypothalamus. These regulatory systems are becoming understood in greater detail in ways that are likely to inform clinically relevant aspects of sleep problems. Two specific examples discussed briefly include the role of sleep in learning and sleep and emotion regulation. Sleep, Learning and the Developing Brain One of the most intriguing lines of sleep research with clinical relevance to child and adolescent psychiatry is the growing understanding of the role of sleep in the consolidation of learning (Hobson & Pace-Schott, 2002; Peigneux, Laureys, Delbeuck, & Maquet, 2001; Stickgold, 2005; Stickgold & Walker, 2005; Walker & Stickgold, 2004). A central hypothesis is that sleep facilitates brain plasticity – the ability to constantly modify cerebral structures and functions depending on specific experience and environmental inputs. This view is supported by studies showing impaired memory consolidation following episodes with sleep loss (Pilcher & Huffcut, 1996), and recently there have been findings suggesting such a link at the cellular and molecular levels (Benington & Frank, 2003; Steriade, 1999). Studies have shown the influence of sleep on post-training consolidation, as well as initial memory encoding (Walker & Stickgold, 2006). One recent study demonstrated that sleep can enhance specific components of language learning in infants (Gomez, Bootzin, & Nadel, 2006). These data indicate that adequate sleep may be essential to making enduring neural changes through learning. This role of sleep in learning may be particularly important during periods of rapid brain maturation in childhood and adolescence. SLEEP DISORDERS 895 Sleep disorder or behavior Prevalence estimate (%) Reference Behavioral insomnia of children 10–30 AASM (2005) (including bedtime refusals and night wakings) Sleep talking 55 Owens et al. (2003) Bruxism 28 Owens et al. (2003) Night terrors 17 Owens et al. (2003) Rhythmic movements 17 Owens et al. (2003) Confusional arousals 17 Littner et al. (2005) Sleepwalking 14 Owens et al. (2003) Nightmares 10–50 Sheldon et al. (2005) Insomnia Children 1–6 AASM (2005) Adolescents 11 Hoban & Chervin (2005) Delayed sleep phase 7–17 Lu, Manthena & Zee (2006) Restless leg syndrome/PLMD 1–17 Chervin et al. (2002) Narcolepsy 0.05 Dauvilliers et al. (2003) AASM, American Academy of Sleep Medicine; PLMD, period limb movement disorder. Table 54.1 Prevalence estimates for sleep disturbance in children and adolescents. 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 895

These findings have implications regarding the importance of sleep to educational goals as well as clinical relevance, because learning (and its consolidation) underpins many clinical interventions such as cognitive–behavioral therapy. Sleep and Emotion Regulation Over the past decade there has been increasing interest in understanding the bidirectional relationships between sleep disturbances and problems with emotion regulation in children and adolescents. There is growing evidence that problems with sleep can create and/or exacerbate emotional problems and that emotional difficulties can interfere with sleep. These bidirectional interactions (and the potential for a vicious cycle of negative effects) may have particular clinical relevance in several clinical domains including anxiety, depression and bipolar disorders in youth (Dahl, 1996a; Harvey, Mullin & Hinshaw, 2006) and with aggression, anger and impulse control (Haynes, Bootzin, Smith et al., 2006). With respect to sleep and mood, evidence from the sleep deprivation literature suggests that one of the strongest adverse effects of sleep deprivation is increased negative mood (Dinges, Pack, Williams et al., 1997; Pilcher & Huffcutt, 1996; Van Dongen, Maislin, Mullington, & Dinges, 2003). This makes sense given evidence suggesting that REM sleep has an emotional processing and mood regulation function (Stickgold, Hobson, Fosse, & Fosse, 2001). In adults with bipolar disorder, the case for sleep contributing to mood disturbance is fairly compelling: sleep loss is highly correlated with daily manic symptoms (Barbini, Bertelli, Colombo, & Smeraldi, 1996); among patients with bipolar disorder, sleep disturbance is the most common prodrome of mania and the sixth most common prodrome of depression (Jackson, Cavanagh, & Scott, 2003); and induced sleep deprivation triggers hypomania or mania in a proportion of patients (Colombo, Benedetti, Barbini, Campori, & Smeraldi, 1999; Kasper & Wehr, 1992; Leibenluft, Albert, Rosenthal, & Wehr, 1996; Wehr, Sack, & Rosenthal, 1987; Wu & Bunney, 1990; Zwi, Shawe-Taylor, & Murray, 2005). In youth with bipolar disorder, rates of significant sleep disturbance range from 35 to 45% (for review see Harvey, Mullin, & Hinshaw, 2006). However, in these younger samples, research and clinical questions relating to a possible link between sleep disturbance and mood are yet to be addressed. With respect to sleep and aggression, there are both human and animal data showing increased aggression and impulsivity following experimental sleep loss. For example, rats show increases in aggression and defensive fighting after sleep deprivation. One recent study found that animals who were easy to handle at baseline became irritable and aggressive following modest amounts of sleep deprivation, with evidence of related changes in synaptic plasticity associated with these behavioral changes (Marks & Wayner, 2005). Clinical studies of children and adolescents also have revealed associations between sleep deprivation and irritability/aggression and difficulties with self-regulation in youth (Chervin, Dillon, Archbold, & Ruzicka, 2003; Dahl, 1996b; Ireland & Culpin, 2005). Most relevant is a recent study by Haynes, Bootzin, Smith et al. (2006) which examined behavioral and emotional changes in adolescents with substance-related difficulties undergoing a behavioral sleep treatment. This study reported that improvements in sleep time were associated with significant decreases in the reporting of aggressive thoughts and actions. Taken together, these data suggest that inadequate sleep in adolescence may contribute to aggressive thoughts and actions and that increased or improved sleep may reduce problematic aggression, at least in some cases. While the clinical research questions about the interrelationships between sleep and emotional regulation in children are compelling, it is also important to acknowledge that from a clinical practice perspective, traditional polysomnographic studies have not yielded clear evidence of specific sleep abnormalities associated with particular psychopathologies in youth (e.g., ADHD, major depressive disorder [MDD] ). Thus, polysomnographic studies are best utilized clinically when a specific sleep disorder is suspected. Genetic Contributions To date, scant attention has been paid to investigations of possible genetic contributions to sleep disturbance in children and adolescents. The exception is a study by Gruber, Grizenko, Schwartz et al. (2006), who investigated the hypothesis that the catecholamine-O-methyltransferase (COMT) polymorphism modulates disturbance in children with ADHD. Sleep was measured over 2 weeks with actigraphy. The rationale for investigating COMT is its association with sleep in narcolepsy and Parkinson’s disease and disorders related to dopamine dysfunction. Gruber, Grizenko, Schwartz et al. found that children who had the COMT high-activity allele, val (i.e., val/val or val/met) exhibited greater sleep disturbance compared with children who were met homozygous genotypes (i.e., met/met). The authors interpreted these findings as adding to the accruing evidence base suggesting that sleep disturbance is core to the pathology underpinning ADHD and that there may be a genetic basis for the association between ADHD and sleep disturbance. Specific Clinical Problems Problems Going to Sleep and Staying Asleep Bedtime struggles and middle of the night wakings are not only a source of sleep disruption for children and their parents, but also can be a source of conflict and negative emotion among family members, contributing to negative parent–child interactions, marital discord and, in some rare cases, child abuse. One aspect of the difficulty is that young children often show a “paradoxical” reaction when obtaining insufficient or inadequate sleep. That is, sleep-deprived young children (whether from insufficient or disrupted sleep) often look irritable and impulsive, with some symptoms of distractibility and emotional lability, and may seem overly active. 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are: (i) creating an emotional state of calmness and safety; (ii) consistent limit setting; and (iii) establishing good habits. Bedtime habits include a wind-down period and a sequence of activities that begin 30–60 min before bedtime. If a child continues to test limits and not to respond to strategies to reward desirable behaviors, it may become necessary to set consequences for the undesirable behaviors. However, this can still be done within a positive framework to avoid the escalating emotional arousals of yelling, screaming and flaring tempers. Insomnia is a common complaint among older children and adolescents, particularly sleep-onset insomnia. Two theoretical perspectives are helpful for conceptualizing insomnia in children and adolescents. First, a vigilance–avoidance model has particular relevance to understanding sleep problems in childhood and adolescence (Dahl, 1996b). At the most fundamental level, sleep and vigilance represent opponent processes. Physiologically, going to sleep requires turning-off awareness and responsiveness to threats in the external environment. Because of this loss of vigilance and responsiveness, sleep is naturally restricted to “safe” times and places (e.g., animals and birds tend to sleep in safe burrows, nests or temporal niches that minimize dangers). In humans, one important domain for assessing the safe conditions that permit and promote sleep focuses on social and emotional appraisals of threat (Dahl, 1996b; Worthman & Melby, 2002). Individuals experiencing high levels of anxiety, vigilance and perceived threats often have difficulties going to sleep. While these tendencies have adaptive advantages (inhibiting sleep in truly dangerous environments), they also have costs: high levels of anxiety and vigilance based on perceived threats can lead to chronic sleep difficulties. A major source of difficulty for children and adolescents is rooted in presleep onset worries and ruminative thinking at bedtime – which naturally increase vigilance and interfere with going to sleep. Yet, it can be equally problematic if anxious youth avoid these distressing bedtime worries by staying up (or getting back out of bed) to engage in very late hours of school work, watching TV or socializing (via the Internet or cell phones). These patterns of behavior often lead to difficulty getting to sleep until very late on school nights and thus insufficient sleep because of the need to get up early for school. Without effective intervention, this pattern of vigilance and avoidance can lead to extremely problematic sleep habits and patterns in adolescents with anxiety disorders. Second, a conditioning model of insomnia (Bootzin, 1979; Perlis, Giles, Mendelson, Bootzin, & Wyatt, 1997) emphasizes the contribution to insomnia of multiple pairings of the bed and not sleeping. This set of associations likely results in conditioning between the bedroom environment and heightened arousal, which serves to maintain the sleep disturbance. The initial assessment of youth with insomnia should start with a thorough clinical interview with the patient and their family members to obtain information about the duration, frequency and severity of night-time sleep disturbance, including estimates of the key sleep parameters: sleep onset latency (SOL), number of awakenings after sleep onset (NWAKE), total amount of time awake after sleep onset (WASO), total sleep time (TST) and an estimate of sleep quality (SQ). This information should encompass both weekday and weekend schedules and should assess the regularity of the schedule as well as the average schedule. The onset and duration of the insomnia and type of symptoms (i.e., sleep onset, sleep maintenance, early morning or late morning awaking problem or combinations of these) should be assessed. A description of the daytime correlates and consequences of insomnia is a key domain of importance as is gauging parental responses to the insomnia. In addition, it is crucial to obtain information about medications (prescription and over-the-counter) and conduct a screen for the presence of psychiatric disorders and medical problems (including other sleep disorders). Asking the patient to complete a sleep diary each morning, as soon as possible after waking, for 1–2 weeks provides a wealth of information including an insight into the night-to-night variability in sleeping difficulty and sleep–wake patterns and the presence of circadian rhythm disorders, such as delayed sleep phase disorder. An assessment of insomnia in youth should include an exploration of the child’s fears, because fear, distress or any cognitive or emotional cue that prevents an overall sense of safety can be antithetical to sleep (because sleep onset requires turning off awareness, responsiveness and vigilance). Children and adolescents can often get into the habit of mentally reviewing or replaying worrisome thoughts or memories of stressful events. Some youth with stressful memories or specific fears manage to avoid these during the day through distracting activities, but at night – especially when trying to fall asleep – the lack of distracters can result in rumination on these thoughts. The treatment principles are threefold. First, helping the child feel emotionally and physically safe is often a critical component. (The emphasis on the word feel is because the affective appraisal of being safe appears to be extremely important for some youth – they may logically understand the absence of specific threats but unless they have an emotional sense of safety they may experience heightened vigilance that interferes with sleep.) Depending on the age and inclinations of the child, this can involve creative imagery, self-hypnosis techniques or systematic relaxation exercises. In addition, parents are encouraged to contribute to an emotional sense of safety, positive emotions and good associations at bedtime. The second treatment principle is to establish a regular bedtime routine and consistent sequences of steps and habits. In children, this can involve a bedtime story, special quiet time with the parent and pleasant wind-down time. In adolescence, multiple sleep-interfering factors (e.g., cell phones, the Internet) interact with the pubertal phase delay in the circadian system causing a tendency to shift toward later bedtimes and increased autonomy to decide on bedtimes. Hence, the intervention is to identify sleep-interfering behaviors and work collaboratively with the young person to assist them to find internal motivations for enhancing their sleep by reducing the use of sleepinterfering technologies around bedtime. In addition, derived from the conditioning theory just discussed, there is preliminary evidence that the stimulus control intervention that has SLEEP DISORDERS 897 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 897

been so successful for adult insomnia (Morin, Bootzin, Buysse et al., 2006) can also be helpful for youth (Bootzin & Stevens, 2005). The therapist provides a detailed rationale for and assists the patient to achieve the following: 1 Use the bed and bedroom only for sleep (i.e., no TV watching or text messaging). 2 Get out of bed and go to another room whenever you are unable to fall asleep or return to sleep within approximately 15–20 min and return to bed only when sleepy again. 3 Arise in the morning at the same time (no later than plus 2 hours on weekends) and gradually move closer to a regular schedule 7 days a week. Third, based on the vigilance–avoidance model, there is a clear need to address bedtime worry, rumination and vigilance (Dahl, 2002). Interventions for worry and rumination include: diary writing or scheduling a “worry period” to encourage the processing of worries several hours prior to bedtime, creating a “to do” list prior to getting into bed to reduce worrying about future plans or events, training to disengage from presleep worry and redirect attention to pleasant (distracting) imagery, demonstrating the adverse consequences of thought suppression while in bed and scheduling a presleep “wind-down” period prior to bedtime. The Adolescent with Daytime Sleepiness/ Difficulty Waking up for School After the onset of puberty and throughout the adolescent period, the most prevalent sleep complaints tend to center on difficulty waking up for school in the morning and the associated difficulties with daytime sleepiness, tiredness and irritability. It is important to emphasize that there is a broad continuum of severity. This ranges from normative or mild difficulties that appear to affect up to half of all high-school students in the USA and result in at least some symptoms (National Sleep Foundation, 2006), to a much smaller subset of adolescents with severe difficulties with sleep and schedules that result in significant impairments (such as failures in school) and often meet full criteria for a delayed sleep phase syndrome (DSPS). At the current time, there is an absence of empirical data to help delineate precisely when to diagnose adolescents as having DSPS from the much larger set of youth with mild to moderate problems with erratic and late sleep schedules. One pragmatic approach advocated in this chapter is that clinicians understand both the physiological and social influences that contribute to these problems, and apply sound behavioral principles aimed at improving and increasing sleep in any adolescent who shows evidence of suboptimal sleep as a result of late nights and erratic schedules. In other words, many of the simple behavioral interventions to regularize sleep–wake schedules and increase total sleep can be tried even in the case of mild to moderate problems (because these interventions have little to no risks or side effects and can result in improved alertness as well as improved mood regulation). It is important to understand the interaction between biological and social processes underpinning these common problems. Biological changes in the circadian system at puberty shift sleep timing preferences in the direction of delayed sleep phase (Carskadon, 2005; Lee, Hummer, & Jechura, 2004). Unfortunately, these biological changes interact with early school start times, increase in paid work responsibilities and increased amounts of time devoted to socializing in a way that can spiral quickly into a pattern of extreme sleep deprivation. Then most “catch-up” sleep occurs on weekends and holidays on an extremely phase-delayed schedule. The human circadian system adapts more easily to phase delays as endogenous rhythms of body temperature and neuroendocrine function are able to reset quickly to later bed and wake times. However, the circadian system has more difficulty accommodating to phase advances (earlier sleep schedules). This creates an effect that is a bit like “jet-lag” on Monday morning; students try to adjust to a sudden phase advance in which they need to get up for school at a time that is often 3–4 hours (or more) earlier than their biological clock “expects” to wake up. A thorough and detailed history is essential to evaluate and to characterize the nature of the sleepiness. This should include an assessment of whether the young person is fatigued or unable to stay awake, the frequency and duration of symptoms and whether the symptoms occur at particular times of day or only in certain situations. A family history of increased sleep needs and sleepiness can be important in the consideration of narcolepsy. In adolescents with true sleepiness (not simply complaints of fatigue), two main categories of problems should be considered: 1 Inadequate amounts of sleep; and 2 Circadian and scheduling disorders. Inadequate Amounts of Sleep The most common cause of mild to moderate sleepiness in adolescents is an inadequate number of hours in bed. A combination of social schedules leading to late nights with early morning school requirements can significantly compress the number of hours of sleep. Part-time jobs, sports activities, hobbies and active social lives can exacerbate this problem. The catch-up sleep of naps, weekends and holidays can also contribute to the problem by leading to erratic schedules and even later nights. In taking a sleep history, it is important to ask specific questions about bedtime schedules. Many families will say the adolescent “usually” goes to bed at a certain time, but when asked for an exact time covering the previous few nights, a much later hour is reported. When assessing the amount of sleep an adolescent is getting, it is important to obtain details of bedtime (such as when the child gets into bed as well as lights-out time), estimates of sleep latency, nighttime arousals, time of getting up in the morning, difficulty getting up, and the frequency, timing and duration of daytime naps. It is also essential to get details of sleep–wake schedules on weekends, as well as during the school week. When this type of specific information is obtained either by interview or by having the family maintain a sleep diary, evidence of inadequate sleep is often apparent. A prospective detailed sleep diary provides the most reliable information. CHAPTER 54 898 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 898

When inadequate sleep is identified, simply recommending that the adolescent go to bed earlier is not likely to be effective. Often, the primary role of the clinician is to help the entire family understand and acknowledge the consequences resulting from the inadequate sleep. Sleep deprivation frequently contributes to many factors that the family identifies as problems, including falling asleep in school, oversleeping in the morning, fatigue and irritability. In cases in which the adolescent’s school or social functioning is significantly impaired by sleep problems, a strict behavioral contract that is agreed upon by the family can be essential. The contract should specify hours in bed (with only small deviations on the weekends) and should target the specific behaviors contributing to bad sleep habits, such as specific late-night activities, erratic napping or oversleeping for school. The choice of rewards for successes and negative consequences for failures, as well as an accurate method of assessing compliance, are essential components of the contract. Circadian and Scheduling Disorders The most common specific problem that is relevant to adolescents is delayed sleep phase syndrome (DSPS). DSPS often begins with a tendency to stay up late at night, sleeping in late and/or taking a late afternoon nap. This process often begins on weekends, holidays or summer vacations. Problems become apparent when school schedules result in morning wake-up battles and difficulties in getting to school. Often, these adolescents cope by taking afternoon naps and getting catchup sleep on the weekends. Although some of these behaviors occur in many normal adolescents, in extreme cases the circadian system can become set to such a late time that even highly motivated adolescents can have difficulty shifting their sleep back to an earlier time. In some instances, the attempts by the adolescent (and their families) to correct the problem go against circadian principles. For example, an adolescent who has been going to bed at 3 am and getting up at noon during a vacation tries to go bed at 10 pm the Sunday night before the first day back at school, and finds that her physiology is quite resistant to sleep. For a few days, she manages to get up for school by overriding the system (despite inadequate sleep) but then takes a long nap after school. Despite numerous nights of trying to go to bed at 10 pm she is unable to shift her temperature cycle and circadian system back to an earlier phase. There is a subgroup of adolescents who appear to have trouble following an early schedule but are not particularly troubled by their late schedule. These adolescents are not motivated to correct the problem, are not particularly troubled by their recurrent experiences of being late for or missing school, and do not show great motivation to change their late-night habits. These adolescents are essentially choosing a late-night schedule. Unless the clinician is able to alter the larger realm of priorities and motivators, these adolescents are very unlikely to respond to any treatment of a sleep schedule problem. The treatment of DSPS consists of two parts. The first is gradually to align the sleep system to the desired schedule. The second is to maintain that alignment. The process of alignment consists of gradual consistent advances in bedtime and wake-up time (15 min per day). It is often best to begin from the time the adolescent usually goes to sleep without difficulty. It is important during this process to avoid any naps and to be consistent across weekends and holidays. In severe cases, some adolescents on very late schedules respond more favorably to going around the clock with successive delays in bedtime. This process has been described as phase delay “chronotherapy.” As already highlighted, because the biological clock tends to run on a 25-hour cycle, it accommodates phase delays more easily than phase advances. Hence, the schedule changes can proceed with larger (2–3 hour) delays per day. An example is described for an adolescent who has been falling asleep at 3 am and getting up at noon. On day 1, he stays up until 6 am then sleeps until 3 pm. On day 2, he goes to bed at 9 am and sleeps until 6 pm. On day 3, he sleeps from noon until 9 pm; day 4, from 3 pm until midnight; day 5, from 6 pm until 3 am; day 6 from 9 pm until 6 am; and day 7, from 10 pm until 7 am. It is important that the adolescent take no naps during the chronotherapy. Upon waking up, he or she should be active and, if possible, during the day have bright light exposure, such as walking outdoors. Although many adolescents do very well with this type of phase-delay chronotherapy, the first weekend or vacation of returning to old habits can undo a lot of hard work. Particularly in the first 2–3 weeks following chronotherapy, rigid requirements should be set about wake-up time. Maintenance of the new circadian phase is based on the same principles but usually can be somewhat less rigorous. For example, if the adolescent wants to stay up late on an occasional weekend night, he or she may be able to do so but should not be permitted to sleep more than 1–2 hours later than the usual wake-up time for school. Strict behavioral contracts worked out with the parents, having specific rewards for success and serious consequences for failures, are essential in this type of intervention. Before moving to the consideration of bright light or melatonin therapy for DSPS in adolescents, it is important to place these comments in a larger context. In our experience, if an adolescent complies with the behavioral program the additional use of a light box or evening melatonin is often not necessary. Moreover, if the adolescent does not comply with the behavioral contract, light and melatonin alone are extremely unlikely to be successful. If the adolescent remains asleep in bed with eyes closed, the bright light beside the bed is not going to exert the needed physiological effect, and if adolescents are continuing to engage in arousing social activities or use of electronic media late at night, taking melatonin is not going to make them go to sleep any earlier. Thus, we conceptualize the use of light or melatonin as a way to augment the (primary) behavioral intervention. For example, an adolescent living in a far northern latitude may experience a complete absence of morning bright light exposure during the winter, awakening and travelling to school in darkness or dim light. The use of melatonin has also been suggested as a potential treatment for this problem. Currently, there are some preliminary data supporting the use of low-dose melatonin in SLEEP DISORDERS 899 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 899

the evening to assist with sleep-onset insomnia, presumably resetting of the timing of sleep to an earlier phase (Smits, Nagtegaal, van der Heijden, Coenen, & Kerkhof, 2001; Smits, van Stel, van der Heijden et al., 2003). In some ways, melatonin appears to act as “chemical darkness” to offset the effects of late-night artificial lights in influencing the biological clock. It is important to note the lack of any safety data regarding melatonin use in adolescents. Others have argued that direct control over exposure to light (darkness in the evening and bright light exposure on awakening) can be more effective than melatonin. Use of Medications for Treating Insomnia and DSPS in Youth One of the most contentious issues in the domain of treating sleep problems in children and adolescents with difficulty going to sleep is the use of medications to promote sleep onset (Owens, Rosen, & Mindell, 2003). Indeed, there are large numbers of medications prescribed by pediatricians, child psychiatrists and family practitioners for children of all ages in attempts to hasten sleep onset. The medications include an unusual array, ranging from relatively ineffective choices like antihistamines to powerfully sedating medications with an absence of any empirical data on risks or benefits in children and adolescents. Some of the most common agents, such as chloral hydrate and clonidine, not only lack empirical data, but would be regarded by adult sleep clinicians as poor choices regarding the relative degree of somnogenic properties compared to potential side effects (e.g., the dose of clonidine needed to induce sleepiness is relatively close to the dose that lowers blood pressure). Adult sleep pharmacology has seen numerous advances, including new generation agents with a much better specificity for sleep, duration of action, and relatively low risk and side effects in adults. Therefore, one might logically argue that some of these medications might represent better treatment options for insomnia in children and adolescents. However, until we have empirical data and controlled trials using these medications in youth (including dosing, side effects and efficacy), we are reluctant to advocate any specific medication at this time. Weiss, Wasdell, Bomben, Rea, and Freeman (2006) have reported data on a small sample of stimulant-treated children with ADHD (n = 27) and sleep onset difficulty. A sleep hygiene intervention was administered first (effect size = 0.67). This intervention involved regularizing the sleep–wake schedule (negotiating a consistent bed and wake time), addressing parental unrealistic expectations about the child’s sleep, and reducing caffeine and naps. Those who did not respond were then randomized to a double-blind cross-over design of 10 days of 5 mg melatonin or placebo. There was a reduction in sleep onset latency of 16 min during melatonin use, relative to placebo (effect size = 0.6). The effect size for the combination of sleep hygiene and melatonin was 1.7. An important next step in evaluating the use of sleep hygiene and melatonin for sleep disturbance in ADHD will be to assess the longevity of the reported improvements in sleep. Ramchandani, Wiggs, Webb, and Stores (2000) published a systematic review of treatments for settling problems in children aged 5 years or younger. Nine studies met the inclusion criteria: four testing a drug, four testing a behavioral treatment and one testing non-directive education. Although the drugs appeared to reduce sleep disturbance in the short term, long-term effects were considered by the authors to be questionable. In contrast, behavioral interventions showed both short-term and longer-term efficacy. The behavioral interventions included positive routines and graduated extinction of parental involvement in resettling across the night. The Child with Parasomnias Parasomnias occur commonly in children as sudden partial awakenings from deep NREM sleep into a mixed state which has some aspects of being awake and some aspects of being in a deep sleep. There appear to be at least two routes to this mixed state: 1 Difficulty leaving deep sleep (stage 4) at the end of the first sleep cycle; or 2 A sudden disturbance or disruption during the middle of deep sleep. The events include sleepwalking, night terrors and confused partial arousals (some enuretic events can also occur as partial arousals). Although the specific types of arousal (sleepwalking versus night terror) are sometimes considered as separate entities, these behaviors represent a spectrum of related phenomena with respect to sleep physiology. The intense events (with screaming, agitated flailing and running) represent the extreme end of a spectrum of partial arousal behaviors that occur in mild forms (such as calm mumbling or a few awkward movements) in many children. The events can last from a few seconds to 20 min, with an average duration of about 3 min. The termination is usually as sudden as the initiation, with a rapid return to deep sleep. During the events, the children may seem confused, often not recognizing their parents, being inconsolable and often appearing incoherent. Overtiredness from any source (whether sleep deprivation, sleep disruption or erratic schedule) can increase or precipitate partial arousals. Any time a child is adjusting to getting less sleep, or has disturbed night-time sleep, the physiological compensation is to get deeper sleep (especially in the first 1–2 hours after sleep onset). This deep “recovery” sleep appears to be fertile ground for partial arousal events. A second theme of associated features is in the realm of psychological and emotional factors. Particularly with respect to night terrors, much has been written about the association with particular psychological states, such as anxiety, trauma, stressful events and repressed aggression (Ferber, 1989; Klackenberg, 1982). These psychological factors may contribute to sleep loss. For example, emotional and behavioral problems are also associated with difficulty falling asleep, as occurs with depression and anxiety in children (Dahl & Puig-Antich, 1990; Ryan, PuigAntich, Ambrosini et al., 1987). Likewise, disruptive disorders, such as ADHD and conduct disorder, can be accompanied by oppositional behaviors around bedtime, which may delay CHAPTER 54 900 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 900

sleep onset, as well as occasional difficulties falling asleep (Dahl & Puig-Antich, 1990). When a parasomnia is suspected, the clinical interview should include obtaining a careful history of all sleep-related habits, as well as the characteristics, pattern and frequency of the events. The pattern of events should also be assessed. Occurrence in the first third of the night and an increased frequency corresponding to periods of being overly tired are strongly suggestive of typical partial arousal events. Given the wide range of intensity and clinical significance of these nocturnal partial arousals, the key to therapy is matching the appropriate intervention to the degree of problem. Mild sleepwalking or an occasional night terror in a young child requires only some parental reassurance with general suggestions regarding improved sleep habits and the likelihood that the events will decrease over time. In contrast, frequent, repetitive, intense and agitated arousals in an older child may require very aggressive treatments, because of the risk of self-injury during the events and the possibility of associated pathology. There are three broad principles that guide intervention. First, educate the family about the events (and what to do during the actual partial arousal event). For mild to moderate events, the parents should try to direct the child to go back to bed and back to sleep. Physically taking the child by the hand and leading him or her back to bed during a mild event can also be effective. Usually, the event needs to take its course and will end spontaneously. During more agitated arousals, interventions trying to direct the child back to bed can result in increased arousal and can inadvertently prolong the event. In general, if mild directing of the child does not work, the parents should let the episode run its course. One very important caveat to this advice is with respect to the need to prevent self-injury. Eliminating factors such as sleeping on the top bunk bed, having a bedroom near the top of the stairs or having windows or dangerous objects in the room are major considerations. In severe cases, in which partial arousals are frequent and not responding to treatment, it may be necessary to install window guards or Plexiglas. Second, encourage a regular sleep–wake schedule with good sleep habits. Parents should consider an earlier bedtime if there is any evidence that the child is getting inadequate sleep. Specific causes of delayed bedtime or difficulty falling asleep should also be directly addressed. It is important to try to improve the overall quantity and/or quality of sleep of the child when applicable. Third, help the child feel safe, secure and relaxed at bedtime and to identify and express sources of anxiety and fear in a supportive environment. If there is evidence that anxiety and unexpressed anger may contribute to the frequency of these partial arousal events, the family should be encouraged to facilitate their child expressing sources of anxieties, fears, anger and conflicts in healthy ways while awake. Age-appropriate suggestions of positive family interactions within this realm can be very helpful. Also, helping children focus on positive images and positive relaxation exercises can help to foster feelings of safety and security at bedtime. Tricyclic antidepressants and benzodiazepines have been shown to have positive effects on partial arousal events. Both medications decrease arousals from sleep, and lighten the deepest stage 4 sleep. Difficulties with pharmacological interventions include tolerance effects, withdrawal effects and questions concerning possible long-term detrimental effects on sleep. Sleep-Disordered Breathing It is important for child and adolescent psychiatrists to understand how sleep apnea and a related set of problems called sleep-disordered breathing can contribute to fragmented sleep (and possibly intermittent hypoxia; Gozal & Kheirandish, 2006; Kheirandish & Gozal, 2006), which can contribute to daytime difficulties with cognitive and affective functioning. In particular, difficulties with irritability, attentional difficulties and emotional lability can be created or exacerbated by sleep-disordered breathing in children. During sleep, and particularly in REM sleep, there is a considerable drop in muscle tone. This decreased tone affects the musculature maintaining the airway and the muscles assisting in respiration. In susceptible individuals, these physiological changes can lead to obstructive sleep apnea syndrome (OSAS). In adults, the clinical picture of OSAS is typically an obese hypersomnolent lethargic individual. In children, the clinical appearance is quite different. The most common cause of OSAS in children is hypertrophy of adenoids and tonsils. Many of these children have little difficulty breathing when awake; however, with decreased muscle tone during sleep, the airway becomes smaller, airway resistance increases and the work of breathing increases. Brief arousals from sleep increase the muscle tone to the neck and pharyngeal muscles, open the airway and allow the child to resume breathing. Although the actual number of minutes of arousal during the night may be small, the repeated chronic but brief disruptions in sleep can lead to significant daytime symptoms in children. It is important to note that the child is usually unaware of waking up, and the parent often describes very restless sleep but usually does not describe the child’s waking up completely. The most frequent symptoms reported by families include loud chronic snoring (or noisy breathing), restless sleep with unusual sleeping positions (attempts by the child to move and open the airway), a history of problems with tonsils, adenoids and/or ear infections, and signs of inadequate nighttime sleep. There is still a great deal of uncertainty and controversy about the exact clinical threshold for performing formal sleep studies. Treatment decisions and options are also complex clinical decisions. In medical centers with pediatric sleep facilities, consultation with a child sleep specialist can be extremely helpful in making decisions in these cases. Some recent studies provide excellent reviews of these complex issues (Ferber, 1996; Marcus, 1997). A similar (and even more controversial) situation exists regarding restless legs syndrome (RLS) and periodic limb movement disorder (PLMD) in children. Periodic limb movement SLEEP DISORDERS 901 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 901

in sleep (PLMS) is characterized by periodic episodes of repetitive and highly stereotypic limb movements during sleep (American Academy of Sleep Medicine, 2005). PLMD is defined by the presence of periodic limb movement during sleep associated with symptoms of insomnia or excessive daytime sleepiness (Chesson, Wise, Davila et al., 1999). RLS is a clinical diagnosis characterized by disagreeable leg sensations that usually occur prior to sleep onset. PLMD and RLS are distinct entities but can coexist. Most patients with PLMD do not manifest RLS symptoms; however, approximately 80% of patients with RLS have PLMS (Montplaisir, Boucher, Poirier et al., 1997). PLMD has been described in children with somewhat different clinical presentations. Children with PLMD may present with non-specific symptoms such as growing pains, restless sleep and hyperactivity (Picchietti & Walters, 1999). These symptoms are most often unnoticed by their parents, although a family history of RLS and PLMD is common. In a series of studies, Picchietti, England, Walters, Willis, and Verrico (1998) suggested an increased prevalence of PLMD among children with ADHD. Other studies have suggested that the sleep fragmentation secondary to these disorders is an important contribution to ADHD and that, particularly in the context of symptoms and/or a positive family history of RLS/PLMS, sleep studies should be performed if there is any question that sleep fragmentation is contributing to daytime impairments with attentional control (Cortese, Konofal, Lecendreus et al., 2005; Hoban & Chervin, 2005). Treatment The guideline from the Standard of Practice Committee of the American Academy of Sleep Medicine states that no specific recommendation can be made regarding treatment of children with RLS or PLMD (Littner, Kushida, Wise et al., 2005). There is limited information on the dopaminergic medications in children and other medications have not been adequately studied in children. There is some evidence that children with PLMD may have low iron storage as evidenced by low serum ferritin and iron. Children with low serum ferritin (<50 μg/L) have been reported to respond favorably to iron therapy (Simakajornboon, Gozal, Vlasic et al., 2003). However, currently, there are no long-term data on the use of iron treatment. These children also appear to benefit from avoiding caffeine and behavioral interventions to improve sleep habits. Narcolepsy Narcolepsy is a chronic neurological disorder characterized by excessive daytime sleepiness. The classic tetrad of symptoms in narcolepsy comprises: 1 Sleep attacks; 2 Cataplexy (sudden loss of muscle tone without change of consciousness); 3 Sleep paralysis (inability to move after waking up); and 4 Hypnogogic hallucinations (dream-like imagery before falling asleep). These symptoms do not all occur together or consistently in many cases of narcolepsy. Particularly in younger patients, signs of sleepiness may be the only initial symptom. Cataplexy is typically provoked by laughter, anger or sudden emotional changes. It may be as subtle as a slight weakness in the legs, or as dramatic as a patient falling to the floor limp and unable to move. If cataplectic attacks last long enough, full sleep can occur. It is not a rare disorder, affecting approximately 1 in 10,000 people in the USA. Narcolepsy is a neurological disorder that appears to be caused by an abnormality in the hypocretin–orexin system – often by a loss of hypocretin neurons in the lateral hypothalamus and usually low cerebrospinal fluid (CSF) levels of hypocretin. About 10% of narcoleptics are members of familial clusters; however, genetic factors alone are apparently insufficient to cause the disease. A mutation on chromosome 6 affecting a human leukocyte antigen ([HLA] DR2/DQ6 [DQB1*0602]) is seen in 90– 100% of patients; however, this also occurs in as many as 50% of people without narcolepsy. Hence, obtaining a family history of narcolepsy or excessive sleepiness can be helpful, although it is negative in many cases. Traditionally, the onset of narcolepsy is thought to occur in late adolescence and adulthood, but there are well-documented cases that begin in early childhood. The diagnosis of narcolepsy requires evaluation in a sleep laboratory. Patients with narcolepsy show early REM periods near sleep onset, fragmented night-time sleep, excessive daytime sleepiness in objective nap studies during the day and sleep-onset REM periods in naps. In prepubertal children, this diagnosis can be very difficult to establish (Kotagal, Hartse, & Walsh, 1990). Repeat studies may be necessary before reaching a final diagnosis. At this time, neither HLA typing nor CSF hypocretin levels are routinely indicated in the clinical evaluation of narcolepsy; however, in unusual cases CSF hypocretins can be helpful. Treatment of narcolepsy is generally focused on: 1 Education and counseling of the patient and family; 2 Adherence to a regular schedule to obtain optimal sleep with good sleep habits (often including scheduled naps); 3 Use of short-acting stimulant medication for treatment of daytime sleepiness (with drug holidays to avoid build-up of tolerance); and 4 Use of REM-suppressant medications (such as protriptyline) when symptoms of cataplexy are problematic. The differential diagnosis of narcolepsy includes consideration of the following two sleep disorders. Idiopathic Hypersomnolence Some patients have significantly increased sleep needs without evidence of the REM abnormalities seen in narcolepsy. This condition has been called idiopathic hypersomnolence. There is often a familial history of excessive sleep needs, and these individuals show clear objective sleepiness in nap studies despite having obtained what appears to be adequate amounts of night-time sleep. These disorders are also frequently CHAPTER 54 902 9781405145497_4_054.qxd 29/03/2008 02:56 PM Page 902

treated with stimulant medication when the diagnosis is definitively established and daytime functioning is impaired. Kleine–Levin Syndrome Symptoms of excessive somnolence, hypersexuality and compulsive overeating were first described in adolescent boys by Kleine (1925) and Levin (1929). Mental disturbances (irritability, confusion and occasional auditory or visual hallucinations) have also been reported in these cases. This syndrome (with more than 100 published cases) occurs more frequently in males (3:1). Typically, symptoms start during adolescence, either gradually or abruptly, and in about half of cases the onset follows a flu-like illness or injury with loss of consciousness. Frequently, there is an episodic nature to the symptoms, with cycles lasting from 1 to 30 days. The syndrome usually disappears spontaneously during late adolescence or early adulthood. Laboratory tests, imaging studies, EEGs and endocrine measures do not appear to be helpful in making the specific diagnosis of Kleine–Levin syndrome. It is important to rule out other organic causes of similar symptoms such as a hypothalamic tumor, localized CNS infection or vascular accident. The presence of neurological signs, evidence of increased CNS pressure, abnormalities in temperature regulation, abnormalities in water regulation or other endocrine abnormalities point to an organically based abnormality. A family history of bipolar illness or other signs suggesting an early onset bipolar illness should also be ruled out. Although stimulant medication or use of lithium carbonate has been reported to be helpful in individual cases, there is no clear consensus on treatment. Conclusions In this chapter we reviewed several important principles relevant to the assessment, diagnosis and treatment of sleep problems in children and adolescents. We have underscored the evidence for interactions between sleep and the regulation of behavior, emotion and learning and the importance of sleep disturbance to many aspects of child psychiatry (and behavioral pediatrics). We have consistently emphasized behavioral principles and behavioral approaches to these problems because we believe that there is great pragmatic value in understanding and implementing these in many domains of clinical practice. There are few risks – and potentially many benefits – from increasing and enhancing sleep in youth through cognitive–behavioral interventions. There is also a clear need for more studies to provide stronger empirical support and further insight into the benefit of these interventions. Finally, it is important to emphasize the exciting advances in research in neuroscience, genetics and development in understanding the role of sleep in the developing brain. 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906 Although attachment disorders have received increased attention in contemporary literature, there have been more reviews of these kinds of disorders published than systematic studies. In fact, although these disorders have been described for more than 50 years in the literature and have been defined by specified criteria in psychiatric nosologies for nearly 25 years, they have been studied directly only in the past decade. There is now a small but growing database, allowing for a beginning consensus on some points and clearer questions about areas of controversy. The purpose of this chapter is to review attachment disorders, especially in the context of severe deprivation, and in light of what has been learned in recent investigations. Although different or more expanded versions of attachment disorders have been proposed by some (Minnis, Marwick, Arthur, & McLaughlin, 2006; van IJzendoorn & BakermansKranenburg, 2002; Zeanah & Boris, 2000), these alternative approaches have not been well studied, and we have limited our review to the traditional conceptualization of the disorder, as defined in contemporary nosologies. Definitional Challenges The term “attachment” itself has been used variably to refer to somewhat different constructs, and this has led to confusion in the literature. Bowlby, who developed attachment theory, asserted that attachment referred to the young child’s strong disposition “to seek proximity to and contact with a specific figure and to do so in certain situations, notably when . . . frightened, tired, or ill. The disposition to behave in this way is an attribute of the child, an attribute which changes only slowly over time and which is unaffected by the situation of the moment” (Bowlby, 1982, p. 371). In addition to this description of behaviors, it is often useful to consider attachment as a relational construct. We have suggested, using Emde’s (1989) model, that functions of the parent–child attachment relationship include: 1 Providing and seeking comfort for distress; 2 Providing and experiencing warmth, empathy and nurturance; 3 Providing emotional availability and regulating emotion; and 4 Providing and seeking physical and psychological protection (Zeanah, Mammen, & Lieberman, 1993). Because the relationship is asymmetrical, the young child is the seeker and the adult caregiver or parent is the provider. A significant challenge to the definition of attachment disorders is the very ubiquity of attachment in young children. Under species-typical rearing conditions, virtually all children form selected attachments to their caregivers. In children with clinical disturbances of a variety of types, attachment formation or expression may be compromised. Thus, an important question is when the disturbed attachment is the primary problem, in which case it comprises an attachment disorder, and when attachment is merely one of a number of developmental domains that is compromised, in which case it may be an associated feature of some other disorder. An appreciation of the development of attachment under usual circumstances provides a useful foundation from which to begin to answer this question. Development of Attachment Parents’ affiliative feelings for their infants often begin prenatally. Studies have indicated that parents are willing to describe a sense of who the baby is and assert that they have a relationship with the baby even before the baby is born (Benoit, Parker, & Zeanah, 1997). However, infants are not born attached to their caregivers. In fact, two conditions appear to be necessary for infants to become attached. First, they must have a significant amount of interaction on a regular basis with the caregiver and, second, they must reach a cognitive age of about 7–9 months. Phases in the development of attachment, modified slightly from Bowlby’s (1982) original formulation, are described in Table 55.1. By the end of the first year of life, it is possible to assess the quality of the young child’s attachment to a particular caregiver. Usually, this is accomplished by means of a laboratory paradigm known as the Strange Situation Procedure (Ainsworth, Blehar, Waters, & Wall, 1978). Alternating episodes of separations and reunions between a young child and a familiar caregiver and an unfamiliar adult or stranger, the procedure yields classification of a young child’s pattern of attachment to the caregiver with whom he or she is assessed as secure, insecure/avoidant, insecure/resistant or disorganized. Although secure attachment appears to confer some protection Attachment Disorders in Relation to Deprivation Charles H. Zeanah and Anna T. Smyke 55 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 906 Rutter’s Child and Adolescent Psychiatry, 5th Edition, Edited by M. Rutter, D. V. M. Bishop D. S. Pine, S. Scott, J. Stevenson, E. Taylor and A. Thapar © 2008 Blackwell Publishing Limited. ISBN: 978-1-405-14549-7

for young children in high-risk environments, the major risks for psychopathology appear to be associated with disorganized attachments (Green & Goldwyn, 2002). Attachment Classifications and Psychopathology Infant behaviors in the presence of the parent or caregiver suggesting fearfulness, freezing or other contradictory attachment behaviors (e.g., initial approach followed by avoidance while in close contact with the caregiver) during the Strange Situation Procedure are indices of a disorganized attachment classification. Although disorganized attachment has a prevalence of 14% in low-risk samples (van IJzendoorn, Schuengel, & Bakermans-Kranenberg, 1999), it is as high as 75–80% in maltreated and institutionalized samples (Carlson, Cicchetti, & Barnett, 1989; Vorria, Papaligoura, Dunn et al., 2003; Zeanah, Smyke, Koga, & Carlson, 2005). Furthermore, disorganized attachment is associated with various disturbances in caregiving behavior, including frightening/frightened behavior, as well as antagonism, role confusion, withdrawal and contradictory cues (Lyons-Ruth, Bronfin, & Parsons, 1999; Schuengel, Bakermans-Kranenburg, & van IJzendoorn, 1999). Of the attachment classifications, disorganized attachment has the strongest links to subsequent psychopathology, including social difficulties, role-inappropriate parent–child interactive behavior, peer rejection and poor social adjustment, aggression and disruptive behavior in middle childhood, in addition to emotional problems, low self-esteem and dissociative disorders in adolescence (Green & Goldwyn, 2002). At least one group has suggested that because disorganized attachment represents failure of a stage-salient developmental task, is strongly linked to concurrent and subsequent psychopathology, is reliably identified in a laboratory procedure and is associated with “pathogenic” patterns of caregiving (e.g., frightening/frightened patterns), it should be considered a disorder of attachment (van IJzendoorn & BakermansKranenburg, 2002). There are a number of problems with this suggestion, including the high prevalence of disorganized attachment in low-risk samples, identification based only on a child’s behaviors (perhaps lasting only a few seconds) in a laboratory paradigm with no established connection to conventional signs or symptoms, an uncertain relation to impairment and the relationship specificity of the disorganized classification. That is, the child’s attachment to one caregiver may be classified disorganized and the child’s attachment to another caregiver as another attachment classification, meaning that the classification is a characteristic of the relationship rather than the child. Therefore, the prevailing consensus is that disorganized attachment is a risk factor rather than a clinical diagnostic category (for a discussion of the relationship between attachment and psychopathology see chapter 13). Most descriptions of attachment as a clinical disorder have derived from studies of the effects on social and emotional development in young children who have experienced severe deprivation, such as that associated with institutional rearing. Because of that, we turn next to a brief review of the association between institutional care and attachment disorders and consider how studies of institutionalized children informed psychiatric nosologies. Historical Considerations Institutional Care and Deprivation Maternal deprivation and the material deprivation that often accompanies it have long been recognized as important risk factors to development in humans (Bowlby, 1965; Chapin, 1915; Rutter, 1981; Spitz, 1945), primates (Suomi, Collins, Harlow, & Rupenthal, 1976) and rodents (Hofer, 2006; Zhang, Levine, ATTACHMENT DISORDERS 907 Table 55.1 Development of attachment in early childhood. Age Phase Characteristics Newborn to 2 months Limited discrimination Ability to discriminate among different caregivers is limited and preferences are not readily observable 2–8 months Discrimination with Clearly discriminates different caregivers as evident by different patterns of interacting limited preference but usually engages readily with strangers as well as familiar caregivers 8–12 months Focused attachment Begins when separation protest and stranger wariness appear. Infant becomes notably more wary of interacting with strangers and protests readily if he or she anticipates or experiences separation from a preferred caregiver (i.e., attachment figure). In typical rearing circumstances, infants attach to more than one caregiver and demonstrate a hierarchy of preferences evident when stressed, fatigued or frightened 12–20 months Secure base Independent ambulation ushers in increasing exploratory forays away from the attachment figure, alternating with returns in which proximity is sought. Different patterns of attachment reflect differences in balance between exploration and secure base behavior 20 months and beyond Goal corrected partnership Attachment relationship shifts from nearly complete dependency on the caregiver to one in which the preschooler becomes aware that others, including the caregiver, have intentions and wishes different from their own 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 907

Dent et al., 2002). For the past 50 years, children raised in institutions have been studied to assess the risks associated with this atypical rearing environment because it has been associated with deprivation. A series of small-scale descriptive studies began to raise questions about the consequences of institutionalization in the 1940s. Early studies focused on behavioral and cognitive manifestations of the effects of poor rearing environments (Goldfarb, 1945; Levy, 1947). In addition to disturbances of growth, cognitive development and language, these studies also documented that children who had been institutionalized during the first 3 years of life demonstrated consistently greater levels of problem behaviors, including feeding and sleeping difficulties, aggressive behavior and hyperactivity, and excessive attention-seeking and sociability with strangers when compared with children in foster care. A recent resurgence in studies of children adopted out of Romania has demonstrated some significant problems across a range of outcomes, including disturbed attachment (Zeanah, 2000). In spite of these problems, Romanian children who have been adopted following institutional care have demonstrated varied developmental effects from their institutional experience (Kreppner, Rutter, Beckett et al., 2007; Rutter, Beckett, Castle et al., 2007), and many children seem to have recovered fully. Tizard’s Study Tizard’s (1977) landmark study deconstructed the institutional experience by removing the material deprivation often associated with institutional care and focusing on the variable of caregiver–child relationship. She described the cognitive and socioemotional development of a group of children who experienced varying lengths of institutional care in residential nurseries in Great Britain. Caregiver to child ratios were substantially better than those found in older studies (Goldfarb, 1945; Provence & Lipton, 1962). Children were maintained in small, mixed age groups, in a stimulating environment which included books, toys and instruction during the preschool years (Tizard, 1977). Three groups of children with histories of early institutional rearing were studied at age 4 years: 24 children who were adopted between 2 and 4 years of age, 15 children who were restored to their biological families (often despite marked ambivalence on the part of the mother) and 26 children who remained institutionalized (Tizard, 1977). By age 4, this latter group had experienced an average of 50 different caregivers who had cared for the child for at least a week. Of the 26 children who remained in the residential nurseries, eight had developed a preferred attachment, eight were withdrawn and unresponsive and 10 were indiscriminate, attention-seeking and socially superficial (Tizard & Rees, 1975). The latter two groups, which had been noted in previous studies and were replicated in subsequent studies, formed the basis of reactive attachment disorder in contemporary nosologies. Attachment Disorders in Nosologies Reactive Attachment Disorder of Infancy first appeared in the DSM-III (American Psychiatric Association, 1980). Disordered social and emotional development was a hallmark of the disorder which was signaled also by the presence of failure to thrive. Inexplicably, there also was a requirement that the disorder be manifest prior to 8 months of age; that is, prior to the age at which preferred attachment ordinarily emerges (Zeanah, 1996). Subsequent versions of the DSM removed this curious requirement and made the age of onset below 5 years of age. However, in DSM-IV-TR (American Psychiatric Association, 2000), a specified etiology – the child’s signs of disturbed attachment are in response to “pathogenic care” – is included. ICD-10 (World Health Organization, 1996) criteria are generally consistent with DSM criteria. ICD-10 criteria do not specify pathogenic care, but they do caution against making the diagnosis in the absence of a history of maltreatment. All revisions in criteria through 2000 occurred in the absence of systematic research of the disorders; in fact, such research is only beginning to appear. In addition to DSM-IV-TR and ICD-10, two other sets of criteria for disorders of attachment have been published recently. The Research Diagnostic Criteria–Preschool Age (RDC-PA; Task Force on Research Diagnostic Criteria: Infancy and Preschool, 2003) and the Diagnostic Classification: 0–3, Revised criteria (Zero to Three, 2005) are quite similar, although they differ from DSMIV-TR and ICD-10 in focusing more explicitly on disturbed attachment behaviors rather than disturbed social behaviors more generally. Clinical Disorders of Attachment: The Phenotype Attachment disorders describe a constellation of aberrant attachment behaviors and other social behavioral anomalies apparent in early childhood which are believed to result from limited opportunities to form selected attachments. Historically, attachment disorders have been linked to deprivation or social neglect. The diagnosis is excluded in the presence of pervasive developmental disorders to distinguish social abnormalities associated with those disorders. Consistent with the notion of a disorder, both DSM-IV and ICD-10 require that the signs of disorder be evident across situations and relationships. Two clinical patterns of attachment disorders have been described: (i) an emotionally withdrawn/inhibited pattern, in which the child exhibits limited or absent initiation or response to social interactions with caregivers, and a variety of aberrant social behaviors, such as inhibited, hypervigilant, or highly ambivalent reactions; and (ii) an indiscriminately social/disinhibited pattern, in which the child exhibits lack of expectable selectivity in seeking comfort, support and nurturance, with lack of social reticence with unfamiliar adults, including a willingness to “go off” with strangers. Emotionally Withdrawn/Inhibited Core features of this subtype of attachment disorder include lack of social and emotional responses, an absence or near-absence CHAPTER 55 908 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 908

of attachment behaviors even in times of stress, and serious problems of emotion regulation. Most strikingly, the emotionally withdrawn subtype of attachment disorder involves a paucity of positive affect responses. It also often includes bouts of fear or irritability that are seemingly unprovoked, or at least out of proportion to the putative stimulus. Social and emotional reciprocal interactions are absent or at least severely dampened. Although few data have addressed the issue directly, cognitive delays are an expected associated feature, given that the conditions of neglect believed to lead to this pattern of attachment disorder also are associated with developmental delays. These signs have been described in young children with a history of severe maltreatment (Zeanah, Scheeringa, Boris et al., 2004) and those being raised in institutions (Smyke, Dumitrescu, & Zeanah, 2002; Zeanah, Smyke, Koga et al., 2005). Interestingly, they have been much less noted in children adopted out of institutions (Chisholm, 1998; Chisholm, Carter, Ames, & Morison, 1995; O’Connor, Bredenkamp, & Rutter, 1999; O’Connor & Rutter, 2000). Indiscriminately Social/Disinhibited Core behavioral features of disinhibited/indiscriminately social attachment disorder in young children include inappropriate approach to unfamiliar adults, a lack of wariness of strangers, failure to check back with a caregiver in unfamiliar settings, and a willingness to accompany a stranger and wander away from a familiar caregiver. Indiscriminate behavior also may be associated with lack of appropriate physical boundaries, so that children may interact with strangers intrusively and aggressively and may even seek out physical contact (O’Connor & Zeanah, 2003). In comparing three different investigative teams’ approaches to indiscriminate behavior, Zeanah, Smyke, and Dumitrescu (2002) were able to show that despite differences in definition, there was overall a substantial convergence about how this type of disorder is identified. Nevertheless, the signs of this type of attachment disorder are not particularly clearly described in DSM-IV-TR or in ICD-10, and concerns remain about the heterogeneity of what we mean by indiscriminate behavior. For example, some investigators have referred to “indiscriminately friendly” behavior (Chisholm, 1998; Chisholm, Carter, Ames et al., 1995), but others have suggested that these terms imply more than is actually known about the nature of the child’s behavior (O’Connor, 2002; O’Connor & Zeanah, 2003; Zeanah, 2000). Some children exhibit passive lack of reticence and willingness to accompany a stranger, but this may be quite distinct from children who eagerly accompany a stranger without even checking back with their putative attachment figure. Other behaviors subsumed under this description include lack of initial reticence around unfamiliar adults (e.g., sitting in the lap of an unfamiliar adult after just meeting him or her), actively choosing to approach an unfamiliar adult over an attachment figure in times of need, failing to monitor the whereabouts of the attachment figure in unfamiliar settings, displaying inappropriate social boundaries (e.g., making eye contact for too long or asking overly personal questions on initial meeting), or verbally or physically aggressive engagement of unfamiliar adults. These behaviors are not experienced as sociable or friendly by those on the receiving end, and they are not always indiscriminate either (O’Connor & Zeanah, 2003). There is much room for more precise delineation of behaviors subsumed by this term. Measurement Issues and Differential Diagnosis Attachment disorders have been assessed both continuously and categorically. Boris, Zeanah, Larrieu, Scheeringa, and Heller (1998) were the first to examine the reliability of categorical diagnostic criteria. This and subsequent categorical studies have demonstrated that attachment disorders can be reliably diagnosed in young children (Boris, Hinshaw-Fuselier, Smyke et al., 2004; Zeanah, Scheeringa, Boris et al., 2004). Studies using continuous measures of disordered attachment also have demonstrated adequate inter-rater reliability in identifying signs of both the emotionally withdrawn/inhibited and the indiscriminately social/disinhibited types of disorder (O’Connor, Bredenkamp, & Rutter, 1999; O’Connor, Marvin, Rutter et al., 2003; Smyke, Dumitrescu, & Zeanah, 2002; Zeanah, Smyke, & Dumitrescu, 2002; Zeanah, Scheeringa, Boris et al., 2004; Zeanah, Smyke, Koga et al., 2005). The two patterns have been reliably identified in extreme populations, such as severely neglected children and those being raised in institutional settings, and they appear to be extremely rare in other populations of children (Boris, Hinshaw-Fuselier, Smyke et al., 2004; Roy, Rutter, & Pickles, 2004). Regarding convergent validity, Boris, Zeanah, Larrieu et al. (1998) demonstrated more severe parent–child relationship impairments in children with attachment disorders compared to children with other clinical problems. More specifically, Zeanah, Smyke, Koga et al. (2005) showed a moderate convergence between ratings of young children’s attachment behaviors in the Strange Situation with their caregivers and caregivers’ reports of signs of emotionally withdrawn/inhibited attachment disorder in the children. Also, the same group reported convergence of caregiver reports of signs of indiscriminately social/disinhibited attachment behavior and a procedure demonstrating the child’s willingness to “go off” with a stranger (Zeanah, 2006). These preliminary findings are encouraging that there is reasonable convergence among different methods used to identify attachment disorders. Nevertheless, it must be acknowledged that there remains no gold standard method of identification at this point. Given the earlier noted ubiquity of attachment as a developmental issue, it seems useful to explore whether putative attachment disorders can be distinguished from other types of disturbance. In fact, both types of attachment disorder have been shown to be only modestly or moderately correlated with aggressive behavior problems, language delays and stereotypies ATTACHMENT DISORDERS 909 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 909

(O’Connor & Rutter, 2000; Smyke, Dumitrescu, & Zeanah, 2002). The emotionally withdrawn/inhibited type of attachment disorder shares some features with autistic spectrum disorders, including disturbances in emotion regulation, impaired or absent social and emotional reciprocity and, often, cognitive delays. However, children with reactive attachment disorder should not have a selective impairment in pretend play, repetitive preoccupations or language abnormalities other than delays. In addition, they should have a history of seriously adverse caregiving. Because both the inhibited/emotionally withdrawn subtype and cognitive delays have been associated with severe deprivation (Zeanah, Smyke, & Settles, 2006), it is reasonable to assume that the emotionally withdrawn/inhibited type also may be confused with intellectual disability. Young children with emotionally withdrawn/inhibited attachment disorder should be distinguishable from children with intellectual disability because the latter group have social and emotional behaviors that are consistent with their developmental age, whereas children with emotionally withdrawn/inhibited attachment disorder have clear evidence of deviance in their social responsiveness and regulation of emotion. Clearly, both conditions may co-occur. Disinhibited attachment may share features of attention deficit/hyperactivity disorder, particularly social impulsivity. Roy, Rutter, & Pickles (2004) found that school-age children with a history of institutional rearing had high levels of both disinhibited attachment behavior and inattention/hyperactivity, as well as a strong association between the two. In addition to lack of selectivity in relationships with caregivers, the children also showed lack of selectivity with peers, echoing the findings of Hodges and Tizard (1989) with adolescents who had formerly been indiscriminate with caregivers. Because both inattention/hyperactivity and disinhibited behavior have been reported as sequelae of institutional rearing, these findings raise questions about whether they may be part of a unitary “post-institutional” syndrome, at least in a subset of cases, or whether they are distinct disorders that occur comorbidly. Of note, in Roy, Rutter, & Pickles (2004), the combination of lack of selectivity in relationships and inattention/hyperactivity occurred only in boys, whereas there has been no gender specificity reported in other studies of disinhibited attachment. Prevalence Signs of attachment disorders and prevalence of attachment disorders are greater in more extreme populations; that is, in young children who have experienced severe deprivation (Boris, Hinshaw-Fuselier, Smyke et al., 2004; Chisholm, 1998; O’Connor, Marvin, Rutter et al., 2003; Smyke, Dumitrescu, & Zeanah, 2002; Zeanah, Scheeringa, Boris et al., 2004). Still, there is a consensus that attachment disorders are rare, even though there have been no careful epidemiological studies. In a sample of 300 children aged 2–5 years recruited from pediatric clinics in Durham, North Carolina, Egger, Erkanli, Keeler et al. (2006) found no cases of attachment disorders using DSM-IV or the RDC-PA criteria (Boris & Zeanah, 2005). Several recent studies of clinic-referred and high-risk children have assessed signs of disordered attachment using structured interviews and observational assessments. These studies have found that, even among maltreated children, few met criteria for attachment disorders, with the exception of maltreated children in foster care (Boris, Zeanah, Larrieu et al., 1998; Boris, Hinshaw-Fuselier, Smyke et al., 2004; Zeanah, Scheeringa, Boris et al., 2004). In a sample of 12- to 30-month-old children living in institutions in Bucharest, Romania, 10% met criteria for emotionally withdrawn/ inhibited attachment disorders and 24% for indiscriminate/ disinhibited attachment disorder (Zeanah, 2006). Thus, although signs of the disorder may be common in extreme populations, a minority of children seem to reach the threshold of disorder. Etiology Together with post-traumatic stress disorder (PTSD), attachment disorders are one of the few psychiatric disorders in which the etiology is specified as one of the diagnostic criteria. A requirement of DSM-IV is that the signs and symptoms of attachment disorder be brought about by “pathogenic care.” Although pathogenic care is not defined, maltreatment and multiple moves are cited as examples. Although ICD-10 is less insistent on requiring pathogenic care, the clinician is cautioned against making the diagnosis in the absence of a history of maltreatment. Many questions about the nature of the caregiving compromise necessary to produce attachment disorders remain unanswered. For example, it seems reasonable to presume that neglect is the key feature, but the question of whether physical or sexual abuse alone could lead to a similar clinical picture has not been examined carefully, in part because of the difficulty of determining the nature of maltreatment in preverbal or barely verbal children. There does seem to be some component of institutional rearing that is specifically implicated in underlying indiscriminate behavior. In Roy, Rutter, & Pickles (2004), indiscriminate behavior occurred only in children with a history of institutional rearing and not in those placed in foster care, and in O’Connor, Marvin, Rutter et al. (2003) only in those with a history of institutional rearing and not those adopted within the UK. Because these studies involved institutional experiences that ranged in quality, it appears that even in better quality institutions, children remain at risk. It is also notable that the lone study to consider age at entry into institutions found that only children placed in institutions early showed a pattern of indiscriminate behavior (Wolkind, 1974). However, anecdotal evidence suggests that even in the absence of pathogenic care, some young children display signs CHAPTER 55 910 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 910

of disinhibited/indiscriminate attachment behavior. For example, individuals with Williams syndrome have been reported to show high levels of indiscriminate behavior (Jones, Bellugi, Lai et al., 2002). Williams syndrome, a microdeletion on chromosome 7q11.23, is characterized by a tendency for affected individuals to approach persons they do not know and interact with them verbally. Parents of young children with Williams syndrome expend much effort to monitor their children’s whereabouts and teach them to refrain from approaching strangers (Plesa-Skwerer, personal communication). Compared with non-affected individuals, they are unable to distinguish threatening faces, considering them just as trustworthy as nonthreatening faces (Jones, Bellugi, Lai et al., 2002). Fetal alcohol syndrome (FAS) may be associated with indiscriminate behavior in the absence of pathogenic care. Alcohol acts as a central nervous system poison, and FAS results from prenatal exposure to high levels of alcohol. Signs of the syndrome in young children include a specific pattern of facial features as well as cognitive and socioemotional abnormalities, including children being talkative, affectionate, outgoing and overly friendly to strangers (see chapter 30; Jacobson & Jacobson, 2003). FAS has been associated both with increased risk of disorganized attachment (O’Connor, Sigman, & Brill, 1987) and with increased risk of reactive attachment disorder. Course Over Time The major impediment to understanding the course of attachment disorders is the paucity of longitudinal data available for review. Based upon preliminary results from several studies, however, it is possible to suggest that the two types of attachment disorder generally have different courses. Studies of children adopted out of institutions have not reported evidence of the emotionally withdrawn/inhibited type following adoption (Chisholm, 1998; Tizard & Hodges, 1978; O’Connor, Marvin, Rutter et al., 2003). Furthermore, the Bucharest Early Intervention Project (BEIP), the first randomized controlled trial to evaluate an intervention for attachment disorders, included baseline assessments and follow-up at 30, 42 and 54 months of age (Zeanah, Nelson, Fox et al., 2003). At baseline, quality of caregiving, defined by parental sensitivity, stimulation of development, positive regard towards child, absence of detachment, absence of flat affect and absence of intrusiveness, was inversely related to signs of emotionally withdrawn/inhibited attachment disorder, although it was unrelated to signs of indiscriminate/disinhibited attachment disorder (Zeanah, Smyke, Koga et al., 2005). Preliminary results from this study indicate that foster care leads to significant reductions in signs of attachment disorder in young children who had signs of the disorder when they were living in institutions. In the group of young children randomized to foster care following early institutional rearing, such placement effectively eliminated emotionally withdrawn/inhibited attachment disorder and reduced indiscriminately social/disinhibited attachment disorder, whereas both persisted in children who remained in institutions (Zeanah, 2006). Over a 2-year period in the BEIP, there was no significant reduction in signs of inhibited/emotionally withdrawn reactive attachment disorder for young children who remained in institutions. This latter finding is important, as it represents preliminary evidence of persistence of the disorder in children who remain in an adverse caregiving environment. In contrast, there is reasonably clear evidence demonstrating that indiscriminate behavior persists over time – sometimes even after improvements in the caregiving environment. This was demonstrated originally in the work of Tizard and colleagues, who showed that young children who had been indiscriminate with caregivers at age 4 years were more likely to have serious peer disturbances at 16 years, including superficial and even indiscriminate behavior with peers (Hodges & Tizard, 1989). Subsequent studies of children adopted into Canada and the UK from Romanian institutions have also demonstrated a tendency for indiscriminate behavior to persist even after the child forms attachments to the adopted family (Chisholm, 1998; O’Connor, Marvin, Rutter et al., 2003). In fact, of those children adopted into the UK from Romanian institutions who had high levels of indiscriminate behavior at age 6 years, more than half continued to show high levels at age 11 years (Rutter, Colvert, Kreppner et al., 2006). Furthermore, in the same randomized controlled trial of foster care described above, indiscriminate behavior was only weakly responsive to removal from institutions and placement in foster care, and persistence was noted in all children who had been reared in institutions (Zeanah, 2006). Clinical Assessment In keeping with recent trends in the assessment of early childhood psychopathology (delCarmen-Wiggins & Carter, 2004), attachment disorders are best identified through a combination of caregiver interview and observed interactions. In our view, there are no gold standard interviews nor observational paradigms of interactions between parents and children that must be included, but several useful approaches have been described. Interviews Structured interviews specifically focused on the child’s attachment behaviors and signs of disordered attachment (Boris, Hinshaw-Fuselier, Smyke et al., 2004; O’Connor, Bredenkamp, & Rutter, 1999; Smyke, Dumitrescu, & Zeanah, 2002; Zeanah, Scheeringa, Boris et al., 2004) are more likely to be useful than interviews that are less specifically focused. At least two structured psychiatric interviews, the Psychiatric Assessment Preschool Age (Egger & Angold, 2004) and the Diagnostic Infant/Preschool Structured Interview (Scheeringa, 2005) include modules on attachment disorders. These approaches have been able to characterize attachment disorders both categorically and continuously. ATTACHMENT DISORDERS 911 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 911

Observations Observations of parent and child together include procedures designed specifically to examine attachment behaviors (Boris, Hinshaw-Fuselier, Smyke et al., 2004; Marvin, Cooper, Hoffman, & Powell, 2002; O’Connor, Marvin, Rutter et al., 2003), or those in which other aspects of parent–child interaction are also assessed (Zeanah, Larrieu, Valliere, & Heller, 2000). It is generally useful to include in the observation some degree of distress that is likely to activate the child’s need for the attachment figure. Conventionally, this has involved brief separations followed by reunions, but other approaches such as a novel (i.e., scary) toy also have been used (Boris, Hinshaw-Fuselier, Smyke et al., 2004). Although it is neither necessary nor necessarily advisable to use the Strange Situation Procedure in clinical assessments, in some circumstances, it may be helpful. The Circle of Security Intervention, for example, includes review of videotaped recordings of a child’s Strange Situation behavior with the parent as a part of the intervention (Marvin, Cooper, Hoffman et al., 2002). What is important clinically is not to confuse attachment classifications derived from the Strange Situation with attachment disorder diagnoses. Although systematic observations of the child’s behavior in paradigms designed to elicit attachment behaviors are often clinically valuable, naturalistic observations of a child’s attachment behaviors also may yield important data. Interventions Given that the database on disordered attachment is so small and so recent, it is not surprising that studies of intervention efforts for reactive attachment disorder are limited. Intervention efforts to date have been guided by the principle that enhancing the caregiving environment will ameliorate signs of attachment disorder. There are case reports documenting substantial improvements in young children removed from severely neglecting environments and placed in foster care (Hinshaw-Fuselier, Boris, & Zeanah, 1999; Zeanah & Boris, 2000; Zeanah, Mammen, & Lieberman, 1993). Stovall and Dozier (2000) also reported a descriptive study indicating that attachment behaviors in a small sample of young children became organized towards their new caregivers within days to weeks of placement in foster care. Studies of children raised in institutions have also begun to appear. Smyke, Dumitrescu, & Zeanah (2002) studied signs of attachment disorder in young children being raised in a large institution for young children in Bucharest. This institution had implemented a pilot unit designed to reduce the number of caregivers responsible for each child. In fact, compared to the more standard units, which included large numbers of rotating caregivers, on the pilot unit no more than four caregivers were responsible for each child on the day and evening shifts. Caregivers on the pilot unit reported that children had significantly fewer signs of both emotionally withdrawn/ inhibited attachment disorder and indiscriminately social/ disinhibited attachment disorder. Importantly, this reduction was evident even though the ratio of caregivers to children remained at 1:12 on the pilot unit (the same as on the standard units). Studies of young children adopted out of institutions are also useful to examine because they represent such a dramatic change in caregiving environments. Two longitudinal studies of young children adopted out of Romanian institutions are particularly instructive. A longitudinal study of young children adopted into Canada from Romanian institutions used parent reports of attachment and indiscriminate behavior. They found significant increases in attachment to adoptive parents during the first several years following adoption, although indiscriminate behavior persisted in a minority of children (Chisholm, 1998; Chisholm, Carter, Ames et al., 1995). Another longitudinal study of children adopted from Romanian institutions into families in the UK assessed signs of disordered attachment from parent reports at ages 4, 6 and 11 years (O’Connor & Rutter, 2000; O’Connor, Marvin, Rutter et al., 2003; Rutter, Colvert, Kreppner et al., 2006). They reported little change in the numbers of children with high levels of indiscriminate behaviors between 4 and 6 years, but some decline by age 11 years (O’Connor & Rutter, 2000; Rutter, Colvert, Kreppner et al., 2006). To date, however, there has been no demonstration that quality of care in adoptive homes is inversely related to signs of indiscriminately social/ disinhibited attachment in children. In summary, findings from studies of children living in institutions and those adopted out of institutions suggest that signs of the indiscriminate/disinhibited type are remediated by enhanced caregiving only in some children. It is unclear whether this represents individual differences among children or partial remediation with persistence in those most severely affected initially. To date, adoption studies have not included assessments of children in the institutions prior to adoption, making it difficult to explain individual differences in response. The only contemporary intervention study that included assessments of children’s attachment prior to removal from institutions and placement in an enhanced caregiving environment is the BEIP, described earlier. Preliminary results from this study indicate that foster care leads to significant reductions in signs of attachment disorder in young children who had had signs of the disorder when they were living in institutions (Zeanah, 2006). At this stage, clinicians must be guided by data indicating that secure attachment is fostered by caregivers who are emotionally available and sensitively responsive, who know and value the child as an individual, and who place the needs of the child ahead of their own needs. Preliminary data indicate that psychoeducational approaches involving training parents to respond sensitively to their child (van den Boom, 1995) and to understand their own and their child’s attachment pattern (Hoffman, Marvin, Cooper, & Powell, 2006) have been shown to promote attachment security in the child. These interventions are reasonable starting points for developing an evidence base about intervening in disordered attachments. 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Conclusions Our understanding of attachment disorders has deepened considerably in the past decade as a result of the publication of a number of systematic studies of children with histories of severe neglect or institutional rearing. These studies have affirmed the reliable identification of both the emotionally withdrawn/inhibited and the indiscriminately social/disinhibited types of attachment disorder described in contemporary nosologies. They also have supported the link between pathogenic care and attachment disorders, and have suggested that the specific “pathogen” is any environment in which young children have limited opportunities to form selective attachments, as happens in cases of severe social neglect or in settings in which the child lacks regular and consistent contact with an emotionally involved caregiver (e.g., institutional settings). These studies have suggested that although the two types of attachment disorder arise from similar conditions of risk, they have different correlates and different courses. In particular, placement in enhanced caregiving environments seems to lead to rapid and complete elimination of signs of emotionally withdrawn/inhibited attachment disorder, but the indiscriminately social/disinhibited attachment disorder remains persistent in some children, apparently for years. However, a number of questions remain to be addressed. First, nothing is known about individual vulnerabilities to the disorder – in particular why such phenomenologically distinct syndromes arise from similar conditions of risk. Genetic polymorphisms, temperamental dispositions and differential caregiving experiences should all be examined. In addition, the relationship between the two types of disorder – if any – needs to be clarified. Second, why some children with indiscriminately social/disinhibited attachment disorder seem to respond to enhanced caregiving and others do not is unclear. Third, the specific components of enhanced caregiving that are crucial for amelioration of signs of the disorder are unclear. This is especially important given the heterogeneity of outcomes in post-adoption studies. Fourth, the putative social cognitive abnormalities that characterize indiscriminate behavior need to be delineated. It seems reasonable that such abnormalities are the most concerning aspect of the disorder from the standpoint of functional impairment. Finally, the underlying neurobiology of these disorders is largely unexplored. The links between indiscriminate behavior and Williams syndrome and FAS, each of which has begun to be explored neurobiologically, may suggest hypotheses about functional impairments to be examined in children with deprivation-induced indiscriminate behavior. 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The effect of early institutional rearing on the development of eight year old children. Journal of Child Psychology and Psychiatry, 19, 99–118. Tizard, B., & Rees, J. (1975). The effect of early institutional rearing on the behaviour problems and affectional relationships of four-year-old children. Journal of Child Psychology and Psychiatry, 16, 61–73. van den Boom, D. (1995). Do first-year intervention effects endure? Follow-up during toddlerhood of a sample of Dutch irritable infants. Child Development, 66, 1798–1816. van IJzendoorn, M. H., & Bakermans-Kranenburg, M. J. (2002). Disorganized attachment and the dysregulation of negative emotions. In B. Zuckerman, A. Lieberman, & N. Fox (Eds.), Emotion regulation and developmental health: Infancy and early childhood (pp. 159–179). Somerville, NJ: Johnson and Johnson Pediatric Institute. van IJzendoorn, M. H., Schuengel, C., & Bakermans-Kranenburg, M. J. (1999). 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Zeanah, C. H. (2000). Disturbances of attachment in young children adopted from institutions. Journal of Developmental and Behavioral Pediatrics, 21, 230–236. Zeanah, C. H. (2006, February). Bucharest Early Intervention Project: Psychiatric outcomes. Presented at the American Association for Advancement of Science Annual Meeting, St. Louis, MO. Zeanah, C. H., & Boris, N. W. (2000). Disturbances and disorders of attachment in early childhood. In C. H. Zeanah (Ed.), Handbook of infant mental health (2nd edn., pp. 353–368). New York: Guilford Press. Zeanah, C. H., Larrieu, J. A., Valliere, J., & Heller, S. S. (2000). Infant–parent relationship assessment. In C. H. Zeanah (Ed.), Handbook of infant mental health (2nd ed., pp. 222–235). New York: Guilford Press. Zeanah, C. H., Mammen, O., & Lieberman, A. (1993). Disorders of attachment. In C. H. Zeanah (Ed.), Handbook of infant mental health (pp. 332–349). New York: Guilford Press. Zeanah, C. H., Nelson, C. A., Fox, N. A., Smyke, A. T., Marshall, P., Parker, S. et al. (2003). Effects of institutionalization on brain and behavioral development: The Bucharest Early Intervention Project. Development and Psychopathology, 15, 885–907. Zeanah, C. H., Scheeringa, M. S., Boris, N. W., Heller, S. S., Smyke, A. T., & Trapani, J. (2004). Reactive attachment disorder in maltreated toddlers. Child Abuse and Neglect, 28, 877–888. Zeanah, C. H., Smyke, A. T., & Settles, L. (2006). Children in orphanages. In K. McCartney, & D. Phillips (Eds.), Blackwell handbook of early childhood development (pp. 224–254). Malden, MA: Blackwell Publishing. Zeanah, C. H., Smyke, A. T., & Dumitrescu, A. (2002). Disturbances of attachment in young children. II. Indiscriminate behavior and institutional care. Journal of the American Academy of Child and Adolescent Psychiatry, 41, 983–989. Zeanah, C. H., Smyke, A. T., Koga, S. F. M., Carlson, E., & the BEIP Core Group. (2005). Attachment in institutionalized and noninstitutionalized Romanian children. Child Development, 76, 1015– 1028. Zero to Three. (2005). Diagnostic classification: 0–3R: the Diagnostic classification of mental health and developmental disorders of infancy and early childhood, Revised edition. Washington, DC: Zero to Three Press. Zhang, L. X., Levine, S., Dent, G., Zhan, Y., Xing, G., Okimoto, D., et al. (2002). Maternal deprivation increases cell death in the infant rat brain. Developmental Brain Research, 133, 1–11. ATTACHMENT DISORDERS 915 9781405145497_4_055.qxd 29/03/2008 02:56 PM Page 915

916 Wetting and soiling are widespread, and can be distressing. A recent epidemiological study of over 13,000 English children (Avon Longitudinal Study of Parents and Children [ALSPAC]) found from parental reports that at 7 years of age 15.5% children wet the bed sometimes, 7.7% sometimes wet during the day, 6.8% had daytime soiling and only 0.8% had night-time soiling (Butler, Golding, Northstone et al., 2005a). There is an overall reduction in prevalence with age: by late adolescence 1–2% continue bedwetting (Verhulst, van der Lee, Akkerhuis et al., 1985), 1% day wet (Hellström, Hanson, Hansson, Hjälmås, & Jodal, 1990) and very few soil (Bellman, 1966). However, the picture is not one of universal improvement. Children with the severest form of wetting or soiling (at least twice a week, in line with the DSM-IV diagnostic criteria) are much less prevalent (2.6% for bedwetting at age 7, 1% for daytime wetting and 0.8% soiling; Butler, Golding, Northstone et al., 2005a), but an epidemiological survey by Yeung, Sreedhar, Sihoe, Sit, and Lau (2006) suggested that for these more severely affected individuals the prevalence profile remains flat, with enuresis tending to continue into late adolescence. There is accordingly a complex issue about the point at which these forms of incontinence should be defined as problematic. One way of answering is developmental: to define an age at which there is a discontinuity and a sudden increase in the proportion becoming dry or clean. On this basis, Verhulst, van der Lee, Akkerhuis et al. (1985) identified a rapid decrease in the prevalence of nocturnal enuresis at about age 5 for girls and 8 for boys. A definition on this basis, however, would overlook the psychological distress for families with children (especially boys) who are enuretic but younger than the age of rapid decrease. A different approach, in line with recent evidence, suggests intervention to meet the child’s needs. Both Yeung, Sreedhar, Sihoe et al. (2006) and Butler, Golding, Northstone et al. (2005a) suggest that those wetting less frequently than the DSM-IV definition of twice a week, now termed “infrequent wetters,” will fairly quickly achieve dryness, and so are inappropriate for evidence-based treatment interventions, and are best supported in terms of management (e.g., regular drinking, regular toileting). However, children meeting the DSMIV definition (wet at least twice a week) appear likely to continue wetting throughout childhood and need to be identified early and treated appropriately. This chapter follows the DSM-IV approach of definitions based on both chronological age and frequency of occurrence, as set out in the following sections; but with the option of recognizing that treatment may be appropriate for some children who are distressed or impaired by the symptom even if they do not meet all the other criteria. Children with wetting and soiling are usually seen in primary care or in community-based clinics, with more complex problems referred into pediatric or child mental health services. Wetting and soiling become of particular interest to clinical psychology and psychiatry in the context of comorbidity, family dynamics and lack of response to treatment intervention. Coexistence of the problems is common; in the ALSPAC study, the small proportion (2.6%) with severe bedwetting (more than twice per week) also had a substantially increased rate of severe day wetting and a few also showed daytime soiling (Butler, Golding, Northstone et al., 2005a). Behavioral problems, including attention deficit/hyperactivity disorder (ADHD), are often identified as coexisting with nocturnal enuresis (von Gontard, Pluck, Berner, & Lehmkuhl, 1999). Urinary tract infection (UTI) can provoke overactivity of the detrusor muscles leading to painful voiding, urgency and day wetting (Hellström, Hanson, Hansson et al., 1990). An accumulation of fecal mass in constipation can compress the bladder, leading to uninhibited bladder contractions and day wetting (Kodman-Jones, Hawkins, & Schulman, 2001). Girls present a variable epidemiological profile (Verhulst, van der Lee, Akkerhuis et al., 1985) with a higher likelihood of secondary wetting (a return of wetting after being dry for at least 6 months). Secondary enuresis has repeatedly been found to be associated with a high incidence of distressing events, including parental separation and disharmony (Jarvelin, Moilanen, Vikevainen-Tervonen, & Huttunen, 1990; von Gontard, Hollmann, Eiberg et al., 1997). A minority of parents may hold the belief that wetting or soiling is under the child’s control, leading to intolerance, and such reactions seem antagonistic to some of the treatments available. An awareness of the heterogeneity of each problem can prove beneficial, both in terms of understanding the psychological ramifications and deciding on the apposite intervention. Nocturnal enuresis is divisible into monosymptomatic or polysymptomatic forms (based on the absence or presence of Wetting and Soiling 56 Richard J. Butler 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 916 Rutter’s Child and Adolescent Psychiatry, 5th Edition, Edited by M. Rutter, D. V. M. Bishop D. S. Pine, S. Scott, J. Stevenson, E. Taylor and A. Thapar © 2008 Blackwell Publishing Limited. ISBN: 978-1-405-14549-7

bladder overactivity); day wetting can be considered as either urge incontinence, voiding postponement or detrusor–sphincter dyscoordination; and soiling can reflect a lack of bowel control, intentional inappropriate deposition or constipation with overflow. Clinicians enhance their effectiveness by assessing a child’s particular needs, applying evidence-based treatment interventions and drawing, where possible, on a knowledge of pretreatment predictors of outcome (Butler & Stenberg, 2001). Nocturnal Enuresis Although parental concern and child distress should undoubtedly be considered in determining clinical significance, nocturnal enuresis is defined as an involuntary voiding of urine during sleep in children over 5 years with a severity of at least twice a week for at least 3 consecutive months when not provoked by congenital or acquired defects of the central nervous system (American Psychiatric Association, 2000). The psychological associations of bedwetting have been reviewed by Butler (2001) and although the diversity of methodology hinders firm conclusions, the ALSPAC study suggests a tendency towards social avoidance, vulnerability to victimization and increased activity and impulsivity at 7 years compared with controls (Joinson, Heron, Emond, & Butler, 2007a). However, as a group, there are few differences between those with primary nocturnal enuresis (in the absence of daytime wetting) and controls in terms of emotional adjustment, self-construing (Butler, Redfern, & Holland, 1994; Joinson, Heron, Emond et al., 2007a) or behavioral presentation (Friman, Handwerk, Swearer, McGinnis, & Warzak, 1998; Hirasing, Van Leerdam, Bolk-Bennink, & Bosch, 1997). However, children with more complex presentations appear more psychologically vulnerable than controls. Emotional vulnerability and behavior problems have both been linked to secondary nocturnal enuresis (von Gontard, Hollmann, Eiberg et al., 1997) and associated daytime wetting. There are probably several developmental pathways accounting for the associations between wetting and mental disturbances. The epidemiological basis of the findings makes it clear that referral bias is not the explanation. The timing of associations is an important clue: Rutter, Yule, and Graham (1973) drew on epidemiological data and reported that children who developed secondary enuresis were more likely to have shown psychological deviance before the enuresis started. This indicated that the psychological symptoms did not wholly result from the impact of wetting; rather, they either led to the wetting or resulted from a common cause, such as stressful life events and social adversity (see Chapters 25 and 26). There may well be a further pathway from enuresis to psychological symptoms. Fergusson and Horwood (1994) reported longitudinal epidemiological data showing a predictive effect of childhood wetting on later adolescent disturbance. The effect survived allowance for potentially confounding factors, but its size was small. Clinically, it is common to find that children are upset by both the experience of wetting and the reaction of other people; specific treatment of enuresis, such as the alarm, has reduced psychological symptoms in randomized trials (Longstaffe, Moffatt, & Whalen, 2000). The higher rates of disturbance when daytime wetting is present suggest that the public nature of the problem may provoke a troublesome dilemma (Feehan, McGee, Stanton, & Silva, 1990). While most parents remain tolerant towards the wetting, a notable proportion become intolerant and punitive (Tissier, 1983), which can lead to early withdrawal from alarm-based treatment. Where parental intolerance is apparent, a package of interventions is advocated – designed to maintain contact with the family, immediately reduce hostile feelings and challenge parental attributions of controllability (Butler & McKenna, 2002). Assessment Physical examination should exclude organic causes such as neurological incontinence. Urine analysis is necessary to check for infection (UTI; Mikkelsen, 2001). A primary clinical objective is the identification of bedwetting as either monosymptomatic (MSNE) or polysymptomatic (PSNE) based on the absence or presence of bladder overactivity, respectively (Table 56.1). The three systems approach (Butler, 2004; Butler & Holland, 2000), a conceptual formulation drawing on empirical evidence, offers an explanatory model for children while aiding practitioners in selecting apposite treatment interventions. Urine volume is normally reduced and concentrated through arginine vasopressin (AVP) secretion, enabling continuous sleep throughout the night. However, children with MSNE appear to lack circadian rhythmicity of AVP leading to urine volumes that exceed bladder capacity (Nørgaard, Pedersen, & Djurhuus, 1985; Rittig, Knudsen, Nørgaard, Pedersen, & Djurhuus, 1989). Clinical identifiers of low AVP include wetting soon after going to sleep and large wet patches (Butler & Holland, 2000; Nørgaard, Rittig, & Djurhuus, 1989). Studies of overnight cystometry recordings of children with nocturnal enuresis suggest 30–32% have uninhibited bladder contractions during sleep with small functional bladder capacities at the point of wetting (Kruse, Hellström, & Hjälmås, 1999; Watanabe, Imada, Kawauchi, Koyama, & Shirakawa, 1997). Such children are regarded as having PSNE and are identified by daytime urgency, frequent daytime voiding (more than 7 times per day), low voided volumes, variability in size of the wet patch and waking during or immediately after wetting (Butler, Robinson, Holland, & Doherty-Williams, 2004a; Yeung, Chiu, & Sit, 1999). Voided volumes are assessed by inviting the child to void into a measuring jug when the bladder “feels full,” avoiding the first void of the morning (Watanabe & Kawauchi, 1995). The highest measurement provides an estimate of maximum voided volume, which is compared with expected voided volume, by employing the formula (up to 14 years), age × 30 + 30 (mL). Approximately 80% of non-enuretic children sleep through the night without waking to toilet, yet they are more likely to wake spontaneously to void compared to enuretic children (Bower, Moore, Shepherd, & Adams, 1996), leading both WETTING AND SOILING 917 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 917

children and parents to believe bedwetting is a deep sleep phenomenon. However, electroencephalography (EEG) investigations of sleep physiology suggest similar sleep architecture to that of non-enuretic children (Mikkelsen, Rapoport, Nee et al., 1980; Neveus, Stenberg, Lackgren, Tuvemo, & Hetta, 1999). Wetting episodes occur during all sleep stages in proportion to the amount of time spent in that stage (Wolfish, 1999) and occur when the bladder is filled to the equivalent of maximal voided volume (Nørgaard & Djurhuus, 1993). Further, there are no apparent differences in sleep pattern on dry compared with wet nights (Gillin, Rapoport, Mikkelsen et al., 1982). What appears problematic for bedwetting children is being unable to arouse from sleep to bladder signals (Watanabe, Imada, Kawauchi et al., 1997). Etiology The heterogeneity of nocturnal enuresis suggests the possibility of differing etiologies. Arguably, the influential effect of any etiological determinant is through increasing vulnerability towards suboptimal nocturnal AVP secretion, bladder overactivity and/or reduced arousal response to bladder signals during sleep. Genetic influences play a part. Community surveys have shown high rates of enuresis in the families of people who are slow to acquire continence of urine at night (Fergusson, Horwood, & Shannon, 1986; Jarvelin, Moilanen, Kangas et al., 1991). Familiality by itself does not imply heritability, but the rate of nocturnal enuresis in the identical twins of bedwetters is much higher than in non-identical, suggesting a genetic contribution (Bakwin, 1971); and a genetic contribution to the acquisition of night-time dryness is clear from twin studies even in 3-year-olds (Butler, Galsworthy, Rijsdijk, & Plomin, 2001a) and 4-year-olds (Hublin, Kaprio, Partinen, & Koskenvuo, 1998). Psychosocial theories of etiology are supported by findings indicating the coexistence of bedwetting with low socioeconomic status (Chiozza, Bernardinelli, Caione et al., 1998), families in overcrowded housing (Foxman, Valdez, & Brook, 1986), unemployment of father (Devlin, 1991), large family size (Hanafin, 1998) and, interestingly, the absence of breastfeeding (Kalo & Bella, 1996; Liu, Sun, Uchiyama, Li, & Okawa, 2000). A well-conducted study with matched controls drawn from an unselected population (Jarvelin, Moilanen, Vikevainen-Tervonen et al., 1990) found that disruptive experiences (particularly splitting of the family through separation or divorce) during a sensitive stage in the development of bladder control (around 2–3 years) were related to the later development of nocturnal enuresis. Such disruptions may contribute to other potentially adverse experiences, including poorer living conditions, emotional turmoil and adaptation to new family dynamics and structures. The consequent anxiety could interrupt either the acquisition of co-ordinated muscular responses, leading to bladder overactivity, or the inhibition of AVP secretion (Houts, 1991). Management Facilitating an understanding of the three systems, often through pictures, enables an awareness of cause and the direction of treatment and emphasizes the importance of not attaching blame. Many parents adopt strategies for helping their child, with lifting and fluid restriction (Haque, Ellerstein, Gundy et al., 1981; Butler, 1987) being the most frequently reported. However, both may inadvertently prolong bedwetting. Lifting (toileting at night, usually without waking the child) in effect encourages children to empty the bladder while asleep, while fluid restriction may precipitate bladder overactivity. Good clinical practice encourages regular daytime drinking and invites CHAPTER 56 918 Table 56.1 Types of nocturnal enuresis. Definition Proportion of the enuretic population Assessment: signs Comorbidity Treatment Key references Butler & Holland (2000) Neveus (2001) Nijman et al. (2002) Butler et al. (2006) Nørgaard et al. (1989) Yeung et al. (1999) Butler et al. (2004a) Joinson et al. (2007a) Butler & Holland (2000) Polysymptomatic (PSNE) Bedwetting precipitated by increased frequency and amplitude of unstable detrusor contractions. Detrusor dependent Approx 1/3 Daytime urgency Frequent day time toileting (>7/day) Low voided volumes Variable wet patch Wake after wetting Day time wetting Fecal soiling Bladder training Anticholinergic medication Monosymptomatic (MSNE) Normal, complete, coordinated void occurring when bladder filling has reached its capacity. Diuresis dependent Approx 2/3 Large wet patches Wets soon after sleep Dilute urine on wetting Normal voided volumes Few related problems Enuresis alarm Desmopressin 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 918

the child to test what fluid intake (both volume and type) is appropriate before bedtime. The only parental strategy that appears effective is the adoption of regular daytime toileting (Butler, Golding, Heron et al., 2005b), which encourages development of control over the bladder. Effective Treatment Treatments for nocturnal enuresis are broadly categorized as either psychological, which typically aim for complete cessation, or pharmacological, which usually aim for wetting reduction. The three systems approach (Butler & Holland, 2000) encourages consideration of the child’s needs, with desmopressin or the enuresis alarm being appropriate for MSNE, and bladder training, usually combined with anticholinergic medication, being applicable for PSNE (Table 56.1). Desmopressin Desmopressin, a synthetic analog of vasopressin, is considerably more resistant to metabolic degradation than natural AVP. The mode of action is antidiuretic, with decreased urine production and increased urine concentration (Rushton, Belman, Skoog, Zaontz, & Sihelnik, 1995). An excellent review of randomized controlled trials found 70% response rate with 24.5% complete cessation of bedwetting while taking desmopressin (Moffatt, Harlos, Kirshen, & Burd, 1993). When employed specifically with MSNE, higher response rates are reported (Caione, Arena, Biraghi et al., 1997; Devitt, Holland, Butler et al., 1999). With the correct dose, dry nights are realized immediately. Thus, where a rapid response is desired (e.g., sleepover, parental intolerance), desmopressin is preferable to the enuresis alarm. Because desmopressin has no detectable effect on endogenous AVP release (van Kerrebroeck, 2002), relapse is often evident with removal of medication. Thus, long-term treatment is often advocated. Pretreatment predictors of success include older children, less severe nocturnal enuresis and no bladder overactivity (Butler & Stenberg, 2001). There are nasal spray, tablet and melt formulations with doses of 20–40 μg, 200–400 μg and 120–240 μg, respectively, taken just before bedtime with a break after 3 months to check if further treatment is indicated (Rittig, Knudsen, Nørgaard et al., 1989). Very few side effects, even with long-term treatment, are reported (Hjälmås, Hanson, Hellström, Kruse, & Sillen, 1998; Miller, Goldberg, & Atkin, 1989) provided the child does not drink excessively before bedtime or throughout the night. However, there are rare reported cases of hyponatremia although of 11 cases reported, 6 were caused by excess fluid intake (Robson, Leung, & Bloom, 1996). Enuresis Alarm The enuresis alarm is considered the optimal intervention for MSNE (Mellon & McGrath, 2000). The essential principle is to alert and sensitize the child to respond quickly and appropriately to full bladder signals, converting the signal from one of urination to that of inhibition of urination and waking (Butler, 1994). A key to success is not stimulus intensity of alarm triggering, but the child’s preparedness to wake and respond to the signal (Wolfish, 1999, 2001). Reviews of controlled studies with undifferentiated samples indicate 65–75% success with duration of 5–12 weeks and relapse of 30–45% in the 6 months following treatment (Butler & Gasson, 2005; Forsythe & Butler, 1989). Compared with desmopressin, rate of response with alarm treatment is slower but relapse less likely (Houts, Berman, & Abramson, 1994; Monda & Husmann, 1995). The expanding literature pertaining to pretreatment predictors of outcome should enhance clinical efficacy. Failure is associated with multiple wetting at night, associated daytime wetting, lack of motivation, family distress and concomitant behavioral or psychiatric problems (Devlin & O’Cathain, 1990; Moffatt & Cheang, 1995). Early withdrawal has repeatedly been found where there is parental intolerance (Butler, Brewin, & Forsythe, 1988) and relapse tends to occur with multiple wetting, associated daytime wetting and secondary enuresis. With regards to within-treatment variables, failure is associated with lack of waking to alarm triggering (Butler & Robinson, 2002). The mode of action remains open to conjecture, particularly as the majority of children who become dry sleep through the night, although theoretically it is assumed they learn to arouse to full bladder sensations (Butler & Robinson, 2002). Possible explanations include avoidance conditioning whereby alarm triggering is avoided by inhibition of micturition, increased functional bladder capacity or increased production of AVP in response to waking to the alarm (Butler & Stenberg, 2001). Bladder Training Bladder training aims to promote voluntary control over voiding, generating a change from unstable to stable bladder functioning at night. Fundamentally, children are encouraged to drink regularly (6–7 times per day), void at predetermined times, deal with urgency by voiding immediately if the sensations persist and keep regular checks of voided volumes. An uncontrolled clinical trial of children with PSNE found 68% response (>50% reduction in bedwetting) after 1 month of bladder training (Kruse, Hellström, & Hjälmås, 1999). Anticholinergic Medication Oxybutynin is a smooth muscle relaxant specifically targeting the detrusor muscles, both reducing overactivity and increasing functional bladder capacity (Kosar, Arikan, & Dincel, 1998). With unselected samples, oxybutynin has proved ineffective (Lovering, Tallett, & McKendry, 1988), yet in selected groups of children with PSNE, in uncontrolled case studies, success rates of 67–90% are reported (Kosar, Arikan, & Dincel, 1998; Watanabe, Kawauchi, Kiramori, & Azuma, 1994). Potential side effects include dry mouth, constipation and flushing. Relapse Prevention Although there are no published reports on relapse prevention with anticholinergic medication, clinical practice suggests removal is effective once the voided volume is age appropriate. Traditionally, overlearning, which involves increased WETTING AND SOILING 919 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 919

drinking in the hour before bed, has been advocated with the enuresis alarm although there are both theoretical (regarding the mechanism of action) and clinical concerns (children being dispirited by emergence of wetting). With desmopressin, gradual reduction of dosage is advocated, but reduction is prolonged (up to 3 years) and emphasizes medication as the effective agent, and at best only 50% of children remain dry (Miller, Goldberg, & Atkin, 1989). In becoming dry, children tend to attribute success to treatment (alarm or medication) so, in effect, externalize success. Relapse may arise through removal of what the child believes to be the effective agent. Recently, Butler, Holland, and Robinson (2001b) have reported a program specifically designed to prevent relapse after removal of medication, with 75% success. The program focuses on engaging the child in a process of internalizing success and highlighting the effective process by which this is accomplished, whether it is increased arousability or improved AVP release. Daytime Wetting According to DSM-IV, daytime wetting is considered an involuntary voiding of urine into clothing, with a severity of at least twice a week, in children over 5 years when not provoked by congenital or acquired defects of the central nervous system. The age criterion is not based on secure evidence, and Robson, Leung, & Bloom (1996) contended that daytime wetting is problematic at 4 years. Assessment All children deserve a careful history, pediatric physical examination, urinalysis to detect UTI and abdominal examination to rule out fecal impaction. Children with a difficulty initiating voiding, interruptions of the stream or constant dribbling require urological investigation. Von Gontard (1998) suggested non-invasive techniques should include a 24 h diary of fluid intake and urinary output; ultrasound to detect structural CHAPTER 56 920 Table 56.2 Types of daytime wetting. Other terms Definition Proportion Gender Impact Presentation Phase Assessment: signs Comorbidity Treatment ADHD, attention deficit/hyperactivity disorder, PSNE, polysymptomatic nocturnal enuresis; UTI, urinary tract infection. Urge incontinence Bladder–detrusor instability Wetting accompanied by sudden, unexpected urge symptoms Most common 82% girls; 74% boys More common in girls Emotional disturbance Wetting of small volumes Worse in the afternoon Filling phase Sudden urge to void Frequent micturition (>7/day) Holding maneuvers Small voided volumes UTI PSNE Cognitive–behavioral with anticholinergic medication Voiding postponement Wetting following postponement of micturition with urinary retention More common in boys Disruptive behavior Wetting with large volumes Normal urodynamics Full bladder Infrequent urination (<5/day) Holding maneuvers Fidgeting Stool retention Soiling Behavioral problems ADHD Cognitive–behavioral Non-directive prompts Bladder awareness Detrusor–sphincter dyscoordination Hinman’s syndrome Dysfunctional voiding with contraction instead of relaxation of the external urethral sphincter during micturition Rare More common in girls Voiding only possible with straining Prolonged micturition time Flow rate partially or completely interrupted Voiding phase Normal micturition frequency Straining on voiding Staccato voiding Residual urine Constipation Soiling Biofeedback Key reference van Gool & de Jonge (1989) Hellström et al. (1990) von Gontard et al. (1998) van Gool & de Jonge (1989) von Gontard (1998) von Gontard et al. (1998) 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 920

anomalies and residual urine; and uroflowmetry to identify detrusor–sphincter dyscoordination, although some clinicians rely on listening to the stream. Table 56.2 outlines the three main forms of daytime wetting. Urge Incontinence Urge incontinence results from sudden unexpected detrusor contractions. When access to a toilet is restricted children are unable to hold and countering the urge to void with pelvic floor contraction may lead to small voided volumes, inappropriate postponement of defecation and constipation. Wetting tends to occur when playing outdoors, shopping or walking home from school (van Gool & de Jonge, 1989). Voiding Postponement Voiding postponement is common in preschool children (Robson, Leung, & Bloom, 1996) although there are no reliable epidemiological estimates. During absorbing activities (play, television, computers), children ignore (or fail to register) a need to void, with bladder pressure building until the detrusor contracts and wetting occurs. Dysfunctional Voiding Several patterns are apparent, with overactivity of the pelvic floor muscles during micturition being a common denominator. Detrusor–sphincter dyscoordination is the most common. Although rare, because of potentially severe medical consequences, diagnosis and specific treatment are vital. Epidemiological data are lacking, although von Gontard (1998) suggested relatively high rates in psychiatric settings. Less common forms of day wetting include the following: Giggle Micturition This is sudden involuntary complete bladder emptying, usually provoked by laughter, tickling or excitement. Of the few cases reported, 70% are girls. It may resolve in adolescence but can persist into adulthood. There are no evidence-based treatments but Schmitt (1982) advocated stream interruption exercises. Stress Incontinence Involuntary loss of urine when, in the absence of detrusor contractions, intravesical pressure exceeds the maximum urethral pressure (Nørgaard, van Gool, Hjälmås, Djurhuus, & Hellström, 1998). Voiding may be provoked through coughing and exertion such as running, jumping and gymnastics. Resistive Wetting Schmitt (1982) suggests that some children become resistive about using the toilet and deliberately wet themselves. Usually occurring with boys, with a history of pressurized or punitive toilet training, there is often non-retentive soiling. There is a need to reduce tension within the parent–child relationship, foster praise for appropriate toileting and encourage the child to take positive responsibility for bladder functioning. Etiology From inconclusive evidence, von Gontard, Schaumburg, Hollman, Eiberg, and Rittig (2001) suggested an autosomal mode of inheritance in urge incontinence; environmental and psychological determinants with voiding postponement, whereas dysfunctional voiding was considered to have a genetic predisposition in the form of a delayed development of sphincter– detrusor coordination. Management Although lacking empirical evidence, many strategies continue to be adopted by clinicians. These include explanation of bladder function; regular daytime fluid intake; keeping records of voiding; sitting on the whole seat when voiding with feet supported; voiding with a relaxed pelvic floor, bending slightly forwards; completely emptying the bladder; voiding in one go, listening to the sound; developing bladder awareness through linking behaviors (e.g., fidgeting) with a desire to void; measuring voided volumes; undertaking action replay (waiting a short while after voiding and trying again, to prevent residual urine); reinforcing all actions that improve the chances of staying dry; remaining calm as a parent as overeager toileting may foster anxiety and fear over the toilet; and treating any wetting as a matter of fact and a chance for both renewed efforts and an opportunity to learn about what provoked wetting. Effective Treatment Cognitive–Behavioral Therapy For children with urge incontinence, bladder training as described for nocturnal enuresis is the preferred intervention, with the aim of establishing a sense of voluntary control over bladder functioning. Briefly, it consists of regular drinks (6–7 times per day), regular toileting (approximately 6 times per day), coupled with toileting rather than holding when urgency is experienced. Non-directive prompts to assist a child in making decisions to use the toilet regularly are preferable to direct prompts where parents are often construed as “nagging.” For children with voiding postponement, the goal is to increase frequency of daytime voiding with a watch triggering every 2 h to aid remembering. Body Worn Alarm Halliday, Meadow, and Berg (1987) reported 66% success although a non-contingent alarm schedule was equally as effective as triggering contingent on wetting, suggesting that regular toileting, rather than conditioning bladder signals, is the important factor (in contrast with nocturnal enuresis, where alarm triggering contingent with wetting is important). Biofeedback Van Gool, Vijverberg, and de Jong (1992) employed a program embracing bladder training (voiding at first sensation of urge, decreasing number of voids) with biofeedback involving visual (uroflow) and acoustic (electromyography [EMG]) readings of flow curves displayed for the child, who is encouraged to WETTING AND SOILING 921 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 921

urinate with a steady flow. They reported 60% success in disappearance of urge syndrome and voiding dysfunction. Medication There is little evidence of medication being helpful for children with daytime wetting, although good controlled trials are lacking. Anticholinergic medication (oxybutynin), with a specific action on reducing detrusor overactivity, is regularly employed for children with urge incontinence. Oxybutynin is usually prescribed at 5 mg twice a day, with slow-release preparations administered only once a day (Hjälmås, PasseriniGlazel, & Chiozza, 1992). Side effects include dry mouth, constipation, skin flushing, blurred vision and headache (Robson, Leung, & Bloom, 1996). Combination Therapy Hellerstein and Zguta (2003) used bladder training coupled with anticholinergic medication with urge incontinence and found 47% complete recovery and 42% improvement. Vijverberg, Elzinga-Plomp, Messer, van Gool, and de Jong (1997) described a 10-day in-patient program for both urge incontinence and dysfunctional voiding involving biofeedback with uroflowmetry to display good (smooth curve) or poor (interrupted curve) micturition patterns; bladder training (regular fluid intake, voiding to a regular pattern and reacting appropriately to urge signals); and goal setting (six voids per day). They reported 68% success, especially with older children. The challenge is to develop similar programs for out-patients and discover the effective components of the program. Fecal Soiling According to DSM-IV (American Psychiatric Association, 2000), this is the repeated passage of feces into inappropriate places, after the chronological or mental age of 4 years, at least once a month for 3 months without any structural abnormality or physical cause. I would prefer to reduce the age criterion to 3 years to avoid excluding such youngsters in need of treatment. In terms of the usual development, Clayden (2001) suggested the sensation to defecate occurs when rectal contents impinge on the upper part of the temporarily relaxed anal canal. With increasing rectal filling and distension, defecation is postponed by contraction of the external sphincter and pelvic floor muscles. The rectum contracts every 1–2 min and the sensation to defecate increases in intensity until the rectum is emptied. The sensation may be diminished where there is either sensory nerve loss (neuropathic rectum) or where the rectum is persistently full, especially where the fecal mass is firm (constipation) and prevents all but loose or liquidized feces passing through (overflow incontinence). The strength of the anal sphincter is likely to be affected by trauma such as abuse. Appropriate defecation is dependent on this physiological response and is assisted where there is sufficient diet (fiber and fluid) to promote movement of feces through the bowel. The child needs to attend and respond to bodily cues related to peristalsis, rather than ignore them, to discriminate between appropriate and inappropriate places for defecation, have an inclination to use the toilet and to relax and tighten the stomach muscles to eliminate feces. Thus, the physiology is aided by behavioral responses. Stein and Susser (1967) showed bowel control was achieved by 50% of children at 24 months and by almost all children (both day and night) by 2.5 years. Findings relating to impact of fecal soiling are equivocal. There is a need to clarify type of soiling in future empirical work. The clinical impression is that children with fecal soiling often live in fear of discovery, have poor peer relationships, tend to withdraw socially and avoid social events (Landman, Rappaport, Fenton, & Levine, 1986). The ALSPAC study showed that those soiling more frequently have greater levels of emotional problems (e.g., separation anxiety, social fears, general anxiety and sadness) and were more likely both to report being victimized and to bully others compared with controls (Joinson, Heron, Butler et al., 2007b). Parental ratings of behavior can be influenced by the fact of soiling (halo effects), but an association between soiling and psychological disturbance is still present when the latter is rated by teachers (Rutter, Tizard, & Whitmore, 1970). With respect to behavior, studies with strict inclusion criteria have found increased rates of behavior problems in those with fecal soiling compared with controls (Benninga, Bueller, & Heymans, 1994; Loening-Baucke, Cruikshank, & Savage, 1987). Foreman and Thambirajah (1996) reported more behavioral problems in those with secondary encopresis compared to primary encopresis. Other behavioral issues found to be associated with fecal soiling are obsessive compulsiveness and attention and activity problems (Joinson, Heron, Butler et al., 2007b). Both Landman, Rappaport, Fenton et al. (1986) and Joinson, Heron, Butler et al. (2007b) reported reduced self-esteem in children with soiling problems. Psychologically, vicious cycles are likely to develop as a result of soiling (Levine, 1982). Parents may attribute avoidance of toileting as willful or controlling, and diarrhoea-like soiling as laziness. With the resultant blame and admonishment, children experience hostile parental reactions despite being unable to feel a need to toilet. Consequently, they may hide soiled underclothes to avoid detection and humiliation. Children may fail to notice the resultant smell as the olfactory apparatus accommodates to incessant odors and thus children may have limited awareness of soiling and the consequences. It is also possible that soiling can result from pre-existent disturbance or from stresses (like those affecting wetting, see above) on the child that may be linked to psychological problems. Assessment Possible physical and organic factors need screening. The list of rare organic conditions involving soiling is long and well elucidated by Loening-Baucke (2002). Urinalysis and urine culture are necessary in the evaluation with girls because CHAPTER 56 922 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 922

of a susceptibility to UTI. Clayden (2001) suggests the more normal the consistency of the stool in the clothing, the less likely there is to be a structural abnormality of the anus or rectum. Levine (1982) favored X-ray of the lower abdomen for the detection of abundant retained feces, or a clinical examination may reveal a constipated colon. Careful history-taking is imperative to elucidate the type of soiling. Areas to cover include: 1 Precipitating factors: life events such as parental separation, birth of siblings, traumatic accidents, abuse, starting school, hospitalization; 2 History of soiling including age of onset; 3 History of toilet training; 4 Current toilet use, which defines pattern of toileting, and parental and child motivation; 5 Antecedents of soiling accidents; 6 The child’s attitude and access to toilet/s (at home and school) and reasons for avoidance; 7 Severity of soiling, eliciting the intervals, amount and consistency of bowel movements (with the aid of the Bristol Stool Chart) deposited into the toilet and clothing; 8 Dietary habits; 9 Co-occurring problems; 10 Family attitudes to the child; 11 Family dynamics and coping strategies; 12 How accidents are dealt with: parental reactions, responsibility, denial, hiding underwear; 13 The child’s understanding of the problem; and 14 Exceptions: are there times when motions and toileting are appropriate and what defines such episodes? Empirically validated subtypes of fecal soiling have yet to be established (Mellon, Houts, & Lazar, 1996) leaving classification dependent on clinical acumen. There is potential confusion arising from interchangeable terminology yet a functional classification proves valuable in differentiating treatment. Clayden (2001) provided a useful three-way clinical classification (Table 56.3) based on descriptions of soiling behavior and proposed underlying psychological mechanisms. This chapter used “encopresis” to define one pattern of secondary non-retentive soiling in which children control the physiological process of defecation but deposit normal motions in unusual places (e.g., behind sofa, in the deep freeze, parents’ WETTING AND SOILING 923 Table 56.3 Types of fecal soiling. Other terms Definition History Gender Presentation Assessment: signs Comorbidity Treatment Lack of bowel control Primary non-retentive encopresis Passage of normal stools into clothing Child is either unaware of soiling, or unable to control the bowel Never achieved bowel control Normal stools Deposited randomly in clothing No constipation May be neurological (e.g., cerebral palsy) or cognitive (intellectual disability) which impairs ability to learn bowel control Bowel training Contingent reinforcement Attention to the wider social climate Encopresis Secondary non-retentive encopresis Passage of normal stools in socially unacceptable places Bowel control established and lost Usually boys Normal stools No constipation Smearing feces Understanding the particular stresses on the child Constipation with overflow Soiling Retentive encopresis Continuous overflow of incomplete stools into clothing Toilet avoidance leading to constipation Liquid or loose stools (odorous, thin and ribbon-like) Poor appetite Functional constipation (<3 stools/week) Painful passage Infrequent bowel movement Abdominal pain Nocturnal enuresis Day wetting Stimulant laxative Stool softener Bowel training Key references Benninga & Taminiau (2001) Loening-Baucke (2002) Clayden (2001) 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 923

bed). It is hypothesized that this form of soiling is provoked by psychological events including sexual abuse (Boon & Singh, 1991), family stress and punitive reactions. Encopresis is of particular importance to mental health professionals, who may need to consider family dynamics in order to fully understand the presentation. Constipation with overflow accounts for approximately 85% of fecal soiling (Loening-Baucke, 2002). A typical pattern may involve either physical or psychological factors: 1 Avoidance or fear of the toilet, either because of painful defecation (e.g., hard feces; anal fissure) or tension around toileting (e.g., overdemanding or punitive parental reactions); 2 Stools are held back, the rectal wall is stretched affecting contractibility and sensory feedback from the bowel is reduced with a loss of feeling and diminution in the normal call to stool; 3 With increased water absorption from the fecal matter, feces become larger and harder, leading to constipation and impaction; 4 The functioning of the external and internal sphincters become compromised, allowing seepage of soft diarrhoea-like feces around the impaction; and 5 Further toilet avoidance increases constipation, leading to more concerted efforts and/or punitive reactions on the part of parents. Etiology Primary soiling has been found to be related to developmental delay, whereas secondary soiling is associated with more psychosocial adversity and behavioral problems (Foreman & Thambirajah, 1996). Clinically, fecal soiling is best understood as an interaction of physiological and psychological factors, with differing influences depending on type of soiling. Clayden (2001) outlines the main pediatric causes: • Developmental delay as a result of learning difficulty or social deprivation; • Neuropathic rectum and sensory deficiency as a result of spinal cord defects; • Abnormal internal sphincter as a result of anorectal anomaly or trauma such as abuse; • Inhibited internal sphincter as a result of rectal fecal retention – fear of pain on passing motions is a major factor in constipation (Bernard-Bonnin, Haley, Belanger, & Nadeau, 1993); • Increased motility and diminished awareness of sensation as a result of psychological distress. The “encopresis” subtype particularly can reflect psychological causes, and family interactions need special consideration. Management There are a raft of clinically useful suggestions that appear to help both children and parents understand and approach the problem in a more enlightened manner. Many clinicians now suggest avoidance of early introduction to potty training unless the child makes clear signals they are ready to try. Levine (1982) appropriately discusses the prevalence of soiling, to counter any sense of isolation. Promoting an understanding, through drawings and diagrams, of normal bowel functioning and how constipation leads to soiling is important. Children are often relieved to discover their lack of control is understood, by hearing how a distended colon becomes less sensitive, leading to a lack of a warning of the need to defecate and ultimately to seepage. Discussions with child and parent may highlight that noone is perceived as being at fault or accused of causing the problem. Parents need to refrain from attributing blame or expressing anger and frustration towards the child. Soiling episodes are best handled in a matter-of-fact way, with an older child expected to clean up and place soiled clothing in the laundry. Maintaining a positive focus is imperative with parents encouraged to praise appropriate effort and toileting (e.g., trying; passing motions in toilet). Promoting realistic expectations is important, given that treatment may be prolonged and that success is dependent on the bowel regaining muscle tone. The theme of treatment might be described as similar to a training program an athlete might use to develop better muscles and skills. It is important to enhance the appeal of the toilet at home (with décor, reading matter) and ensure access to toilets at school. Keeping a record of toilet sitting (prompted or child initiated), passage of motions, soiling accident (small stain; small formed bowel movement; full formed bowel movement) is considered useful (Houts, Mellon, & Whelan, 1988; Stark, Opipari, Donaldson et al., 1997). Effective Treatment Few prospective controlled studies have been undertaken, limiting an understanding of effective treatment interventions. McGrath, Mellon, and Murphy (2000) found only 42 well-designed studies, of which 13 were single-case designs. Often, studies failed to specify characteristics of the population, creating problems in knowing whether children had constipation with overflow or encopresis. Treatment aims and interventions evidently differ according to the type of soiling, something future research needs to address before a good clinically relevant evidence base is established. McGrath, Mellon, & Murphy (2000) provided an excellent review of treatment interventions. Although most approaches involve a package of techniques and approaches, interventions will be discussed here in relation to the specified aim. Medical Interventions Regimes for initial clear-out of impacted fecal matter and removal of pain vary across studies but usually include suppositories, enemas, laxatives, mineral oil and stool softeners. Arguably, oral methods of bowel clean-out are preferable, given that enemas may be considered psychologically unsuitable. There is a need to seek an understanding from children as to the impact of various forms of bowel clean-out. For prevention of reaccumulation of stools, long-term laxatives are likely to be required, with the dosage gradually decreased once regular bowel movements (once or twice per day) are established. On their own, enemas, laxatives and mineral oils may increase soiling initially in an effort to CHAPTER 56 924 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 924

correct constipation. The mechanism of action is assumed to relate to the debulking effect of chronically retained stools, although it is possible that the stimulation of colonic smooth muscle not only increases colonic motility itself, but also increases awareness of motility and rectal filling (Nolan, Debelle, Oberklaid, & Coffey, 1991). Medical interventions are rarely employed on their own, with Cox, Sutphen, Ling, Quillan, and Borowitz (1996) finding that only 9% were significantly helped with just a laxative. Diet Diet theoretically offers an alternative means of correcting constipation that, although slower, has a more lasting solution than laxatives. The combination of fiber and water promotes bowel movement. Increased dietary fiber includes fruit, cereals containing bran, brown rice, baked beans, vegetables, baked potato and avoidance of milk products. Houts, Mellon, & Whelan (1988) and Mellon, Houts, & Lazar (1996) outlined an appropriate fiber intake, using points linked to amount of fiber. However, there is little evidence that dietary factors alleviate functional constipation once stool withholding becomes a problem (Loening-Baucke, 2002). Mellon, Houts, & Lazar (1996) found a dietary program to improve fiber intake was no more effective and took longer to take effect than mineral oil. Behavioral Interventions These aim to increase appropriate toilet use and encourage regular bowel movement but do not address the problem of constipation. Bowel training, a customary intervention, consists of regular toilet sitting some 15 min after meals to coincide with the gastrocolic reflex which stimulates movement of feces through the bowel. As toilet sitting twice a day may be construed as tedious, the length of sitting should be a maximum of 5 min and contingent praise and rewards offered for the passage of stools. Children need to perceive the process as training not punitive. Cox, Sutphen, Ling, Quillian, and Borowitz (1998) found bowel training, coupled with medical intervention and positive reinforcement, to be 85% successful. Nolan, Debelle, Oberklaid et al. (1991) found the addition of laxative to bowel training improved success rates, although they found 12.5% failed to comply with bowel training. Other behavioral approaches, often enmeshed within bowel training, include reinforcement (tangible or star charts) contingent on behaviors the child can control (e.g., increased fiber intake, self-initiated toileting, motions in toilet); differential attention (Stark, Opipari, Donaldson et al., 1997), where soiling is treated in a neutral matter-of-fact way; overcorrection or cleanliness training, where the children are involved in washing the soiled clothing and cleaning themselves up; and awareness techniques, where children are encouraged to identify bowel sensations and behaviors as cues for toileting. Biofeedback Children with chronic constipation and encopresis are thought to have abnormal defecation dynamics with an inability to relax the external anal sphincter during attempted defecation. As this sphincter is striated muscle, theoretically amenable to modulation, biofeedback aims to enhance relaxation of the sphincter during defecation. Loening-Baucke (1990) employed a balloon, inserted in the anus at the level of internal and external sphincter, which transmitted pressure changes to an oscilloscope. Children were trained to alter their sphincter responses voluntarily using anal and buttock muscles. With reported success of 86% in relaxing the external anal sphincter and recovery from constipation in 55–60%, it was suggested that biofeedback was a promising intervention, complementary to good conventional treatment. However, being labor intensive, biofeedback may be reserved for those who have failed to respond to conventional treatment. There remains a question of clinical applicability for children and Cox, Sutphen, Ling et al. (1996) described a more convenient and accessible procedure involving electromyographic (EMG) biofeedback of the external anal sphincter. However, Loening-Baucke (1996) and Cox, Sutphen, Ling et al. (1996) reported no advantage of biofeedback over bowel training in randomized controlled studies. Further, van der Plas, Benninga, Buller et al. (1996) found that the addition of five sessions of biofeedback to the normal medical intervention plus bowel training did not improve success rates. They concluded that abnormal defecation dynamics do not have a crucial role in the pathogenesis of childhood constipation. Psychotherapeutic Interventions For children with non-retentive forms of fecal soiling, a raft of psychotherapeutic interventions have been advocated including play therapy, child psychotherapy and family therapy to reduce guilt, shame and anxiety. However, empirical investigations of such approaches are still required. Pretreatment and Treatment Predictors Treatment failure has been found to occur with children with severe constipation with overflow; when soiling happens in school or during sleep; and with associated behavioral problems, intellectual disability or psychiatric problems (Boon & Singh, 1991; Levine & Bakow, 1976; Stark, Spirito, Lewis, & Hart, 1990). There are few treatment predictors, but both lack of compliance in treatment (Levine & Bakow, 1976; Nolan, Debelle, Oberklaid et al., 1991) and lack of initial progress in the first 2 weeks (Cox, Sutphen, Ling et al., 1996) have been associated with failure. Composite Programs Treatment of fecal soiling should involve the employment of a package of interventions; accordingly, comparative studies are limited and could conceal the impact of any one component. Taking those studies with the largest samples, Nolan, Debelle, Oberklaid et al. (1991) with n = 83 found that a combination of medical intervention, diet, bowel training and contingent reinforcement produced 51% success at 12 months. Van der Plas, Benninga, Buller et al. (1996) applied a similar composite program to 94 children, with 33% success after 6 weeks, 52% WETTING AND SOILING 925 9781405145497_4_056.qxd 29/03/2008 02:56 PM Page 925

at 6 months and 59% at 1-year follow-up. Stark, Opipari, Donaldson et al. (1997), employing a similar package for 52 children with retentive encopresis, found soiling decreased by 85%, with 67% considered a success (one or fewer soils per week). On a cautionary note, McGrath, Mellon, & Murphy (2000) suggest that adding components to a treatment package does not necessarily improve outcome, and may indeed decrease efficacy. Dietary recommendations and biofeedback have both been found to reduce efficacy when attached to medical interventions and bowel training (van der Plas, Benninga, Buller et al., 1996). Relapse Prevention When relapse rates are reported, they tend to be high. Nolan, Debelle, Oberklaid et al. (1991) found 51% relapse with laxative treatment, while 37% of those treated with bowel training relapsed. Once soiling is relieved there may remain dynamics of family involvement that recreate the problem. Children with fecal soiling may therefore need continued support for a protracted time. Conclusions Nocturnal enuresis, daytime wetting and soiling often coexist, but are clinically best considered as discrete, each having heterogeneity. A differentiation between subtypes is imperative in both clinical and research contexts. The clinician is faced with an array of potential management and treatment interventions, many of which require an evidence base. However, there are proven treatments for all three problems, both medical and psychological, and with a thorough understanding of the child’s problem the clinician is now well placed to determine an apposite treatment. Pretreatment predictors are potentially useful in fine tuning treatment options although many need replication. Parsimony of treatment is important and combination programs should therefore integrate interventions to meet a child’s particular needs. Composite treatment packages are often applied in clinical practice, but require clarification; component analysis would prove beneficial in determining effective and redundant aspects. There is a need to clarify outcome measures, as has been done with nocturnal enuresis (Butler, Robinson, Holland, & Doherty-Williams, 2004b), and develop consistency across studies. Methodology tends to employ “convenience” samples and, apart from a few notable exceptions, randomization into different treatment groups is lacking. Relapse following treatment is readily apparent and yet methods to help prevent relapse are lacking – particularly in relation to daytime wetting and fecal soiling. Once children overcome wetting and soiling problems there are demonstrated improvements in psychological functioning. For example, successfully treated children with nocturnal enuresis exhibit an increase in self-esteem (Moffatt, Kato, & Pless, 1987) and develop into young adults no different psychologically from adults who were never enuretic (Stromgren & Thomson, 1990). For psychologists and psychiatrists, wetting and soiling provide interesting areas of study, because with clearly defined dependent variables, there is an ideal opportunity to test out particular theories and models. As Houts, Berman, & Abramson (1994) suggest, for most problems mental health workers confront, the therapeutic goal is management and improvement. However, wetting and soiling are exceptions – complete recovery is a realistic goal. Acknowledgments I am extremely grateful to Leeds Primary Care Trust, Dr. Philip Holland, Professor Eric Taylor and Leeds Mental Health Library for the support and assistance they kindly provided in the writing of this chapter. References American Psychiatric Association. (2000). 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930 Chronic somatic disease affects an estimated 10–20% of all children during childhood or adolescence (Abraham, Silber, & Lyon, 1999; Northam, 1997). Pediatric chronic diseases are illnesses that affect a person for an extended period of time, often for life, and that require medical care and attention above and beyond the normal requirements for a child or adolescent. Improved medical outcomes for many childhood diseases mean that a vast majority survive into adulthood, thus emphasizing psychosocial and quality of life issues. This chapter studies both the contribution of psychological factors to physical ill health and the consequences for mental health of physical illness. The approach to the latter topic is that of Wallander and Varni (1998). This differentiates between non-categorical (i.e., general) and categorical (i.e., illness-specific effects). Psychological and Psychosocial Factors in Development of Somatic Disease Evidence of Effects Children with psychosocial stress are significantly more likely to experience illness and hospitalizations as well as use health services more frequently than other children (Haavet & Gruünfeld, 1997). Stress as a precipitating factor has been implicated for asthma, appendicitis, rheumatoid arthritis and leukemia. Prospective and experimental studies have also shown that adverse life events and other stressful experiences significantly increase a person’s susceptibility to acute and recurrent respiratory infections (Cobb & Steptoe, 1998), and often precede acute exacerbations of asthma (Sandberg, Paton, Ahola et al., 2000; Sandberg, Järvenpää, Penttinen et al., 2004). Mechanisms Producing Risk The mechanisms by which psychosocial stress increases the risk of somatic disease involve cognitive, psychological, physiological and immune functions. It has been postulated that the mediators of stress response (glucocorticoids and catecholamines) have an immediate protective function (allostasis). However, when repeatedly activated they generate what has been called allostatic load, which itself creates risk of later diseases (McEwen, 2000; Turner-Cobb, 2005). For example, in the cardiovascular system, catecholamines promote adaptation by modifying heart rate and blood pressure to sleeping, waking and physical exertion. However, a concerted repetition of such blood pressure changes in response to stress can accelerate atherosclerosis and, acting jointly with metabolic hormones, can produce type 2 diabetes (McEwen, 1998). Stress may compromise the body’s immune responses to viral infection, with the individual differences in susceptibility possibly explained by differences in psychobiological reactivity (for review see Segerstrom & Miller, 2004). The immune system and the central nervous system form a bidirectional communication network (Heim, Ehlert, & Hellhammer, 2000) in which proinflammatory cytokines have a pivotal role. Role of Somatic Disease in Increasing the Risk of Psychiatric Illness Evidence of Effects Chronic or life-threatening illness increases the risk of psychiatric disorder in children and adolescents. The risk is highest in conditions affecting the central nervous system or impairing motor functioning (Eiser, 1990). A meta-analysis by Lavigne and FaierRoutman (1992) confirmed the above, with those having a chronic somatic disease showing twice the rate of psychopathology as children in the comparison groups. However, in spite of the higher prevalence, only a minority of children with chronic disorders appeared maladjusted. Few studies have addressed the specific types of mental health problems in children associated with somatic illness. Consequently, the evidence remains non-specific, although it points to anxiety and other emotional disorders being more common than behavior disorders (Thompson & Gustafson, 1996; Wallander & Varni, 1998). Even more notable is the paucity of longitudinal research to allow examination of individual differences in vulnerability and resistance. Mechanisms Producing Risk As searches for distinctive behavioral features or personality characteristics within individual illness groups have been unsuccessful, studies of psychosocial aspects of specific diseases have increasingly been replaced by the examination of various psychological and psychosocial effects associated with Psychiatric Aspects of Somatic Disease 57 Seija Sandberg and Jim Stevenson 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 930 Rutter’s Child and Adolescent Psychiatry, 5th Edition, Edited by M. Rutter, D. V. M. Bishop D. S. Pine, S. Scott, J. Stevenson, E. Taylor and A. Thapar © 2008 Blackwell Publishing Limited. ISBN: 978-1-405-14549-7

somatic disease in general (Geist, Grdisa, & Otley, 2003; LeBlanc, Goldsmith, & Patel, 2003; Lewis & Vitulano, 2003; Stewart, 2003). According to Wallander and Varni (1998), chronic physical disorders are most usefully conceptualized as sources of ongoing chronic strain for both the children and their families. Chronic strains are defined as persistent objective conditions requiring continual readjustment and repeatedly interfering with the performance of ordinary role-related activities. Their impact is determined by the nature of the onset and course of the illness, its potential fatality and the degree of incapacitation caused. Complex vulnerability and protective mechanisms are also involved. These need to be considered in relation to a child’s individual characteristics as well as to their contextual properties. In the context of a child’s chronic physical illness, family stress is negatively associated with child adjustment. However, the effect will vary depending on the presence of other mediating factors. The strength or the direction of the relationship may depend on the type of coping used and a significant association may only emerge when a parent or child copes in a maladaptive manner. Non-categorical Illness, Child- and Family-Related Factors Increasing Risk of Psychiatric Disorder Age of Child at Time of Illness It is important to view the onset of the illness in a developmental context. Early studies of hospitalization suggested that those necessitating separation from primary attachment figures are particularly difficult for children in their second to fourth year of life. Hospitalizations at an even earlier age, especially if reoccurring, have been shown to be associated with increased later behavior problems (Quinton & Rutter, 1976). However, for a school-age child, illnesses that disrupt participation in normal family and school life can become sources of stress (Vitulano, 2003). During this period gender differences also begin to emerge. For boys, illnesses that impair sports abilities are more difficult to cope with, while girls appear more sensitive to health problems that prevent accessing peer group support. In adolescence, illnesses interfering with the strive for greater independence are especially problematic, as belonging to a peer group and developing intimate relationships become critical developmental challenges (Greydanus, Rimsza, & Newhouse, 2002; Meijer, Sinnema, Bijstra et al., 2002). Accuracy and Timeliness of the Establishment of the Diagnosis The process by which the diagnosis is established can affect the child’s and parents’ ability to adapt. Here, important factors include the duration of time required to reach the diagnosis and the certainty with which it can be made, as well as the sensitivity with which the diagnosis and its implications are conveyed. A rapid and accurate diagnosis can facilitate forming a “partnership of care” and improve the prognosis. Conversely, a misdiagnosis can lead to an acutely adversarial situation. However, some illnesses are more easily recognized than others. Prodromal symptoms of some forms of leukemia, for example, can remain vague and their significance minimized for some time. In cases where malignancies are discovered at relatively advanced stages, answering the question why appropriate early investigations were not ordered is always difficult, but avoiding the issue may jeopardize subsequent treatment. Communicating a confirmed grave or terminal diagnosis to a child is a challenging responsibility (Young, Dixon-Woods, Findlay, & Heney, 2002; Young, Dixon-Woods, Windridge, & Heney, 2003). The task can become even more intricate if the physician, or parent, is ambivalent about the need to share the prognosis with the child. When faced with a grave diagnosis to share, many physicians try to cope by focusing on treatment while avoiding addressing the crucial emotional issues. An opportunity to consult a psychologist or child psychiatrist is often helpful in such situations. Interference with Function There is a strong association between the level of disability and the severity of the illness, but there also exists great variability in types and levels of disability (Morad, Kandel, Hyam, & Merrick, 2004). However, this variability in functional capacity is frequently ignored as an independent risk factor despite clear links with long-term adaptation (Vitulano, 2003). Likewise, differences in the impact of the same disability with regard to the child’s developmental level may get overlooked. Illness-related interference with cognitive processing has a negative effect at all stages of development, but cognitive deficits have even wider implications for a child facing academic examinations, for example. Impact on Physical Appearance Some somatic diseases can involve extensive physical disfigurements (e.g., atopic dermatitis), while others, although serious or incapacitating, usually have no effect on the physical appearance (e.g., diabetes or asthma). Some diseases, such as cystic fibrosis, involve complex dietary and medication regimes that older children, and especially adolescents, may perceive as socially stigmatizing. Given the importance of a positive physical appearance to the sense of self-worth, it is not surprising that stigmatizing illnesses are associated with emotional vulnerability (Breslau & Marshall, 1985). Anxiety about being physically different from their peers is a common concern of chronically ill children. This becomes progressively more important during school years and reaches its peak in adolescence. With the peer group assuming greater importance, emotional rejection based on negative response from others can precipitate depression or lead to other emotional problems. Despite the obvious importance of these issues, little systematic research has been carried out on this topic. SOMATIC DISEASE 931 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 931

Persistence of Symptoms Childhood diseases vary in their pattern of symptom expression and consequent impact on the child’s life. Asthma and epilepsy can involve long symptom-free periods and thus allow the child extended opportunities to lead a near-normal life. Diabetes and cystic fibrosis, however, demand strict daily adherence to special diets and medication routines, and in spite of these remain unremitting or result in gradual worsening and disability. Illnesses with persistent or unremitting symptoms would be expected to have greater negative impact on psychological adaptation. Surprisingly, little evidence that this is so exists. However, it is also true that some children with chronic illness cope with their situation by getting compensatory rewards from academic achievement, or by excelling in music, art or some other skill. Hope for Recovery A brief encounter with a serious illness that results in complete recovery is unlikely to have significant impact on emotional health but children with a more precarious prognosis live with persistent uncertainty about their long-term survival. The primary concern for the mental health of children with a terminal prognosis is helping them not to lose hope prematurely or succumb to depression, with a likely detrimental effect on their illness. Some children manage to maintain strong optimism about their eventual recovery even in the face of the gravest of odds. Another core aspect of work with children who have a terminal prognosis is to help them and their family to reach some level of acceptance of the inevitability of their impending death. Child’s Personality Attributes Modifying or Mediating Risk Many studies comparing children with even a severe physical illness with healthy peers have failed to find differences (Hocking & Lochman, 2005; Meijer, Sinnema, Bijstra et al., 2002; Phipps & Steele, 2002). The same studies have also established that the lack of apparent anxiety even in a gravely ill child with cancer, for example, is explainable by the child’s style of coping. Avoidant coping has been shown to predict poor adjustment characterized by low self-esteem and high social anxiety, whereas confrontation and readiness to seek social support predict positive adjustment (Meijer, Sinnema, Bijstra et al., 2002), and defensive coping is associated with low generalized anxiety (Phipps & Steele, 2002). Tendency to repress feelings, especially anger, is another commonly found characteristic of children with serious physical illness. However, there is no evidence that any particular coping style is uniformly the best protection against psychological problems. Rather, it depends on the child’s personality or temperament and social situation, including the availability of emotional support. Psychiatric Aspects of the Child’s Reactions to Somatic Disease Functional impairment may result from psychological distress or psychiatric illness presenting itself in covert forms. Geist, Grdisa, & Otley (2003) list four common presentations, found singly or in combination, often indicating underlying psychosocial issues: medical symptoms unexplainable by organic factors alone; poor adherence to treatment recommendations; school refusal; and engagement in risky behaviors involving sex/sexuality or use of substances. A change in a child’s overall functioning can alert parents and professionals to possible difficulties in the disease management, treatment adherence or adjustment to illness. However, few assessment tools specific to pediatric populations exist, and measures developed for healthy children, or modifications of adult assessments, are often used instead. The questionnaire “Living with Chronic Illness” (LCI; Adams, Streisand, Zawacki, & Joseph, 2002), represents one exception. It provides an assessment from both a parent’s and the patient’s perspective across a variety of chronic conditions and has the advantage of distinguishing between social difficulties related to the illness and those resulting from other factors. Of the therapeutic approaches, cognitive–behavioral methods and psychoeducational interventions have proved most helpful (Barlow & Ellard, 2004; Christie & Wilson, 2005). Parental Responsibility for the Treatment of the Illness Parental response to the illness can affect the child’s adaptation in many ways (Madden, Hastings, & Vant-Hoff, 2002; Wray & Sensky, 2004). Sensitive and effective parenting promotes optimal outcome, but inconsistent parenting (e.g., because of stress or depression) promotes poor adaptation. Age and developmental stage also determine the level of parental responsibility and degree of supervision of the child’s treatment. Protective parenting may be appropriate in the case of a young child, but highly attentive and controlling parental behavior can be problematic for a teenager. Their growing need for independence includes increased responsibility for their treatments – a responsibility that is also open to exploitation. Chronic illness is a stressor that can negatively impact on the parents’ mental health and marital relationship, and impair their ability to parent not only the sick child, but also the non-affected siblings. The increased risk for psychological problems in the siblings of chronically ill children has been well highlighted (Houtzager, Oort, Hoekstra-Weebers et al., 2004; Sharpe & Rossiter, 2002; Van-Riper, 2003). Interventions combining educational and psychological approaches have proved helpful in reducing family stress and promoting better mental health in parents and siblings (Barrera, Fleming, & Khan, 2004; Williams, Williams, Graff et al., 2003). Role of Non-illness-related Psychosocial Stress Children with somatic illness, like any other children, are vulnerable to the effects of psychosocial stressors, both acute life events and long-term adversities. These stresses may stem from the child’s family life (e.g., parental ill health/deviance or marital conflicts/separations), school environment (e.g., academic pressures), peer group (e.g., being a victim of bullying), or result from acute life events (e.g., accidents or loss of CHAPTER 57 932 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 932

someone close). For discussion of the mechanisms through which psychosocial stressors exert their influence see chapters 25 and 26. Protective Factors and Mechanisms Compared with the volume of research on the effects of negative experiences on children’s health, far fewer studies focusing on protective factors exist. A confiding relationship with a close adult relative has been shown to protect children from a psychiatric disorder when faced with stressful life events and chronic psychosocial adversity (Sandberg, Rutter, Giles et al., 1993). It is reasonable to believe that this would also be the case in children with somatic disease, although no empirical evidence so far exists. In children with asthma, family rituals and routines, as a proxy measure of good parenting, have been shown to protect against anxiety in the context of high parental stress (Markson & Fiese, 2000). Positive life events, such as the child’s personal achievements, and minor rebelliousness also protected against the increased risk of new asthma attacks associated with stressful life events (Sandberg, McCann, Ahola et al., 2002; Sandberg, Taskinen, Oja et al., 2003). Specific Conditions Asthma Of the chronic diseases of childhood, asthma is probably the most common, with up to one child in four being affected. Reports from different countries are consistent in indicating an increase in the prevalence of asthma and allergies, especially in industrialized societies (Asher, Montefort, Björkstén et al., 2006). Most childhood asthma begins in infancy. Adverse events in early life, allergen exposure, suboptimal feeding practices, viral infections and environmental pollutants seem important precipitating factors (Wright, Cohen, & Cohen, 2005), with gene–environment interactions determining illness manifestation (Hoffjan, Nicolae, Ostrovnaya et al., 2005). Lately, research has focused on the potential link between exposures to microbial sources and the development of allergic diseases (for review see Schaub, Lauener, & von Mutius, 2006). The term “hygiene hypothesis” refers to a theory attempting to encompass the complex associations between viral and bacterial infections, exposure to substances produced by microbes in the environment, immune responses and the person’s genetic background. A growing body of evidence suggests that psychosocial stress contributes to the development of wheezing illnesses and asthma, especially during early childhood, and predicts greater morbidity in children who already have asthma (Bloomberg & Chen, 2005). Prospective studies of genetically predisposed infants have highlighted the role of early caregiver stress. It has been shown to predict repeated wheeze in the first year and was associated with an atopic immune profile (Wright, Cohen, Carey et al., 2002; Wright, Finn, Contreras et al., 2004) and, together with early parenting difficulties, the onset of asthma by age 3 (Mrazek, Klinnert, Mrazek et al., 1999), and occurrence of asthma up to age 8 (Klinnert, Nelson, Price et al., 2001). Many clinicians also hold the view that asthma frequently gets worse following a stressful experience. That this is so was shown in a prospective study (Sandberg, Paton, Ahola et al., 2000; Sandberg, Järvenpää, Penttinen et al., 2004) involving children with chronic asthma. Severely negative life events significantly increased the risk of a new asthma attack both immediately and in the coming few weeks. The magnitude and timing of the maximum risk were influenced by the presence or absence of chronic psychosocial stress. Subsequently, a molecular genetic study by Miller and Chen (2006) showed that, in children with asthma, concurrent experience of acute and chronic stress alters the properties of genes responsible for fighting infection and keeping airways open. Emotional arousal itself can act as a potential precursor of asthmatic symptoms (for review see Lehrer, Feldman, Giardino, Song, & Schmaling, 2002). The main focus has been on negative emotions such as anger, sadness and depression (Rietveld, Everaerd, & Creer, 2000; Ritz, George, & Dahme, 2000a). A non-reciprocal pattern of autonomic functioning has been proposed as the underlying physiological mechanism (Ritz, Steptoe, DeWilde, & Costa, 2000b), with the immediate and delayed effects of stress likely to involve different processes (Klinnert, 2003). In the presence of chronic or repeated stress, the physiological response to acute stress appears to result in more sustained effects on the immune system (Chen, Hanson, Paterson et al., 2006). A less active hypothalamic–pituitary– adrenal (HPA) system in combination with high stress has been proposed as another explanation for the delayed effects (Ball, Anderson, Minto, & Halonen, 2006; Heim, Ehlert, & Hellhammer, 2000). An association has been demonstrated between asthma severity and the child’s ability to regulate negative emotions (Klinnert, McQuaid, McCormic, Adinoff, & Bryant, 2000), possibly reflecting wider physiological dysregulation indicating a common genetic vulnerability (Mrazek, 2003). These conclusions are supported by the Early Treatment of the Atopic Child study (ETAC; Stevenson, 2003) and a UK birth cohort study (Calam, Gregg, Simpson et al., 2005). Behavior problems, as possible markers for stress, preceded the development of asthma. Regarding psychiatric morbidity, emotional disorders, especially anxiety (separation anxiety being the most common) and depression, has been the most consistent finding (Feldman, Ortega, McQuaid, & Canino, 2006; Klinnert, Nelson, Price et al., 2001; McQuaid, Kopel, & Nassau, 2001). Prior emotional problems also predicted a further increase in the risk of new asthma attacks associated with stressful life events (Sandberg, Taskinen, Oja et al., 2003). Of late, experimental intervention programs have been implemented in school settings with encouraging results. In a controlled study, McCann, McWhirter, Coleman, Calvert, and Warner (2006) showed that an intervention delivered as part science curriculum combined with staff training resulted SOMATIC DISEASE 933 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 933

in significant improvements of subjective well-being and less need for prescribed medication (for a review of psychological interventions see Lehrer, Feldman, Giardino et al., 2002). Atopic Dermatitis Atopic dermatitis, commonly known as eczema, is an allergic skin condition that nearly always begins in infancy, and usually resolves during childhood, often even in the second year of life. It is a familial illness with evidence for a genetic etiology involving heritable hypersensitive immunoglobulin E (IgE) response (Ball, Anderson, Minto et al., 2006; Wright, Cohen, & Cohen, 2005). Studies in the fields of psychoimmunology have highlighted a probable connection between atopy and emotional dysregulation, possibly reflecting a common genetic vulnerability (Wright, Cohen, & Cohen, 2005). An increased rate of behavior problems in young children with atopic dermatitis (Stevenson, 2003), and a genetic association between atopy and emotional disorders in school-age children (Wamboldt, Schmidt, & Mrazek, 1998), indicate early difficulties in emotional regulation. Caring for a young child with atopic dermatitis can be a distressing task for parents as the child is often in discomfort, and therefore miserable, for long periods of time. This situation has the potential for major disruption of the early caregiving relationship. Because of a lack of systematic studies, however, little is known about the long-term psychological consequences of severe eczema in the early years of life. Cystic Fibrosis Cystic fibrosis (CF) is a complex multisystem disease with the patients often facing a number of physical and psychological problems (Bourke, 2003). It is caused by a defective gene, leading to thick sticky mucus that obstructs the airways, the digestive system and other organs, resulting in chronic infections and inflammation of the lungs and difficulty in digesting food and nutrients. CF is usually diagnosed in infancy or early childhood and prenatal screening now enables intrauterine detection. Medical treatment has led to a substantial increase in life expectancy, and more children with CF are surviving into adulthood. Despite this promising outlook, children and adolescents with CF continue to live with a life-limiting disease, with CF influencing their relationships, education and plans for future employment, marriage, etc. Progress in the medical aspects of care has not been matched by comparable advances in psychological interventions. The treatment regimens, involving strict adherence to special diets, multiple administrations of medicines and nutrition replacements, as well as “aggressive” daily physiotherapy, restrict the daily lives of the children and parents and may become a source of stress. “Successful” adherence to these regimens can also promote developmentally inappropriate dependence in the child and make negotiating the inevitable adolescent challenges problematic. Along with adolescence comes a transition to adult medical care with expectations for the patient to take responsibility for their disease management. To many adolescents with CF, moving to an adult clinic means taking a step closer to their death. Clinical problems associated with impending mortality are often challenging to manage. As the disease progresses, the young people and their families need extra support to cope with their feelings of anticipated loss and frustrations associated with the limitations of therapeutic interventions. A full range of psychiatric problems has been reported in children and adolescents with CF, with depression and eating disorders specifically noted (for review see Christian, 2003), and family interactions – especially during mealtimes – are often strained (Janicke, Mitchell, & Stark, 2005). Avoidance and denial are common psychological coping mechanisms. Such coping strategies may be viewed as “negative” or maladaptive by professionals if they include non-compliance with treatments, but from a young person’s perspective they may be adaptive, as a means of escaping from the world of CF for a while. Juvenile Onset Diabetes Mellitus Diabetes mellitus affects approximately 2% of the population in the UK and refers to a group of metabolic diseases characterized by hyperglycemia occurring as a result of defects in insulin secretion, insulin action or both. Long-term hyperglycemia is a serious problem and can lead to dysfunction of a number of organs including eyes, kidneys and the circulatory system. A distinction is made between types 1 and 2 diabetes, with the first requiring the administration of insulin to maintain health. Type 2 diabetes has usually been seen as a condition with onset in middle age. However, there is a substantial increase in type 2 diabetes amongst children and young people. This occurrence of type 2 diabetes in young people is of relatively recent origin and its clinical management is less well understood (Gungor, Hannon, Libman, Bacha, & Arslanian, 2005). The most obvious reason for the rapidly rising rate of type 2 diabetes in children and adolescents is a combination of low physical activity and the absence of a well-balanced low-fat and high-fiber diet. Action to prevent obesity in this age group is the only satisfactory way in which this alarming rise in type 2 diabetes can be controlled (Vivian, 2006). The psychological consequences of type 2 diabetes in children and young people are not well understood. There is some evidence that boys experiencing this condition are at increased risk of depressed mood compared with boys with type 1 diabetes. In turn, depressed mood is related to relatively poor metabolic control as indicated by mean glycosylated hemoglobin (HbA1c; Lawrence, Standiford, Loots et al., 2006). The care of children with type 1 diabetes is well established (Silverstein, Klingensmith, Copeland et al., 2005). It is important to recognize, however, that the care of these children needs to take into account differences in the way in which the disease needs to be managed at different ages. It has long been recognized that psychological factors can play a significant part in the course of diabetic illness (Wysochi, Buckloh, Lochrie, & Antel, 2005). Type 1 diabetes CHAPTER 57 934 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 934

is a condition that arises from the interaction of genetic and environmental risks (Haller, Atkinson, & Schatz, 2005). The significance of emotional stresses in the onset of the disease is equivocal. However, it is clear that the management and course of the disease are strongly influenced by psychosocial factors. For both children and adults, diabetes has been a major area of the application of behavioral science to medicine. The range of analyses and applications of psychobehavioral approaches to the condition has been reviewed by GonderFrederick, Cox, and Ritterband (2002). The significance of the specific association between type 1 diabetes and depression is given particular weight by the finding that suicidal ideation was a feature shortly after the onset of type 1 diabetes symptoms. Although relatively few attempted suicide, the misuse of insulin in the suicide attempts should be noted (Goldston, Kovacs, Ho, Pharrone, & Stiffler, 1994). In addition to the association between the illness and psychiatric disorder, which the above findings suggest have a reciprocal effect on each other, the presence of type 1 diabetes has an adverse effect on family functioning more broadly. In a study of children between the ages of 1 and 14 years, Northam, Anderson, Adler, Werther, and Warne (1996) found that during the immediate period after diagnosis both children and parents exhibited a mild degree of psychological distress. Within 1 year this had largely resolved and the residual impact on the family was largely seen in terms of families becoming less flexible in their functioning. These complex interactions between the course of the disease and family and individual functioning led to an emphasis on psychological interventions to support children and their families with type 1 diabetes. There is an extensive number of studies of efforts to intervene to enhance the psychological health of the children and their families (Northam, Todd, & Cameron, 2005). Many of these studies have suffered from methodological inadequacies including low participation rates, small unrepresentative samples and the use of unstandardized measures (Northam, Todd, & Cameron, 2005). They have tended to use interventions focused on increasing compliance rather than targeting a broader range of psychological distress. There have also been few attempts to evaluate interventions with younger children. Nevertheless, across the adult and child age range, the evidence suggests that interventions can be effective both in terms of enhancing the psychological well-being of individuals with type 1 diabetes and also, but to a lesser extent, promoting good metabolic control (Hampson, Skinner, Hart et al., 2000). Cancer There are a number of childhood cancers, the most common of which is acute lymphoblastic leukemia (ALL). This comprises about 25% of childhood cancers and 75% of leukemias. It is a malignant disease of the lymphoid cells and the bone marrow, which are carried in the blood to most organs including the central nervous system. Brain tumors are the second most frequently diagnosed cancer of childhood (Strother, Pollack, Fisher et al., 2002). Advances in medical treatment have substantially improved the survival rates for children with ALL to about 75%, with higher rates in Hodgkin’s disease and lower rates in other cancers (Eiser, 1998). In considering the physical and psychological consequences of childhood cancer, the following conclusion has been drawn by Patenaude and Kupst (2005, p. 11): “From these studies emerging over the past 2 decades, we have learnt with increasing specificity that no child with cancer remains unchanged by the experience.” The level of adjustment difficulties in pediatric cancer patients is near to the levels of normal healthy children (Patenaude & Kupst, 2005). However, there appear to be about 25–30% of children and families who experience considerable psychological or social difficulties. These include low academic achievement, impaired social relationships and poor self-concept and low self-esteem. It needs to be recognized that for some children and families the experience of childhood cancer has a positive effect on psychological health with increased resilience and enhanced social relationships (Eiser, Hill, & Vance, 2000). Patenaude and Kupst (2005) were able to identify a number of factors that were associated with a worse psychological outcome. Among the disease and treatment factors were diagnoses of cancer involving the central nervous system (CNS), bone tumors, CNS irradiation and intense chemotherapy, shorter time since diagnosis and a shorter period of treatment. The personal characteristics associated with a worse psychosocial outcome were younger age at diagnosis, poor psychological health before diagnosis, higher levels of perceived stress and lower cognitive ability. Those family and environment factors that have a role were low adaptability and cohesiveness, a lack of open communication, low social support and fewer socioeconomic resources. There are a number of instruments that have been developed to measure psychosocial outcome in pediatric cancer patients. An appraisal of the merits of alternative measures of health-related quality of life, including measures specifically targeted at pediatric cancer, can be found in Eiser and Morse (2001). There are still unresolved issues concerning the neurocognitive consequences of some aspects of the treatment of childhood cancers. Waber, Carpentieri, Klar et al. (2000) found that children treated for ALL with dexamethasone performed less well on cognitive tests than those on prednisone. Chemotherapy for children with ALL has also been implicated in generating attentional deficits (Buizer, de Sonneville, Van Den Heuvel-Eibrink, & Verman, 2005). However, others have suggested that treatment for leukemia that does not involve the use of cranial irradiation does not produce cognitive losses or attentional deficits (Rodgers, Marckus, Kearns, & Windebank, 2003). Indeed, Eiser, Davies, Jenney, Stride, and Glaser (2006) suggest that dexamethasone does not have an adverse effect on the health-related quality of life of children. There is some evidence that methylphenidate may help reduce any attentional and social difficulties experienced by children being treated for childhood cancer (Mulhern, Khan, Kaplan et al., 2004). SOMATIC DISEASE 935 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 935

Chronic Fatigue Syndrome The definition of chronic fatigue syndrome (CFS) in children is usually taken to be the same as for adults, and it is generally accepted that the key features are also similar (Garralda & Chalder, 2005; Garralda & Rangel, 2002). These consist of incapacitating chronic fatigue after comparatively little effort, unimproved by rest and unexplained by physical or psychiatric illness. Besides fatigue, CFS involves a characteristic constellation of physical symptoms such as headaches, a variety of other bodily pains and altered sleep and eating patterns. CFS commonly starts with an acute illness episode, such as “flu” or glandular fever, but it can also have insidious gradual onset. The condition can be intensely debilitating and follows a chronic, sometimes fluctuating, course. Children with CFS often receive repeated specialist medical consultations, and a variety of diagnoses, as the parents seek explanations for their ailments. Controversy has surrounded CFS stemming from strong beliefs held by patients and their advocates that the condition has a medical cause. For this reason, some prefer the term ME (myalgic encephalitis). It is also possible that the terms such as fibromyalgia, neurasthenia and post-viral fatigue syndrome are used to refer to the same condition. Emotional distress and mood changes are common associated psychological problems, and psychiatric comorbidity, especially anxiety and depressive disorders, as well as personality problems, are identified in 50–75% of children and adolescents severely affected with CFS (Rangel, Garralda, Levin et al., 2000b; Richards, Turk, & White, 2005), with some of this probably secondary to CFS (Garralda & Rangel, 2005). Independent genetic etiologies for depression and unexplained disabling fatigue were also indicated in a study by Fowler, Rice, Thapar, and Farmer (2006). A variety of interventions have been used in the treatment and management of CFS, with considerable differences of opinion about the most appropriate and effective management. According to a systematic review (Whiting, Bagnall, Sowden et al., 2001) of 44 controlled trials (36 of these randomized), involving 2801 mainly adult participants, the interventions with the most promising results included cognitive–behavioral therapy (CBT) and graded exercise therapy. A controlled clinical study of children and adolescents with CFS similarly demonstrated a clear superiority of active rehabilitation programs over traditional supportive care (Viner, Gregorowski, Wine et al., 2004). Outcome studies have at best involved a few dozen children and adolescents with CFS. Nevertheless, they have produced surprisingly uniform findings, showing that a distinct majority of severely affected young people eventually make a complete recovery, and many more improve sufficiently to lead near-normal lives (Rangel, Garralda, Levin et al., 2000a). Failure to thrive/deprivation dwarfism Failure to thrive (FTT) can be defined as weight for age below the third percentile for a protracted period of time during the first year of life. Children falling into this group may do so for a variety of reasons, and historically a distinction was made between those with a known organic basis for growth faltering and those deemed to be non-organic. This differentiation is far from straightforward (Reilly, Skuse, Wolke, & Stevenson, 1999). The key question is whether such a group, even if heterogeneous in the origins of their FTT, are at risk for continuing developmental difficulties. That this may not be the case is highlighted by the marked developmental catchup children can demonstrate even having experienced severe early deprivation (Rutter, 1998). The prevalence rate of FTT has been estimated to be 3.5% and it is a relatively common cause of referral to pediatricians (Skuse, 1993). Such children are likely to present with a wide array of associated medical conditions. These will reflect both factors leading to their growth deficiency and the consequences of acute or chronic malnutrition. The treatment of FTT will often require the joint expertise of a team of social workers and nutritionists as well as psychiatrists (Iwaniec, 2004). The value of a wide-ranging psychosocial intervention for children with FTT was shown in a study by Black, Dubowitz, Hutcheson, Berenson-Howard, & Starr (1995). They randomized 130 children (mean age, 12.7 months) recruited from low-income families into two groups: clinic plus home intervention or clinic only. All children received services in a multidisciplinary growth and nutrition clinic. Families in the home intervention group received weekly home visits for 1 year by lay home visitors, supervised by a community health nurse. Children’s weight for age, weight for height and height for age improved significantly during the 12-month study period, regardless of intervention status. However, the enhanced cognitive and behavioral changes in the home intervention group led Black, Dubowitz, Hutcheson et al. (1995) to conclude that early home intervention can promote a nurturant home environment effectively and can reduce the developmental delays often experienced by low-income urban infants with FTT. A meta-analysis of the extent to which FTT was associated with later poor cognitive development was produced by Corbett and Drewett (2004). They suggest that the adverse effect of FTT to later cognitive ability was –0.28 standard deviation (95% confidence interval [CI], –0.16 to –0.41), equivalent to 4.2 IQ points. This overall pattern of results is also illustrated by the findings from Boddy, Skuse, and Andrews (2000). Their longitudinal study of children who had FTT (defined as being at 3 months at least below the 3rd percentile of weight) indicated a diminishing effect of early malnutrition on cognitive ability as the children became older. This pattern of emerging results, suggesting a relative benign outcome for children with FTT during early infancy, has led Rudolf and Logan (2005) to suggest that an “aggressive approach to the identification and management of failure to thrive needs reassessing.” The key issue is that of identifying which of the children within this group might benefit from intervention. This is particularly significant given what they suggest are the equivocal results that have emerged from randomized controlled trials of intervention directed at this group as a CHAPTER 57 936 9781405145497_4_057.qxd 29/03/2008 02:57 PM Page 936