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Word-finding difficulties, verbal paraphasias, and verbal dyspraxia in ten individuals with fragile X syndrome. American Journal of Medical Genetics, 60, 39–43. Spreat, S., Conroy, J. W., & Fullerton, A. (2004). Statewide longitudinal survey of psychotropic medication use for persons with mental retardation: 1994 to 2000. American Journal of Mental Retardation, 109, 322–331. Steen, R. G., Xiong, X., Mulhern, R. K., Langston, J. W., & Wang, W. C. (1999). Subtle brain abnormalities in children with sickle cell disease: Relationship to blood hematocrit. Annals of Neurology, 45, 279–286. Stoll, C., Alembik, Y., & Dott, B. (2006). Are the recommendations on the prevention of neural tube defects working? European Journal of Medical Genetics, 49, 461–465. Szymanski, L., & King, B. (1999). 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841 The concept of personality disorder in adult life is important and elusive. It is important because the problems it encompasses are a major cause of burden to patients, to the adults and children with whom they have relationships, and to society. It is elusive because there is relatively little agreement on basic issues such as whether there is one overarching disorder or many, and what the core defining features of a personality disorder are. Nevertheless, there is broad agreement that the concept must include stability of behaviors over substantial periods of time, and extensive or pervasive dysfunction encompassing a range of psychological, behavioral and interpersonal capacities. Prima facie it seems likely that such stable and severe problems will have started before adult life, and there is evidence that in many instances analogous disorders or their antecedents start in childhood, and that childhood experiences may affect risk for subsequent personality disorder. Accordingly, it seems crucial to consider what is known about personality disorders, with special reference to two key questions for child psychiatrists and child mental health practitioners more widely: the extent to which there is continuity in personality disturbance between childhood and adult life, and on whether it is useful to apply the diagnosis in childhood and adolescence. Concepts of Personality Disorder Attempts to classify personality types and dimensions go back to antiquity but concepts of personality disorder are of much more recent origin (Frances & Widiger, 1986; Rutter, 1987; Tyrer, Casey, & Ferguson, 1991). Although there are numerous variations on the specifics, the unifying notion is the idea that there are pervasive and persistent abnormalities of overall personality functioning that cause social impairment and/or subjective distress, which are not caused by episodic disorders of mental state. The basic assumption is that, in some way, personality disorders are different from other psychiatric conditions, such as schizophrenia, depression or anxiety states. Most personality disorder research has made use of the American Psychiatric Association (2000) DSM classification which put forward four diagnostic criteria. There were first enduring patterns of inner experience and behaviours that deviated markedly from cultural expectations (specifying that they should be manifest in at least two of cognition affectivity, interpersonal functioning or impulse control). Second, this enduring pattern had to be inflexible and pervasive across a broad range of personal and social situations. Third, it had to lead to clinically significant distress or impairment. Fourth, the pattern must be stable over time with an onset by adolescence or early adult life. Several attempts to provide a unified concept have been made, notably using the idea that personality disorder represents an extreme of normal personality. One approach, based on the proposal that there are five major domains of personality – the Five Factor Model – (Widiger & Costa, 1994), argues that “Personality disorders are not qualitatively distinct from normal personality functioning. They are maladaptive, extreme variants of common personality traits” (see chapter 14). In discussions of models such as these, two issues are commonly conflated: whether categorical or dimensional approaches to personality disorder are more useful, and what is “normal personality functioning?” The categorical versus dimensional dilemma is discussed later in the chapter. The identity of the key elements of personality remains a matter of debate. In the Five Factor Model, the higher-order traits extraversion/ positive emotionality, neuroticism/negative emotionality, conscientiousness/constraint, agreeableness and openness-toexperience are proposed as providing a succinct yet comprehensive summary of personality dimensions. However, from the perspective of personality development several questions require attention. How coherent or homogenous are the hypothesized traits? Although the big five are characterized as “higher-order,” each includes “lower-order” traits, some of which differ substantially. This issue is reviewed by Caspi, Roberts, and Shiner (2005) (see chapter 14). For example, the higher-order trait neuroticism is well validated and highly predictive in childhood and adult life, but includes both proneness to anxious distress and to anger. These different emotions motivate different behaviors, they are mediated via different neurological substrates, and they have different implications for development and psychopathology (Frick & Morris, 2004; Lemerise & Arsenio, 2000). Some of the personality traits also resemble temperamental characteristics. Thus, proneness to fearfulness and to anger are two aspects of temperament commonly assessed in infancy (Robinson & Acevedo, 2001). More generally, the overlap between temperament and Disorders of Personality 50 Jonathan Hill 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 841 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

personality traits requires further attention (Caspi, Roberts, & Shiner, 2005). Big five personality traits are determined by the frequency of behaviors across context and time; however, traits can be conceptualized in other ways. In particular, coherence across context and time may occur at the level of the organization of behaviors (e.g., from the interplay between social information processing and context; Mischel, 2004). Personality development entails the development of the person as a social organism. Personality traits such as neuroticism undoubtedly influence social relationships (Robins, Caspi, & Moffitt, 2002), but there are also aspects of personality functioning such as social cognitive processes that are inherently social and entail dedicated neuronal networks (Johnson, Griffin, Csibra et al., 2005). The capacity to regulate behaviors and emotions according to the demands of different kinds of social domain (e.g., contrasting teachers and parents) is key to effective personality development. Personality develops in interaction with the environment so that its features are likely to reflect more or less successful adaptations than any simple manifestation of stable traits. An alternative view of personality disorders proposes that the key features entail limitations of social role performance. Studies of adult personality disorders consistently find that they are associated with social dysfunction (Hill, Fudge, Harrington, Pickles, & Rutter, 2000; Seivewright, Tyrer, & Johnson, 2004; Skodol, Pagano, Bender et al., 2005a) and “the characteristic that seems to define them all is a pervasive persistent abnormality in maintaining social relationships” (Rutter, 1987). Causes of social dysfunction may include extremes of personality traits, such as those identified in the big five, and also deficits or biases in social processes including mentalization, attachment and the capacity for social domain management (Hill, Pilkonis, Morse et al., 2008). While these frameworks are promising, it should be borne in mind that the concept of personality disorder did not originate from a model of normal personality, and indeed descriptions of different personality disorder types have come about through several contrasting routes. First, there are those that derive from the clinical observation that there are individuals who show enduring patterns of inflexible and seriously maladaptive behavior that seem to fall outside the traditional criteria for mental illness or psychiatric syndrome. The need to have a term to describe these patterns of abnormal behavior led to the concepts of psychopathy put forward by writers such as Hare (1970), Henderson (1939) and Cleckley (1941) (see chapter 51). Second, there are the categories of personality disorder that identify individuals who are thought to be most prone to episodic mental disorders. Schneider’s (1923, 1950) notion of psychopathic personality (defined quite differently from the psychopathy formulations of Hare, of Henderson and of Cleckley) provides the main historic notion of this approach. Many of the ICD-10 specific personality disorder categories (e.g., paranoid, anankastic and anxious) represent this tradition (World Health Organization, 1996). Third, some categories have their origins in empirical observations of the continuities between psychopathological disorders in childhood, and pervasive social malfunction in adult lifestyle. The DSM-IV (American Psychiatric Association, 2000) diagnosis of antisocial personality disorder is the obvious example of this type, with its starting point in the findings of the longterm longitudinal studies pioneered by Robins (1966, 1978). Fourth, some categories stem fairly directly from theoretical, especially psychoanalytic, notions. The concept of borderline personality organization (Kernberg, 1967, 1975) underlies specification of the DSM-IV diagnosis of borderline personality disorder (Gunderson, Kolb, & Austin, 1981). Finally, a few categories have come, at least in part, from genetic findings. Meehl (1962) coined the term schizotypy to describe the postulated characteristics of schizophrenia-prone individuals. The Danish adoption study showed that the biological families of adopted schizophrenic probands included a raised rate of personality disorders that seemed to exhibit some features reminiscent of schizophrenia (Kety, Rosenthal, & Wender, 1971). Spitzer, Endicott, and Gibbon (1979) developed more explicit criteria for this type of personality syndrome, which has come to be termed “schizotypal personality disorder.” The differences in the origins of personality disorder have contributed to substantial heterogeneity in the balance between behaviors and psychological states that are included in the subtypes of personality disorder. For instance, DSM-IV antisocial personality disorder is based primarily on antisocial behaviors and interpersonal difficulties, and includes only one state of mind item “lack of remorse” which is closely tied to behavior. By contrast, the borderline personality disorder category includes three items “identity disturbance,” “chronic feelings of emptiness” and “transient paranoid ideation” referring solely to states of mind. Validation Stability and Separation from Episodic Disorders It is assumed that the personality disorders should show substantial stability over time. However, there is surprisingly little evidence that this is the case. Several studies of referred adults have found low to moderate stability (Ferro, Klein, Schwartz, Casch, & Leader, 1998). There is increasing evidence that some personality disorders may remit over relatively short periods of time. In a follow-up every 2 years of 290 patients meeting DSM-IV criteria for borderline personality disorder, 74% had remitted after 6 years, and only 6% had remitted and then relapsed (Zanarini, Frankenburg, Hennen, Reich, & Silk, 2005). The distinction between episodic disorders and personality disorder is enshrined in the DSM distinction between Axis I and Axis II. Axis II disorders should show persistence over a period in which there is either recovery from, or onset of, Axis I disorder. It is important to note that this distinction is not made for childhood disorders. Thus, DSM oppositional defiant disorder and conduct disorder are classified on Axis I even though they show strong continuities, and probably share causal mechanisms, with both Axis I and Axis II disorders in CHAPTER 50 842 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 842

adult life. Remarkably few studies have been carried out to test the stability of personality disorder after accounting for episodic disorders in adults. Loranger, Lenzenweger, Gartner et al. (1991) examined changes in the diagnosis of personality disorder (on a standardized interview) in a series of 84 psychiatric patients over 1–6 weeks and found no effect of change in mental state on the diagnosis of personality disorder, supporting the personality disorder/episodic disorder distinction. Nevertheless, remission was associated with some fall in personality disorder symptoms and the diagnosis of personality disorder showed only moderate temporal stability (K = 0.55). Studies over greater time periods have used social dysfunction as the index of personality disorder at baseline and followup (Quinton, Gulliver, & Rutter, 1995; Rutter & Quinton, 1984) or a trait-based assessment of personality disorder at baseline and social dysfunction at follow-up (Seivewright, Tyrer, & Johnson, 2004). All reported substantial stability of personality dysfunction after accounting for duration or severity of symptoms of episodic disorders. While these findings support the distinction between episodic and personality disorders, the two are strongly associated. There are very high rates of personality disorder in patients with episodic disorders – nearly half in in-patient samples and 20– 40% in out-patient groups (Docherty, Fister, & Shea, 1986; Tyrer, Casey, & Ferguson, 1991). These figures are much higher than the rates of 6–13% that are typical of general population samples (Casey & Tyrer, 1986; Merikangas & Weissman, 1986). The meaning of these associations is not clear. It is likely that to a certain extent they are artifacts arising from difficulties in distinguishing Axis I and Axis II items. In clinical and research practice it is often difficult to disentangle symptoms associated with episodes and those that reflect personality disorder. However, this is unlikely to be the whole explanation. There are three main ways in which the associations may arise. First, personality disorder may constitute a risk factor for episodic conditions. This could occur through direct mechanisms whereby a feature of the disorder, such as poor coping (Vollrath, Alnaes, & Torgersen, 1994), leads to helplessness and hence depression or indirect mechanisms in which the personality disorder increases the likelihood of psychosocial stressors that in turn are associated with a disorder such as depression (Daley, Hamman, Davila, & Burge, 1998). Second, episodic conditions may create the conditions in which personality disorders develop because a chain of maladaptive behaviors and adverse environmental responses is set in motion that fosters more persistent psychopathology (Kasen, Cohen, Skodol, Johnson, & Brook, 1999). Third, both the personality disorder and the episodic disorder could reflect one underlying liability. Malone, Taylor, Marmorstein, McGue, and Iacono (2004) assessed antisocial personality disorder and alcohol dependence in 289 twin pairs at ages 17, 20 and 24. There were moderate genetic influences on each disorder at each age, and a substantial proportion of these influences was common to the two disorders. It might be expected that whatever the relationship between episodic disorders and personality disorder, the combination should in some sense reflect a more severe variant, with implications for treatment outcome. However, the evidence has been mixed. One recent review concluded there was no effect of personality disorder on treatment outcomes for depression (Mulder, 2002). Newton-Howes, Tyrer, and Johnson (2006) conducted a meta-analysis of all depression treatment studies in which personality disorder had been assessed and concluded that personality disorder was associated with a doubling of the risk of a poor outcome for depression (Newton-Howes, Tyrer, & Johnson, 2006). The authors point out that even the largest studies reviewed had inadequate statistical power to detect a difference of this size. Very little is known about the ways in which personality disorder may be associated with a poor outcome. Possibilities include that it may be associated with poor treatment compliance, with greater severity of overall psychopathology or with more psychosocial adversities and fewer interpersonal resources. Differentiations Among Personality Disorders The World Health Organization (1996) classification, ICD-10, makes a basic differentiation between organic personality disorders (meaning those resulting from severe head injury, encephalitis and other forms of overt brain damage); enduring personality changes deriving from some catastrophic experience (see chapter 42); personality abnormalities that reflect the residue of some mental illness (e.g., residual schizophrenia); and what they term personality disorders. The American Psychiatric Association (2000) scheme makes broadly comparable distinctions. This chapter is concerned with this last group of specific personality disorders, because the others have only an extremely limited application in childhood and adolescence. The ICD classification specifies nine personality disorder types, and the DSM system eleven. To a certain extent the different categories reflect the different origins referred to earlier. The DSM classification has received a lot of attention and, in spite of the operational definitions of each of the categories, the evidence for the discriminant validity of the majority is not strong. Consistently, studies have shown that individuals who receive one DSM Axis II diagnosis often receive several (Morey, 1988; Oldham, Skodol, Kellman et al., 1992; Pfohl, Coryell, Zimmerman, & Tsangl, 1986). Although there is evidence that the DSM categories cluster into three broad bands of the odd/eccentric, the dramatic/emotional/erratic and the anxious/ fearful (Kass, Skodol, Charles, Spitzer, & Williams, 1985), combinations of personality disorders that cross these bands are common (Oldham, Skodol, Kellman et al., 1992). Support for the discriminant validity of particular personality disorders would be provided if it were possible to show differential association with other psychiatric conditions, family clustering, or associations with genetic or environmental influences. Studies of the association of borderline personality disorder with other episodic and personality disorders have provided some support for its discriminant validity. Zanarini, Frankenburg, Dubo et al. (1998) compared patients with borderline personality disorder with patients with other persoality disorders. Those with borderline disorder had higher rates of DISORDERS OF PERSONALITY 843 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 843

associated paranoid, avoidant and dependent personality disorders, and higher rates of mood, anxiety and eating disorders. The most clear-cut finding from family studies is the association between schizophrenia and a loading of schizotypal personality disorders in the biological relatives (Kendler, Gruenberg, & Strauss, 1981, Kendler, Gruenberg, & Strauss, 1984). The evidence for a link between schizotypal personality disorder and schizophrenia is further supported by findings that there are similarities in magnetic resonance imaging appearances (Dickey, Shenton, Hirayasu et al., 2000) and in deficits of attention and information processing (Cadenhead, Light, Geyer, & Braff, 2000). A significant but weaker association with schizophrenia has been found with paranoid personality disorders (Kendler & Gruenberg, 1982; Kendler, Masterson, & Davis, 1985). There are very few family and genetic data on other personality disorders (Maier, Franke, & Hawallek, 1998; McGuffin & Thapar, 1992). There is some evidence that borderline personality disorders cluster in families and also are associated with affective disorders in other family members, but it remains quite uncertain whether the family loading has a genetic basis. Joyce, McHugh, McKenzie et al. (2006) reported a significant association between a 9-repeat allele of a dopamine transporter gene variant and borderline personality disorder in depressed patients entered into two treatment trials. This requires replication. There is consistent evidence for genetic influences on adult antisocial behaviors (Langbehn, Caderet, Yates, Troughton, & Stewart, 1998), but not adult violence (Carey & Goldman, 1997). Most studies of the association between adverse childhood experiences and personality disorders have reported associations with a wide range of episodic and personality disorders (Johnson, Cohen, Brown, Smailes, & Bernstein, 1999). It should be borne in mind that such associations can reflect genetic as well as environmental influences; nevertheless, specificity with particular disorders would support their validity. Bezirganian, Cohen, and Brook (1993) found that the combination of maternal inconsistency and high maternal overinvolvement assessed in adolescence was associated with the persistence or emergence of borderline personality disorder, and no other episodic or personality disorders. Parker, Roy, Wilhelm et al. (1999) reported that dysfunctional parenting in childhood was associated with the dramatic and anxious DSM clusters, but not the odd/eccentric cluster. Categorical versus Dimensional Characterizations of Personality Disorders Diagnostic systems treat psychopathology as a categorical entity; however, it is evident that in most, and perhaps all, instances there are no natural cut-offs demarcating disorder from absence of disorder (see chapter 2; Pickles & Angold, 2003). The majority of early studies treated personality disorders as categories; however, the advantages of considering personality disorders dimensionally are becoming increasingly apparent. Simply from a measurement perspective, better interreliablity is achieved using symptom counts. Evidence for validity tends to be stronger for dimensional methods. Personality disorder symptoms scores show greater stability than diagnoses (Skodol, Gunderson, Shea et al., 2005b) and there is a greater elevation of borderline symptoms than borderline personality disorder diagnoses in the first-degree relatives of borderline probands (Zanarini, Frankenburg, Yong et al., 2004). Furthermore, there is no evidence that there is a natural diagnostic cut-off (Daley, Burge, & Hammen, 2000). Diagnoses may also lump together symptoms that reflect different processes. Zanarini, Frankenburg, Hennen et al. (2005) have suggested that borderline personality disorder comprises acute symptoms that wax and wane and may be affected by life events, and others are chronic or “temperamental.” Such a proposal requires further investigation. However, it illustrates the advantages of a dimensional framework in relation to the personality disorders. In community studies, increasing personality disorder symptoms below a diagnostic threshold are associated with increasing social dysfunction (Daley, Burge, & Hammen, 2000; Hill, Hope, Lorenz et al., unpublished data). This is of great clinical importance because it suggests that even among patients who do not meet criteria for a diagnosis, personality disorder symptoms may be associated with processes that influence course and treatment responsiveness in common presentations such as adolescent deliberate self-harm. It is also possible that in the same individual, symptoms below the diagnostic threshold from more than one disorder may each have prognostic significance. Specific Personality Disorders In view of the lack of good discriminant validity among the many specific personality disorders, only those particularly relevant in childhood and adolescence are covered in this chapter. Antisocial personality disorder (ASPD) is well validated and important because of its association with conduct disorders in childhood, particularly those with early onset, and because of its implications for the next generation via parenting difficulties and domestic violence (Moffitt, 2006). Psychopathic disorder is identified in a smaller group of antisocial adults, most of whom have ASPD, and is associated with distinctive patterns of offending and neurobiological correlates. In childhood, there appears to be a subset of antisocial children with “callous unemotional” traits with distinctive genetic and neurobiological contributions that may be the precursors of adult psychopathic disorder (Blair, Peschardt, Budhani et al., 2006). Childhood conduct disorders and callous unemotional traits, and ASPD and psychopathic disorder are described in chapter 51. In this chapter we review borderline, schizotypal and schizoid disorders. Borderline Personality Disorder The overall concept of borderline personality disorder (BPD) is of a pervasive pattern of instability of interpersonal relationships, self-image, affects and control over impulses. The operationalization of this concept has varied somewhat over the years but the criteria have usually included a pattern of unstable and intense interpersonal relationships with alternating idealization CHAPTER 50 844 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 844

and devaluation, frantic efforts to avoid real or imagined abandonment, an unstable or distorted self-image, impulsiveness, recurrent suicidal behavior, affective instability with intense episodic dysphoria, chronic feelings of emptiness and a lack of control over anger. In contrast to ASPD, BPD is more commonly diagnosed in females. Very little is known about the childhood antecedents of BPD. Many individuals have both antisocial and borderline features, and some traits such as impulsivity are core to both disorders (Paris, 2005). It is possible that there is a common origin in early-onset disruptive behavior problems, with a gender-based divergence in adolescence or early adult life. Equally, borderline personality difficulties may emerge during early adolescence without a history of overt antisocial behavior problems. Retrospective studies suggest that, as children, adults with BPD may have had difficulties in separating from emotionally important people and show high mood reactivity (Reich & Zanarini, 2001) and that many started to self-harm before the age of 12 (Zanarini, Frankenburg, Ridolfi et al., 2006). Many theories of the origins of BPD have proposed a central role for adverse, and in particular traumatic, experiences in childhood. If this were the case there would be a particularly important preventative role for child care and mental health professionals. It is important to re-emphasize that both genetic and environmental mechanisms may be implicated in associations with childhood adversities, and genetic designs are usually required to tease them out. Moreover, in their meta-analysis covering 21 studies into possible associations between child sexual abuse and BPD, Fossati, Madeddu, and Maffei (1999) found there was only a moderate pooled effect size. It is difficult to disentangle the effects of child maltreatment from other family environmental and genetic risk factors, to identify which aspects of maltreatment may be relevant to BPD, and to determine the specific association with borderline as opposed to other conditions commonly comorbid with BPD. For example, it may be that particular kinds of child maltreatment are specific to BPD (see chapter 28). In a comparison of borderline patients and those with other personality disorders, those in the borderline group were more likely to have been abused by a male non-caretaker, to have experienced denial of their thoughts and feelings by a male caretaker and to have experienced inconsistent treatment by a female caretaker (Zanarini, Ruser, Frankenburg, Hennen, & Gunderson, 2000). We referred earlier to the particular patterns of mother-child interactions found in a general population study of adolescent predictors of borderline personality disorder (Bezirganian, Cohen, & Brook, 1993). There may also be specificities between type of maltreatment and features of BPD. For example, individuals with BPD also report more dissociative experiences (Jones, Heard, Startup et al., 1999). Zanarini, Ruser, Frankeburg et al. (2000) found that dissociative experiences among BPD patients were associated with particular experiences that included inconsistent behavior by a caretaker and sexual abuse by a caretaker. Finally, some effects of childhood maltreatment on BPD may be evident only in subgroups with genetically influenced susceptibilities to experiences (see chapter 23). Theories regarding mechanisms in BPD have focused on impulsivity (Paris, 2005), emotion dysregulation (Putnam & Silk, 2005), unresolved or insecure attachment (Levy, 2005) and failure of mentalization (Bateman & Fonagy, 2004). Some of the features of BPD, including suicide attempts, self-mutilation, substance misuse and sexual promiscuity, may reflect a more general trait of impulsivity. Impulsive spectrum disorders, such as ASPD and substance misuse, are elevated in the firstdegree relatives of borderline patients (White, Gunderson, Zanarini, & Hudson, 2002), and levels of impulsivity predict clinical outcomes in BPD (Links, Heslegrave, & van Reekum, 1999). However, disorders other than BPD are also characterized by impulsivity, notably ASPD, and although there is substantial overlap between the disorders, there are important differences. Emotion dysregulation is likely to be one of the keys to the difference between BPD and ASPD. “The inability to sustain positive affect coupled with the pervasive and unremitting distress as experienced by patients with BPD can become a veritable prison to those with the disorder and often as well to those who attempt to treat the individual with BPD” (Putnam & Silk, 2005, p. 900). Borderline individuals commonly have a low threshold for reacting to events, or to anticipated events (such as abandonment), with intense negative emotion. According to Linehan (1993), BPD arises from an interaction between temperamental emotional reactivity and an “invalidating” environment in which the child’s communication about inner emotional experience is regarded as reflecting an inaccurate perception of themselves and others. One of the major consequences of this response is thought to be that children raised in these environments do not learn to identify emotional states accurately, nor do they learn to regulate affect, as it is more often than not denied. In addition, invalidating environments also intermittently reinforce escalations of emotional displays. This results in two opposing responses to affect being shaped. The first is the inhibition or denial of emotional states, and the second is an extreme response, which often appears to be inappropriate to the level of the stimulus that elicited it. Instability of family and romantic relationships characterized by highly intense interactions, fear of abandonment and emotionally charged breakdown, are also common in individuals with BPD. Several studies have linked this to disturbances of attachment (Levy, 2005), although very few have tested whether attachment difficulties are associated specifically with BPD, as opposed to other personality disorders. Bateman and Fonagy (2004) argued that individuals with BPD have an impoverished model of their own and others’ mental function (mentalization). “Their schematic, rigid and at times extreme ideas about their own and others’ states of mind make them vulnerable to powerful emotional storms and apparently impulsive actions, and create profound problems of behavioral and affect regulation” (Fonagy & Bateman, 2004, p. 2). They also argue that these limitations in mentalization are seen particularly in relation to attachment relationships, thus leading to particular difficulties in close relationships. DISORDERS OF PERSONALITY 845 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 845

The clinical picture of BPD may arise from interacting processes. For example, the combination of intrusive and inconsistent mother–child interactions associated specifically with BPD (Bezirganian, Cohen, & Brook, 1993) may contribute to affect dysregulation and a failure of mentalization. Intrusive parenting is likely to lead to intense negative affect from which the child takes evasive action, hence leading to extremes of high and low emotional intensity, and also to a reduced capacity to monitor mental states when highly aroused. Inconsistent mother–child interactions may inhibit the child’s capacity to read the intentions or states of mind of the parent, and hence limit his or her ability to think of others’ behaviors in mental state terms. Repeated self-injury, cutting, burning or hitting, is frequently observed in individuals with severe personality disorders, and is one of the diagnostic items for BPD. It is a serious and puzzling phenomenon. There is a wide range of theories concerning the motivation for self-injury, and themes that are common to most of them are of regulation of emotions, communication and social reinforcement (Kemperman, Russ, & Shearin, 1997). Linehan (1993) has proposed that self-injury directly relieves painful emotions, and is reinforced by the attention and care given by others. Several studies have supported the role of self-injury in reducing painful affect, although most have used retrospective measures with samples that in different ways were unrepresentative. Proposals have been made that the mechanisms may entail effects on endogenous opioids or serotonin regulation, but results from a small number of investigations have been inconsistent (see chapter 40; Kemperman, Russ, & Shearin, 1997). Dissociative Identity Disorder The category multiple personality disorder (MPD), renamed dissociative identity disorder (DID) in DSM-IV, has provoked considerable controversy. The diagnosis is rarely made outside of the USA and Canada (Merskey, 1995), and in the USA only a minority of psychiatrists believe that DID is supported by strong evidence of scientific validity (Pope, Oliva, Hudson, Bodkin, & Gruber, 1999). Nevertheless, the phenomenon has been described in a range of scientific and clinical publications, and the case has been made that the symptoms can be corroborated, and that in offenders there is a strong association with documented severe abuse in childhood (Lewis, Yeager, Swica, Pincus, & Lewis, 1997). It is likely that this specific manifestation of personality disorder is culturally determined and to a certain extent iatrogenic, but it is possible that disorders of similar severity and with the same risk factors are seen more widely. Schizotypal Personality Disorder The general concept of schizotypal personality disorder is that of a pervasive pattern of social and interpersonal deficits marked by acute discomfort with, and reduced capacity for, close relationships, as well as by cognitive or perceptual distortions and eccentricities of behavior, beginning by early adulthood. It is likely that the concept of a spectrum of schizophrenic disorders, including schizotypal personality disorder, applies in the pre-adult years, as well as later. However, there have been only a small number of attempts to apply the criteria in childhood. One study suggested an overlap with pervasive developmental disorders, although not autism as such (Nagy & Szatmari, 1986); another suggested a course similar to that of schizophrenia arising in childhood (Asarnow & BenMeir, 1988); and a third suggested that there was a pattern of communication deficits similar to, but milder than, those found in schizophrenia (Caplin & Guthrie, 1992). Olin, Raine, Cannon et al. (1997) gathered prospective questionnaire data from teachers on 15-year-olds who subsequently developed schizotypal personality disorder. Compared with a range of other groups at varying levels of risk for psychiatric disorder other than schizophrenia, the schizotypal individuals as adolescents had been more passive and unengaged and more hypersensitive to criticism. Schizoid Personality Disorder The usual concept of schizoid personality disorder is of a pervasive pattern of detachment from social relationships and a restricted range of expression of emotions in interpersonal settings (American Psychiatric Association, DSM-IV, 2000). Thus, ICD-10 criteria (World Health Organization, 1996) include a lack of pleasure in activities, emotional coldness, apparent indifference to praise or criticism, a limited capacity to express either positive or negative emotions, a lack of close friends, a preference for solitary activities, a marked insensitivity to prevailing social norms and an excessive preoccupation with fantasy and introspection. The literature on the syndrome is almost confined to adults. However, since the 1970s, Wolff has pressed the case for the application of the diagnosis in childhood (Wolff & Barlow, 1979). Unfortunately for comparative purposes, her criteria differ in several key respects from those generally applied with adults. Thus, she has specified increased sensitivity and paranoid ideas, whereas ICD-10 and DSM-IV indicate marked insensitivity, and she has included an unusually odd style of communicating which would ordinarily be part of schizotypal, not schizoid, personality disorders (Wolff, 1991a,b; Wolff & Chick, 1980). The initial findings in childhood suggested some similarities with autism (Wolff & Barlow, 1979) and the follow-up showed substantial continuity with schizotypal personality disorder in adult life (Wolff, Townshend, McGuire et al., 1991). However, a study of psychophysiological responses of adults who in childhood met Wolff’s criteria for schizoid disorder failed to find abnormalities previously documented in the relatives of schizophrenic patients and adults with schizotypal personality disorder (Blackwood, Muir, Roxborough et al., 1994). Utility of Personality Disorder Diagnosis in Childhood and Adolescence In the light of these considerations, is there an argument for applying the concept of personality disorder in adolescence or even in childhood? Clearly, given the probable heterogeneity CHAPTER 50 846 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 846

of the personality disorders, it is unlikely that there will be one answer to this question. There would be a strong case if it could be shown that the category unified diverse symptoms with a common cause, prognosis or response to treatment. It is for example clearly the case that the diagnosis of autism, which meets most of the requirements for personality disorder, provides an effective unifying diagnosis for diverse social and behavioral abnormalities. Might this be true also for childhood conditions such as oppositional defiant disorder or conduct disorder? Angold and Costello (2001) reviewed the DSM criteria for personality disorder outlined earlier and concluded that early-onset antisocial behaviors in children satisfy all the DSM requirements. Childhood oppositional and conduct problems associated with poor peer relationships are more likely to persist (Vitaro, Brendgen, & Tremblay, 2000), and adult antisocial outcomes are most common in children with multiple and diverse oppositional, social and educational difficulties (Stattin & Magnusson, 1996). However, the use of the personality disorder designation could introduce the limitations of the adult categories into childhood. Crucially, it might reduce our scope for investigating the links between different types of dysfunction. Vitaro, Brendgen, & Tremblay, (2000) found that, in boys, although having a best friend who was deviant at age 10 predicted delinquency at age 14, this was much less the case among those with warm relationships with their parents. Such a specific mechanism could not be investigated within a personality disorder framework if poor peer relationships was simply one of a number of items within a childhood ASPD. Similarly, notwithstanding the strong association of conduct disorder and ADHD and evidence that ADHD in the presence of conduct disorder increases the likelihood of persistence (Loeber, Green, Keenan, & Lahey, 1995), important genetic and neuropsychological differences would not have been identified if they had been included in a hyperactivity–conduct personality disorder. Whatever the pros and cons of creating childhood personality disorders, or of other kinds of solution such as dissolving the Axis I–Axis II demarcation, the placement of disruptive disorders of childhood on DSM Axis I, and antisocial personality disorder on Axis II, is an anomaly and does not reflect a fundamental distinction between them. Is there a case for applying adult personality disorder criteria in adolescence? Studies of hospital patients, mainly with BPD, have suggested that adolescent personality disorders may be less distinct and less stable than adult disorders, but the profile of symptoms is largely similar (Becker, Grilo, Edell, & McGlashan, 2004). There is a case for using a dimensional approach to personality disorders in adult life, and this is likely to be the case also in adolescence. The stability of personality disorder symptoms is no less during adolescence, and from adolescence to adult life, than it is in adult life (Cohen, Crawford, Johnson et al., 2005). Correlation coefficients of 0.5–0.7 are commonly reported between adolescent and adult measures and over periods of up to 10 years. Equally, there can be informative developmental sequences involving personality disorder symptoms. Hamigami, McArdle, and Cohen (2000) showed that there were reciprocal effects of narcissistic and borderline symptoms on each other from early adolescence into the mid-twenties. Borderline symptoms predicted increasing narcissistic symptoms over time, but narcissistic symptoms predicted lower borderline symptoms! This may indicate that the appearance of borderline symptoms in early adolescence reflects substantial underlying pathology, whereas early narcissistic symptoms may be more developmentally benign. Later narcissistic symptoms, by contrast, may also reflect more problematic functioning, perhaps particularly when associated with borderline features. Borderline Personality Disorder in Adolescence Many of the studies of personality disorder in adolescence have focused on BPD. A substantial proportion of adolescent psychiatric in-patients in the USA and the UK can be diagnosed with the disorder using adult criteria. It resembles the adult diagnosis in that there is a strong association with episodic disorders, particularly depression, and its stability is low. In adolescents, the BPD diagnosis picks out a group who, compared with other adolescent psychiatric patients, have experienced high levels of maternal neglect and rejection, grossly inappropriate parental behavior and a number of parental surrogates, sexual abuse (Ludolph, Westen, Misle et al., 1990) and higher levels of angry irritable interactions in the family (James, Berelowitz, & Verker, 1996). Associations of inconsistent and intrusive parenting with the emergence of BPD were found in the longitudinal study of a general population sample referred to earlier (Bezirganian, Cohen, & Brook, 1993). Clinical Implications In the light of the varied, and in many respects limited, body of research, should we make use of personality diagnoses in adolescence in clinical practice? The answer is, as is often the case, it depends on the purpose. It can serve the purpose of ensuring that attention is drawn to types of persistent dysfunction that are not reflected in the episodic diagnoses such as depression. For example, among adolescents assessed for suicidal behaviors, a substantial minority is depressed and a smaller group has borderline personality disorder. There are implications for treatment depending on the presence both of depression and of BPD. However, current evidence and clinical experience would suggest that the key issue is not whether the threshold for BPD is crossed but how many features of BPD are present. Similarly, the extent to which these are accompanied by symptoms of other disorders, notably ASPD, is equally important. It should be emphasized that the evidence linking different personality disorders to treatment needs is limited in both adults and adolescents. In relation to ASPD, while there is some evidence for the efficacy of treatments such as multi-systemic therapy (MST) for juvenile offending, the studies have not assessed ASPD nor have they examined outcomes in relation to symptoms of other personality disorders such as BPD (see chapters 35 and 62). DISORDERS OF PERSONALITY 847 9781405145497_4_050.qxd 29/03/2008 02:55 PM Page 847

Dialectical behaviour therapy (DBT) is increasingly being used as an approach with suicidal adolescents, many of whom are likely to have BPD symptoms (Katz, Cox, Gunasekara, & Miller, 2004). The “dialectic” refers to helping the adolescent to change what needs to be changed, while accepting what is a valid response to circumstances (Hill, Swales, & Byatt, 2006). Treatment is delivered by a team, and has several elements. Capability enhancement aims to increase the adolescent’s basic capabilities of affect regulation, distress tolerance and interpersonal skills. This function is most often delivered by a skills training group. Motivational enhancement involves assisting adolescents to apply their new-found skills to their own life problems, in particular attending to cognitions, emotions or reinforcement contingencies that interfere with the implementation of skillful behavior. This function is most commonly delivered via individual psychotherapy. Explicit coaching in the application of problem-solutions to the adolescent’s real world situations is designed to increase generalization of skills learned in the program. This may be delivered by between-session skills coaching, by telephone, by case management or by training family members in the same skills that the adolescents are learning. Considerable attention is also paid to supervision by and support of therapists and to the family and wider environment of the adolescent, including social services and education. Studies of adult populations in relation to BPD and chronic self-harm may provide some pointers to provide support for the effectiveness of DBT and other psychosocial treatments in BPD in adolescents. Perry, Bannon, and Ianni (1999) reviewed the results of 15 studies, and Bateman and Fonagy (2000) reviewed 25 reports of a range of psychotherapeutic approaches in adult personality disorders. Both reviews concluded that there was evidence for short- to medium-term effectiveness in a number of indices of disorder including depression and interpersonal problems, as well as personality disorder diagnosis. However, there have been very few randomized controlled studies. By 2004 there had been seven randomized controlled trials of DBT, but only two had sample sizes of greater than 60 (Lieb, Zanarini, Yen et al., 2004). These suggest a major effect on suicidal behaviors sustained for up to 1 year. Bateman and Fonagy (2001) conducted a randomized controlled trial of psychoanalytically oriented partial hospitalization with 38 borderline patients, based on their theory of impaired mentalization in BPD. The treated group had a lower frequency of self-harm, suicide attempts, hospital admissions, use of medication and depression, and better social adjustment over an 18-month treatment period. Generally, studies of effectiveness of treatment of personality disorders have used outcomes such as service utilization or depression levels as proxies for measures of changes in personality functioning. Tackling the difficulties associated with the current conceptualization and measurement of personality disorder should lead to improved treatment and more refined measurement of outcomes. Bateman and Fonagy have summarized the principal features of treatments for which there is some evidence of effectiveness. The approach is active and structured with clear focus, in the context of a strong relationship between therapist and patient over a substantial period of time. The treatment is well integrated with other services available to the patient. Effective treatments for adolescents who have extensive dysfunction, paralleling that of the adult personality disorders, may well require an approach with these features, coupled with active involvement of the family and attention to educational or vocational needs. Studies have been conducted of tricyclic antidepressants, selective serotonin reuptake inhibitors, neuroleptics and mood stabilizers in BPD (Lieb, Zanarini, Yen et al., 2004). Most have shown some evidence of efficacy but with small sample sizes. Of the 16 studies reviewed by Lieb, Zanarini, Yen et al., only two had a sample size of greater than 60, and in most cases they were substantially smaller. Soler, Pascual, Campins et al. (2005) randomized 60 patients with BPD to placebo or to olanzapine together with DBT. In assessments after 4 months, the combined DBT plus drug group had lower affective symptoms and impulsivity/aggression than the DBT only group. Conclusions Much remains to be discovered regarding the forms of dysfunction that best characterize the personality disorders. This will inform, and be informed by, increasing knowledge of childhood antecedents, developmental pathways, the interaction between individual and environmental processes, and basic psychopathological processes in the personality disorders. For all its shortcomings, the concept of personality has endured because it adds to our understanding of risk and psychopathology and, increasingly, research in this area highlights individuals with distinctive treatment needs. Patterns of symptomatology resembling adult personality disorders are seen during adolescence and also indicate distinctive and generally intensive treatment needs. References American Psychiatric Association. (2000). Diagnostic and statistical manual of mental disorders (4th edn.). Text Revision. Washington, DC: American Psychiatric Association. Angold, A., & Costello, J. (2001). 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852 Psychopathy as a Clinical Entity Classification of Psychopathy The origins of the current description of the psychopathy syndrome can be traced back to the work of Cleckley (1941) and his book, The Mask of Sanity. Some of the key features of psychopathy recorded by Cleckley (1941) included: absence of nervousness, interpersonal charm, lack of shame, impoverished affect and poorly motivated antisocial behavior. From the criteria delineated by Cleckley, and his own clinical impressions, Hare (1980) developed the original Psychopathy Checklist (PCL), a formalized tool for the assessment of psychopathy in incarcerated adults. On the basis of considerable empirical work, this has been revised: first in 1991, the Psychopathy Checklist–Revised (PCL-R; Hare, 1991), and then again in 2003 (Hare, 2003). The first extension of the psychopathy concept to children was Bowlby’s (1946) description of “affectionless psychopathy.” It mirrored some of the key features introduced by Cleckley (such as lack of responsiveness to the suffering of others), but lay forgotten for a long time until the extension of the psychopathy construct to children was proposed again by Frick, O’Brien, Wootton, and McBurnett (1994). This chapter considers how far neuropsychological formulations about psychopathy in adult life may be useful for understanding related characteristics in childhood. The current definitions of psychopathy comprise both ratings of emotional dysfunction and ratings of overt antisocial behavior. The emotional dysfunction definition involves reduced guilt and empathy as well as reduced attachment to significant others. The antisocial behavior definition involves a predisposition to antisocial behavior from an early age and comprises overt behavioral problems also manifest in those individuals with antisocial behavior who do not have psychopathy (e.g., adults with antisocial personality disorder [ASPD] and children with conduct disorder [CD]). This two-component structure was identified through factor analysis in samples of clinic children and incarcerated adults (Frick, O’Brien, Wootton et al., 1994; Hart, Forth, & Hare, 1990). Emotional dysfunction is often considered be the core distinctive feature of psychopathy (Blair, Mitchell, & Blair, 2005). However, most of the existing research has concentrated on criminal psychopathy and in order to qualify for the syndrome of psychopathy at the present, an individual must present with both emotional dysfunction and antisocial behavior (Hare, 2003). This has led to the notion of psychopathy as a subgroup within the antisocial behavior diagnoses in childhood and adulthood. Limited research suggests that “successful psychopaths” (i.e., those who manifest the emotional dysfunction, but do not engage in antisocial behavior) do exist and as such psychopathy may not invariably need to be accompanied by antisocial behavior. However, this chapter concentrates on reviewing research on psychopaths for whom the emotional dysfunction is combined with antisocial behavior. Psychopathy is not part of the DSM-IV diagnostic system (see chapter 2). Given the current definition of psychopathy syndrome that comprises both the emotional dysfunction and antisocial behavior, most individuals with psychopathy would receive a childhood diagnosis of CD or adulthood diagnosis of ASPD (see chapters 35 and 50, respectively). The diagnosis of CD groups individuals together because they engage in antisocial behavior. Thus, children with CD can be found whose antisocial behavior is associated with the specific form of reduced emotional responsiveness characteristic of psychopathy as well as children whose antisocial behavior is associated with heightened emotional responsiveness or dysregulated emotional responding (Blair, Mitchell, & Blair, 2005). In short, the DSM-IV CD diagnosis identifies a heterogeneous population who do not appear to share a common etiology and have some distinct clinical features (with resulting troubling implications for successful treatment). In contrast, the available evidence points towards a core neurocognitive profile shared by a number of individuals with psychopathy. Assessment of Psychopathy Following the development of the adult PCL-R, tools for the assessment of psychopathy in childhood and adolescence have also been developed. Here we briefly highlight those measures that directly stem from the PCL-R. These include the Psychopathy Checklist: Youth Version (PCL-YV; Forth, Kosson, & Hare, 2003; Kosson, Cyterski, Steuerwald, Neumann, & Walker-Matthews, 2002) and the Antisocial Process Screening Device (APSD; Frick & Hare, 2001). There is evidence of substantial convergence among these measures (Salekin, Leistico, Trobst, Schrum, & Lochman, 2005). The assessment techniques (PCL-R, PCL-YV and APSD) all involve 20 behavioral items. Both the PCL-R and PCL-YV are Psychopathy 51 R. James Blair and Essi Viding 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 852 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

scored on the basis of an extensive file review and a semistructured interview. The APSD takes the format of a parent or teacher-rated questionnaire. For each behavioral item, an individual can score between 0 and 2 points. The individual’s total score can therefore vary from 0 to 40 points. Adults scoring 30 or above on the PCL-R are generally considered as having psychopathy, at least in the USA, while those scoring less than 20 are considered as not having psychopathy. There are less-well-established criteria for considering whether a child has psychopathy. Typically, in empirical studies, cut-offs of 20+ scores have been used. Given that emotional dysfunction is what sets psychopaths apart, any APSD scoring criteria should ensure a high score on the emotional dysfunction index (i.e., the callous–unemotional scale of the APSD). The inter-rater reliability for the PCL-R (Hare, 1991) and APSD is comparable with other instruments assessing psychopathology (Frick & Hare, 2001; Frick, Kimonis, Dandreaux, & Farell, 2003). At the present, callous–unemotional traits are not routinely assessed when making a clinical diagnosis of CD. The assessment of callous–unemotional traits in research settings has generally relied on parent and teacher ratings. Such assessments could be easily adapted for clinical use to supplement standard diagnostic interviews. Utility and Validity of the Classification Evidence for the validity of the psychopathy construct is extensive for clinic and criminal populations. Much research has been devoted to relating psychopathy construct to other instruments and scales, collecting data on recidivism and violence as well as laboratory measures involving behavioral, psychophysiological and functional magnetic resonance imaging (fMRI) indices (see Patrick, 2006). One of the major strengths of the construct has been its utility in risk assessment. Considerable work has shown the predictive power of scores on the PCL-R with respect to recidivism (for a review of this literature see Douglas, Vincent, & Edens, 2006). Moreover, this work has shown that the correlation between recidivism and psychopathy is significantly higher than that of the DSM diagnoses (Hare, 2006). The evidence of the success of the psychopathy construct in risk assessment has been almost exclusively conducted in adult samples (for data with adolescent samples see Frick & Marsee, 2006; Frick, Stickle, Dandreaux, Farell, & Kimonis, 2005). The available data indicate that children with psychopathy show more conduct problems, more severe aggression, poorer prognosis and more instrumental aggression than other children with conduct problems (see chapter 35; Frick & Marsee, 2006). For children, considerable stability (r > 0.70 across all time points) of the core emotional dysfunction has been demonstrated over a 4-year period (Frick et al., 2003). A long-term follow-up study measuring psychopathy at age 13 years demonstrated that the construct showed moderate stability to age 24 years (r = 0.32), despite different informants and assessment instruments used across the two age periods (Lynam, Caspi, Moffitt, Loeber, & Stouthamer-Loeber, 2007). This 11-year correlation is equivalent to that typically seen when different informants use the same instrument at the same time-point to rate an individual’s behavior on a construct. Importantly, recent data have shown that differentiating children who receive the DSM diagnoses of CD or oppositional defiant disorder (ODD) on the basis of the callous–unemotional (the emotional dysfunction component of psychopathy) traits has clear implications for treatment prognosis (Hawes & Dadds, 2005). Boys with high callous–unemotional traits were less responsive to treatment, and callous–unemotional traits uniquely predicted clinical outcomes when analyzed in relation to CD/ODD severity, other predictors of antisocial behavior, and parents’ implementation of treatment (Hawes & Dadds, 2005). Moreover, children with psychopathy appear more difficult to socialize through standard socialization techniques (Oxford, Cavell, & Hughes, 2003; Wootton, Frick, Shelton, & Silverthorn, 1997). As most of the research on psychopathy has concentrated on clinic or incarcerated populations, or has focused on the impact of psychopathy on antisocial outcomes, much less is known about the utility and validity of psychopathy construct in predicting other outcomes aside from antisocial behavior. Co-occurrence With Other Syndromes Attention Deficit/Hyperactivity Disorder Considerable work has demonstrated that CD and attention deficit/hyperactivity disorder (ADHD) often co-occur (Hinshaw, 1987; Taylor, Schachar, Thorley, & Wieselberg, 1986). More recent work has suggested that psychopathy and ADHD also frequently co-occur (Barry, Frick, DeShazo et al., 2000; Colledge & Blair, 2001; Lynam, 1996). However, it remains unclear whether the ADHD seen in the context of psychopathy can be considered equivalent to that seen in the absence of psychopathy symptoms or whether it should be considered as a behavioral phenocopy with a divergent neurocognitive signature (Blair, Mitchell, & Blair, 2005). Autism and Asperger’s Syndrome Both autism and Asperger’s syndrome are, like psychopathy, associated with problems in “empathy” (see chapter 46). However, the nature of the empathy impairment appears very different between the two populations (Blair, 2003a). Individuals with autism and Asperger’s syndrome, as a group, tend to show impairment in Theory of Mind, the ability to represent the mental states of others (Baron-Cohen, Leslie, & Frith, 1985; Frith & Happé, 2005). Individuals with psychopathy do not (Blair, Sellars, Strickland et al., 1996; Richell, Mitchell, Newman et al., 2003). Individuals with psychopathy show impairment in processing the fearful and sad expressions of others and are poor at feeling for others, while capable of representing mental states (Blair, 2005; Blair, Colledge, Murray, & Mitchell, 2001). Individuals with autism or Asperger’s syndrome, while potentially showing a global impairment in processing emotions, do not show indications of specific impairment for fear and sadness and appear to find others’ distress aversive (Adolphs, Sears, & Piven, 2001; PSYCHOPATHY 853 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 853

Blair, 1999; Rogers, Viding, Blair, Frith, & Happé, 2006). To date there have been no reports of comorbid psychopathy and either autism or Asperger’s syndrome although there have been isolated reports of individuals showing some of the emotion dysfunction associated with psychopathy (Fine, Lumsden, & Blair, 2001; Rogers, Viding, Blair et al., 2006). As pointed out by Rutter (2005), unlike for psychopathy, the impaired socioemotional responsiveness found in autism spectrum disorders does not seem to be generally linked to increased risk for antisocial behavior. In support of this, Hippler & Klicpera (2003), in their study of 177 cases originally diagnosed by Asperger, found no raised incidence of criminal offences compared with general population rates. In contrast, Frick, Stickle, Dandreaux et al. (2005) have demonstrated that elevated levels of psychopathy predict increased antisocial behavior at a later time-point, even in children who do not start off as being antisocial. It thus appears that although both psychopaths and individuals with autism spectrum disorders can appear emotionally blunted, there are likely to be different roots to their emotional problems, which also explains the lack of increased risk for antisocial behavior in people with autism spectrum disorders. However, more research is needed that provides direct comparison of individuals with psychopathy and individuals with autism spectrum disorders. Anxiety and Mood Disorders Individuals presenting with anxiety and mood disorders (e.g., post-traumatic stress disorder and depression; see chapters 42 and 37, respectively) are at elevated risk for aggression at the time of this presentation (Silva, Derecho, Leong, Weinstock, & Ferrari, 2001; Zoccolillo, 1992). For example, in a recent community sample of almost 6000 individuals, over half (54.33%) of the 3.3% of adults with ASPD had a comorbid anxiety disorder (lifetime; Goodwin & Hamilton, 2003). Similarly, 42.31% of adults with a history of CD (9.4%) but who did not meet criteria for ASPD had a lifetime anxiety disorder. In contrast to ASPD, psychopathy has been traditionally considered to be marked by reduced anxiety levels (Cleckley, 1941; Hare, 1970; Lykken, 1957). Moreover, studies have shown that anxiety level is inversely associated with the callous and unemotional/emotional dysfunction dimension of psychopathy (Frick, Lilienfeld, Ellis, Loney, & Silverthorn, 1999; Patrick, 1994; Verona, Patrick, & Joiner, 2001). In short, increases in anxiety are associated with increases in antisocial behavior but decreases in the emotional dysfunction component of psychopathy (for further discussion of this issue see Blair, Mitchell, & Blair, 2005). Epidemiology of Psychopathy The prevalence rates of CD are very high (6–16% for males and 2–9% for females, according to DSM-IV). The prevalence rate of psychopathy is far lower although full epidemiological studies have not been conducted. However, preliminary work conducted by Frick using the APSD, and using a cutoff for psychopathy commonly used in the literature, results in a prevalence rate of psychopathy of 1–3.5% (Frick & Hare, 2001). Using what we know about prevalence rates of ASPD from epidemiological studies and the proportion of psychopaths in the prison ASPD group, we can conservatively estimate the incidence of adult psychopathy at 0.75% (Blair, Mitchell, & Blair, 2005). There is very little known with respect to prevalence and gender. Work on psychopathy has been conducted largely on males and relatively little is known about psychopathy in females (for a review see Verona & Vitale, 2006). Development of the Behavioral Manifestation There are few longitudinal data on the development of psychopathy, making it impossible to effectively describe how its behavioral manifestation varies with age. However, a recent study does suggest that cruelty to animals may be a potent indicator of early callous–unemotional predisposition (Dadds, Whiting, & Hawes, 2006). There is also some suggestion that fearlessness in early childhood may be one index of risk for callous–unemotional traits and conduct problems (Frick & Marsee, 2006; Kochanska, Gross, Lin, & Nichols, 2002). The importance of assessing age-appropriate behavioral features was recognized during the development of assessment instruments such as the APSD and PCL-YV. For example, attention is given to whether a child keeps the same friends rather than whether they have multiple marital partners (Frick & Hare, 2001). However, the extent to which individual components of the psychopathy are associated with the syndrome across timepoints remains unknown. The few existent data indicate reasonable stability of callous–unemotional traits from middle childhood to early adolescence (see Frick & Marsee, 2006 p. 853). Also, it is worth noting the literature on fearlessness. While it is not an explicit measure of psychopathy, it has long been associated with the disorder (Cleckley, 1941; Patrick, 1994). This is interesting because fearlessness also shows moderate stability in early childhood, as well as a clear relationship with empathic and moral development (Kochanska, Gross, Lin et al., 2002). Does Psychopathy Extend into the Normal Distribution? There has been some debate as to whether psychopathy is best characterized as a taxon or a constellation of normal personality traits (Edens, Marcus, Lilienfeld, & Poythress, 2006; Lynam, Caspi, Moffitt et al., 2005; Vasey, Kotov, Frick, & Loney, 2005). This debate remains unresolved. What is clear is that psychopathy can be mapped as a mixture of normal personality dimensions (see chapters 14 and 50; Lynam, Caspi, Moffitt et al., 2005). In addition, differences in brain function relevant to psychopathy can be identified within the healthy population and related to subclinical features of psychopathy (Gordon, Baird, & End, 2004). Although psychopathy can be mapped to a mixture of normal personality dimensions, and community samples high on psychopathic traits can display functional brain differences similar to those seen in incarcerated psychopaths, this does not suggest that psychopathy should necessarily be viewed as “normal.” Cleckley (1941) strongly CHAPTER 51 854 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 854

asserted that psychopathy represented something distinct from normal variations of personality. As pointed out by Rutter (2005), none of the existing measures of normal personality functioning set out to characterize the emotional dysfunction that defines psychopathy. In addition, we have no data addressing the question of whether the differences in brain function that are relevant to psychopathy need to reach a certain quantitative threshold before we see a true risk for the extreme psychopathic personality that might qualify as a taxon. Origins of Psychopathy Genetic Factors A recent comprehensive meta-analysis of behavioral genetic studies on antisocial behavior concluded that antisocial behavior is moderately heritable (Rhee & Waldman, 2002). Work on psychopathy, a major risk factor for antisocial behavior marked by both reactive and instrumental acts, has revealed, using self-report measures in adult and adolescent populations, moderate heritability of callous–unemotional related traits, no shared environmental influence and moderate non-shared environmental influence (Blonigen, Hicks, Krueger, Patrick, & Iacono, 2005; Larsson, Andershed, & Lichtenstein, 2006; Taylor, Loney, Bobadilla, Iacono, & McGue, 2003). The heritability estimates were similar or slightly higher than typically found for self-report personality questionnaires (Loehlin, 1992). However, the sample sizes in these adult and adolescent samples were either relatively small or at best moderate. In recent work with around 3500 twin pairs, we have shown that callous–unemotional traits are strongly heritable (67% heritability) at both 7 and 9 years (Viding, Blair, Moffitt, & Plomin, 2005). Moreover, antisocial behavior in the presence of elevated levels of callous–unemotional traits is strongly heritable (81%). However, heritability of antisocial behavior without callous–unemotional traits was only moderate (30%; Viding, Blair, Moffitt et al., 2005). Importantly, in addition, the high heritability of antisocial behavior in children with callous– unemotional traits has been shown to be independent of cooccurring ADHD pathology (Viding, Jones, Frick et al., 2008). Genes regulating serotonergic neurotransmission, in particular monoamine oxidase A (MAOA), have been highlighted in the search for a genetic predisposition to antisocial behavior (Lesch, 2003). The MAOA gene contains a well-characterized functional polymorphism consisting of a variable number of tandem repeats in the promoter region, with high- (MAOA-H) and low-activity (MAOA-L) allelic variants. The MAOA-H variant is associated with lower concentration of intracellular serotonin, whereas the MAOA-L variant is associated with higher concentration of intracellular serotonin. Recent research suggests that genetic vulnerability to antisocial behavior conferred by MAOA-L may only become evident in the presence of an environmental trigger, such as maltreatment (Caspi, McClay, Moffitt et al., 2002; Kim-Cohen, Caspi, Taylor et al., 2006). Despite the demonstration of genetic influences on individual differences in antisocial behavior, it is important to note that no genes for antisocial behavior exist. Instead, genes code for proteins that influence characteristics such as neurocognitive vulnerabilities which may in turn increase risk for antisocial behavior. Thus, although genetic risk alone may be of little consequence for behavior in favorable conditions, the genetic vulnerability may still manifest at the level of brain or cognition. Imaging genetics studies attest to genotype differences being associated with variation in brain structure and function in non-clinical samples (Meyer-Lindenberg & Weinberger, 2006). We can think of this as the neural fingerprint; ready to translate into disordered behavior in the presence of unfortunate environmental triggers. Meyer-Lindenberg, Buckholtz, Kolachana et al. (2006) recently provided the first demonstration of the MAOA-L genotype being associated with a pattern of neural hypersensitivity to emotional stimuli. Specifically, they reported increased amygdala activity coupled with lesser activity in the frontal regulatory regions in MAOA-L compared to MAOA-H carriers. Meyer-Lindenberg, Buckholtz, Kolachana et al. (2006) speculated that their brain imaging findings of poor emotion regulation in MAOA-L carriers relate to threat reactive and impulsive, rather than psychopathic antisocial behavior. They based this speculation on the findings of individuals with psychopathy displaying under-reactivity of the brain’s emotional circuit, particularly the amygdala (Birbaumer, Veit, Lotze et al., 2005; Kiehl, Smith, Hare et al., 2001). Currently, while the behavioral genetics data suggest heritability of the emotional component of psychopathy, a molecular level account of the disorder remains in its infancy. Suggestions have been made that psychopathy might be related to anomalies in noradrenergic neurotransmission (Blair, Mitchell, & Blair, 2005). It is also interesting to note that a small number of studies have reported increased vulnerability to antisocial behavior in the presence of the MAOA-H allele (Manuck, Flory, Ferrell, Mann, & Muldoon, 2000). These may reflect false positive findings, but it is also possible to speculate that the amygdala hyporeactivity as opposed to hyper-reactivity seen in individuals with psychopathy could be influenced by MAOA-H, rather than MAOA-L genotype. This suggestion remains highly speculative, and as for any behavior, the genetic influences will not be limited to a single candidate gene. Other genes influencing amygdala reactivity, such as genes encoding 5-HTT, could also have a role in psychopathy. Environmental Factors Many environmental causes have been suggested for an increased risk for aggression and, at least in the lay population, psychopathy. We will consider four sets of potential causes: exposure to extreme threats (e.g., in the context of childhood abuse and/or chronic exposure to violence), incompetent parenting, environmental insults and environmental motivators (e.g., a lack of money; see chapter 25). Exposure to Extreme Threats and Incompetent Parenting There are considerable data that exposure to extreme threats increases the risk for aggression or being diagnosed with CD. PSYCHOPATHY 855 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 855

This is seen as a result of physical and sexual abuse (Dodge, Pettit, Bates, & Valente, 1995; Farrington & Loeber, 2000) as well as a result of exposure to violence in the home or neighborhood (Miller, Wasserman, Neugebauer, GormanSmith, & Kamboukos, 1999; Schwab-Stone, Chen, Greenberger et al., 1999). There are also considerable data that exposure to poor parenting increases the risk for aggression or being diagnosed with CD (Snyder, Cramer, Afrank et al., 2005). Moreover, Bowlby attributed “affectionless psychopathy” to family adversity that especially involved family disruption (Bowlby, 1946) and “impaired empathy” is considered an aspect of the “disinhibited attachment” that is characteristic of some children experiencing profound early institutional deprivation (O’Connor & Rutter, 2000; Rutter, 2005). Parental abuse is also frequently reported in forensic samples, including individuals diagnosed with psychopathy (Hare, 2003). A recent study specifically addressed the effect of childhood maltreatment on the different facets of psychopathy and found that maltreatment increases the risk for impulsive and irresponsible lifestyle, but does not have an effect on the core emotional dysfunction in psychopathy (Poythress, Skeem, & Lilienfeld, 2006). The preliminary work by Poythress Skeem, & Lilienfeld (2006) suggests that the core emotional dysfunction of psychopathy may not be affected by maltreatment. In addition, work by Pollak and colleagues suggests that maltreated children show hyper-reactivity to anger, which is in contrast to hyporeactivity to fear and sadness seen in psychopaths (Pollak & Sinha, 2002). Animal studies can also provide some clues about the effects of maltreatment on emotional reactivity, although such work has to be interpreted with caution with regard to humans. Animal studies clearly show that prolonged threat or stress leads to a long-term potentiation of the neural and neurochemical systems that respond to threat (i.e., the individual becomes more responsive to aversive stimuli; Bremner & Vermetten, 2001; King, 1999). Moreover, animal studies also show that poor parenting is also associated with increased responsiveness to aversive stimuli (Rilling, Winslow, O’Brien et al., 2001; Sanchez, Noble, Lyon et al., 2005). Importantly, this concurs with much of the human literature (Pollak & Sinha, 2002). Thus, traumatic exposure, including exposure to violence in the home or neighborhood, appears to increase the risk for mood and anxiety disorders in general, although not all exposed to trauma will go on to develop these disorders (Charney, 2003; Gorman-Smith & Tolan, 1998; Schwab-Stone, Chen, Greenberger et al., 1999). In other words, animal and much human work indicates that trauma and poor parenting increase emotional responsiveness to aversive stimuli (for an account of why this increased emotional responsiveness to aversive stimuli should be a risk factor for increased levels of reactive aggression and some forms of CD see Blair, Mitchell, & Blair, 2005), whereas a defining feature of psychopathy is the reduction, not elevation, in the individual’s responsiveness to threat (Cleckley, 1941; Hare, 1970). More research is needed to investigate whether maltreated and neglected children displaying poor empathy and attachment problems show a similar neurocognitive profile to that seen in psychopathy. Current literature suggests that for the majority of cases, maltreatment produces a neural and behavioral signature that is different from that commonly observed in individuals with psychopathy. However, it is plausible that both biologically and environmentally “weighted” routes to the same pathophysiology can take place, even if one route is considerably more common than the other (for a discussion of this issue with respect to autism spectrum disorders see chapter 12). Environmental Insults Birth complications such as anoxia (lack of oxygen) and preeclampsia (hypertension leading to anoxia) are environmental factors that can give rise to brain damage. Minor physical anomalies (MPAs) are relatively minor physical abnormalities consisting of such features as low-seated ears and a furrowed tongue that are associated with disorders of pregnancy and are thought to be a marker for fetal neural maldevelopment towards the end of the first 3 months of pregnancy. They can be caused by environmental factors acting on the fetus such as anoxia and infection (Guy, Majorski, Wallace, & Guy, 1983) or can also indicate genetic liability. Several studies have shown that babies who have birth complications or present with MPAs are more likely to develop CD and delinquency and commit violence in adulthood, particularly when other psychosocial risk factors are present (Hodgins, Kratzer, & McNeil, 2001; Pine, Shaffer, Schonfeld, & Davies, 1997; Raine, 2002; although see, for example, Laucht, Esser, Baving et al., 2000). Unfortunately, the literature has not considered whether birth complications or MPAs are a risk factor for the emergence of psychopathy or syndromes linked to heightened levels of reactive aggression. However, there is no reason to believe that such environmental insults could not detrimentally affect neural systems associated with the disorder (i.e., they could be causal for the disorder). Future research is needed to clarify this issue. Environmental Motivation Lower socioeconomic status (SES) and lower IQ are both associated with an increased risk for antisocial behavior, including the antisocial behavior component of psychopathy (Frick, O’Brien, Wootton et al., 1994; Hare, 2003). The SES data suggest a social contribution to at least the behavioral manifestation of psychopathy. Importantly, some antisocial behavior shown by individuals with psychopathy is instrumental in nature: it has the goal of gaining another’s money, sexual favors or “respect” (Cornell, Warren, Hawk et al., 1996; Williamson, Hare, & Wong, 1987). Individuals can attempt to achieve these goals through a variety of means. Having a higher SES (or for that matter intelligence) enables a wider choice of available routes for achieving these goals than having a lower SES or IQ. Lower SES or IQ limits the behavioral options. A healthy individual of limited SES or IQ may also have a narrow range of behavioral options but will exclude antisocial behavior because of aversion to this behavior formed during socialization. In contrast, individuals with psychopathy CHAPTER 51 856 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 856

PSYCHOPATHY 857 may entertain the antisocial option because they do not find the required antisocial behavior aversive. SES is also likely to impact on the probability of displaying instrumental aggressions by determining relative reward levels for particular actions. If the individual already has $100,000, the subjective value of the $50 that could be gained if the individual mugged another person on the street is low. In contrast, if the individual has only 50 cents, the subjective value of the $50 will be very high indeed (for a discussion of subjective value see Tversky & Kahneman, 1981). Pathophysiology of Psychopathy Two levels of accounts are considered: neural (which systems are dysfunctional in individuals with psychopathy) and cognitive (what functional impairments are seen in psychopathy and why should they give rise to symptoms). Accounts at the Neural System Level Frontal Lobes There have long been suggestions that frontal lobe executive function dysfunction is causally related to psychopathy in particular or antisocial behavior more generally (Gorenstein, 1982; Moffitt, 1993; Raine, 2002). Three main strands of data support this contention: 1 Neuropsychological data indicate that individuals with antisocial behavior show impaired performance on classic measures of executive functioning (for reviews of this literature see Kandel & Freed, 1989; Morgan & Lilienfeld, 2000); 2 Neuroimaging data indicate that aggressive individuals are marked by reduced frontal functioning (Goyer, Andreason, Semple et al., 1994; Raine, Meloy, Bihrle et al., 1998; Volkow, Tancredi, Grant et al., 1995); 3 Neurological patients with lesions of orbital frontal cortex, whether these occur early in life or adulthood, present with a heightened risk for aggression (Anderson, Bechara, Damasio, Tranel, & Damasio, 1999; Grafman, Schwab, Warden, Pridgen, & Brown, 1996). However: 1 Individuals with psychopathy, unlike more general antisocial populations, show no impairment on measures of executive functioning linked to either dorsolateral prefrontal cortex or regions of medial frontal cortex including dorsal regions of anterior cingulate (Blair, Newman, Mitchell et al., 2006; Kandel & Freed, 1989; LaPierre, Braun, & Hodgins, 1995). Impairment, when seen, is seen strictly for measures of executive functioning linked to orbital and inferior frontal cortex (e.g., response reversal; Blair Newman, Mitchell et al., 2006; LaPierre, Braun, & Hodgins, 1995). 2 The neuroimaging data reveal a complex pattern with respect to frontal cortex and psychopathy. The results of early work indexing activity during rest in generally antisocial populations (Goyer, Andreason, Semple et al., 1994; Raine, Meloy, Bihrle et al., 1998; Volkow, Tancredi, Grant et al., 1995) have not been borne out with more recent event-related fMRI studies. More recent studies have indicated the possibility of dysfunction in orbital frontal cortex and anterior cingulate (Birbaumer, Veit, Lotze et al., 2005; Kiehl, Smith, Hare et al., 2001). 3 Many of the functional impairments shown by neurological patients with acquired lesions of orbital frontal cortex are markedly different from those shown by individuals with psychopathy (Blair & Cipolotti, 2000). Moreover, clinically, while neurological patients with acquired lesions of orbital frontal cortex present with an increased risk for reactive aggression but not instrumental aggression (Anderson, Bechara, Damasio et al., 1999; Grafman, Schwab, Warden et al., 1996), individuals with psychopathy are at increased risk for both forms of aggression (Cornell, Warren, Hawk et al., 1996; Williamson, Hare, & Wong, 1987). We have argued elsewhere that “frontal lobe” explanations of psychopathy need greater specification (Blair, 2004). The current data indicate no impairment in executive functions reliant on dorsolateral prefrontal cortex and medial frontal cortex. However, there are reasons to believe that there may be impairment in executive functions reliant on orbital and inferior frontal cortex. This may indicate orbital and/or inferior frontal cortex dysfunction (Blair, Newman, Mitchell et al., 2006; LaPierre, Braun, & Hodgins, 1995). However, it is worth noting that it may also indicate dysfunction in systems that feed information to orbital and/or inferior frontal cortex. Currently, the fMRI studies that have indicated reduced activity in these regions in adults with psychopathy have all used tasks where there is also reduced amygdala activity and where the amygdala is necessarily involved in the transmission of information to these frontal regions (Birbaumer, Veit, Lotze et al., 2005; Kiehl, Smith, Hare et al., 2001). Therefore, studies that employ tasks that do not recruit amygdala are needed to ascertain whether the differences in orbital and/or inferior frontal cortex are independent from amygdala input. It is important to note that the executive function consistently identified as impaired in individuals with psychopathy is response reversal (Budhani & Blair, 2005). Response reversal is the ability to make a new response to gain a reward when the old behavior that previously gave rise to the reward no longer does so (Rolls, 1997). It is unlikely that impairment in this executive function is causally related to psychopathy per se. This is because impairment in response reversal is seen in other clinical populations, in particular children with bipolar disorder (Gorrindo, Blair, Budhani et al., 2005). Importantly though, a common feature of both childhood bipolar disorder and psychopathy is an increased risk for reactive aggression (Frick, Cornell, Barry, Bodin, & Dane, 2003; Leibenluft, Blair, Charney, & Pine, 2003). Reactive aggression can be associated with frustration, as well as perceived threat. An individual who cannot change their behavior to accommodate to changing real-life contingencies, an individual who cannot perform response reversal, is someone at increased risk for reactive aggression. It therefore appears plausible that while response reversal impairment is not causally related to the development of the full syndrome of psychopathy, it is causally related to the increased risk for reactive aggression and that 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 857

CHAPTER 51 858 this is also seen in other childhood psychiatric conditions (Blair, Mitchell, & Blair, 2005). Amygdala Patrick (1994) originally suggested that amygdala dysfunction might be causally related to the development of psychopathy, and Blair, Mitchell, & Blair (2005) have developed and extended this position. Two main strands of data support the suggestion of amygdala dysfunction in psychopathy: 1 Neuropsychological data indicate that individuals with psychopathy show impairment on tasks indexing the functional integrity of the amygdala. Thus, individuals with psychopathy show impairment in aversive conditioning (Flor, Birbaumer, Hermann, Ziegler, & Patrick, 2002), the augmentation of the startle reflex by visual threat primes (Levenston, Patrick, Bradley, & Lang, 2000), passive avoidance learning (Newman & Kosson, 1986) and fearful expression recognition (Blair Colledge, Murray et al., 2001); and 2 Neuroimaging data indicate that adults with the disorder are marked by reduced amygdala responsiveness during emotional memory (Kiehl, Smith, Hare et al., 2001) and aversive conditioning tasks (Birbaumer, Veit, Lotze et al., 2005; although see Muller, Sommer, Wagner et al., 2003). Importantly, the impairments seen in individuals with psychopathy closely mirror those seen in patients with amygdala damage. Thus, lesions of the amygdala disrupt aversive conditioning (Bechara, Tranel, Damasio et al., 1995), the augmentation of the startle reflex by visual threat primes (Angrilli, Mauri, Palomba et al., 1996), passive avoidance learning (Ambrogi Lorenzini, Baldi, Bucherelli, Sacchetti, & Tassoni, 1999) and fearful expression recognition (Adolphs, 2002). However, there is a caveat. Patients with acquired damage to the amygdala do not typically show increased levels of aggression. This dissociation between the clinical implications of acquired amygdala damage and psychopathy likely relates to the developmental trajectory of the disorder (Blair, Mitchell, & Blair, 2005). The Paralimbic Hypothesis On the basis of neuroimaging data, Kiehl (2006) has argued that, in individuals with psychopathy, there is dysfunction within paralimbic cortex (i.e., amygdala, anterior superior temporal gyrus, rostral and caudal anterior cingulate, posterior cingulate, ventromedial frontal cortex and parahippocampal regions). However, neuroimaging data are notoriously unable to localize deficits; impairment in any region will lead to anomalous activity in any region reliant on the dysfunctional region for input. We can only be sure that an area is dysfunctional in a population if both neuroimaging and neuropsychological data indicate impairment. Indeed, anterior cingulate, at least, does not appear globally impaired in individuals with psychopathy. Damage to anterior cingulate is known to increase the Stroop effect (i.e., the interference by distracter information; Stuss, Floden, Alexander, Levine, & Katz, 2001). However, individuals with psychopathy show no evidence of increases in the Stroop effect; if anything the opposite (Blair, Newman, Mitchell et al., 2006; Hiatt, Schmitt, & Newman, 2004). Specificity It is important to note that for any of the brain areas of proposed involvement in psychopathy (e.g., orbitofrontal cortex and amygdala), associations with other psychopathologies also exist. For example, autism has been claimed to reflect, at least in part, aberrant functioning of the orbitofrontal– amygdala circuit to much the same direction as is seen in psychopathy (Bachevalier & Loveland, 2006). As another example, major depressive disorder is also associated with orbitofrontal cortex underactivity (Steele, Currie, Lawrie, & Reid, 2007). Clearly, these two disorders are not synonymous with psychopathy, although they share some features at the neural level and may even share overlapping behaviors. As we highlighted above, it is clearly important to combine information from both neuroimaging and neuropsychology to gain a more thorough understanding of a specific deficit profile. In addition, any other brain abnormalities associated with a given disorder, and which can potentially influence behavioral outcome, must be accounted for. Thus, with regard to autism, several other key regions, in addition to the orbitofrontal–amygdala circuitry, have emerged as important in providing explanations for the behavioral manifestations of the disorder. Finally, cognitive accounts of the disorder may work hand in hand with the neural system level of explanations to provide differentiation between disorders. Accounts at the Level of Cognitive Function Executive Accounts Executive dysfunction in psychopathy has been linked to impulsivity (Miller, Flory, Lynam, & Leukefeld, 2003; Whiteside & Lynam, 2001). This in turn has been conceptualized as the lack of premeditation (i.e., the “inability to inhibit previously rewarded behavior when presented with changing contingencies”) and the lack of perseverance (the “inability to ignore distracting stimuli or to remain focused on a particular task”; Whiteside & Lynam, 2001). In addition, it can be linked to response set modulation. This is characterized as the “rapid and relatively automatic (i.e., non-effortful or involuntary) shift of attention from the effortful organization and implementation of goal-directed behavior to its evaluation” (Newman, 1998). Both Lynam’s impulse control and Newman’s response set modulation models can be considered executive accounts because they suggest the existence of general systems operating on multiple domains. These accounts suggest that the emotional deficits seen in individuals with psychopathy are secondary to executive deficits. Both positions should predict that individuals with psychopathy would be impaired on a broad range of tasks; many tasks can be considered to involve inhibition or response set modulation. However, more recent data suggest that there are finer empirical cuts to be made than those predicted by such general models. Thus, for example, dorsolateral prefrontal cortex is involved in processes that allow the alternation of responding to different conceptual categories (shapes versus lines) or spatial locations as a function of contingency change while inferior frontal cortex is involved in 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 858

PSYCHOPATHY 859 processes that allow the alternation of responding to different objects within the same conceptual category as a function of contingency change (Roberts, Robbins, & Weiskrantz, 1998). The functions of both these regions of frontal cortex have been characterized in terms similar to those proposed by the impulse control and response set modulation positions. However, whereas individuals with psychopathy show impairment on measures of inferior frontal cortex functioning, they show no impairment for tasks that index the functional integrity of dorsolateral prefrontal cortex (Blair, Newman, Mitchell et al., 2006; LaPierre, Braun, & Hodgins, 1995). In short, even if a characterization of the impairment in individuals with psychopathy in terms of inhibition or response modulation was correct, we argue that it would be necessary to constrain such accounts such that they were not domain general but rather specific to particular neurocognitive systems. Stimulus–Reinforcement Associations, Fear, Empathy, Moral Socialization and Instrumental Antisocial Behavior A considerable animal literature shows that the amygdala is necessary for the formation of stimulus–reinforcement associations (Baxter & Murray, 2002; Everitt, Cardinal, Parkinson, & Robbins, 2003). With respect to psychopathy, it has been argued that individuals with psychopathy are impaired in the formation of stimulus–reinforcement associations (Blair, 2004). Impairment in the formation of aversive stimulus– reinforcement associations would give rise to the observed deficits in individuals with psychopathy in aversive conditioning, the augmentation of the startle reflex following the presentation of visual threat primes and passive avoidance learning (Flor, Birbaumer, Hermann, Ziegler, & Patrick, 2002; Levenston, Patrick, Bradley, & Lang, 2000; Newman & Kosson, 1986). In short, this impairment would give rise to the deficits consistent with previous suggestions that there is fear system dysfunction in psychopathy (Lykken, 1957; Patrick, 1994); for a re-evaluation of this literature see Blair (2004) and Blair, Mitchell, & Blair (2005). One important class of aversive stimuli for socialization is the distress of other individuals, the expressions of fear and sadness (Blair, 2003b). The amygdala is crucially involved in the response to these stimuli (Adolphs, 2002; Blair, 2003b). In line with suggestions of a specific form of empathy deficit, individuals with psychopathy show reduced autonomic responses to the distress cues of other individuals and impaired fearful facial and vocal expression recognition (for a review see Blair, 2003b). The argument has been made that the expressions of fear and sadness serve as social reinforcers allowing conspecifics to teach the societal valence of objects and actions to the developing individual (Blair, 2003b); actions/objects associated with the sadness/fear of others acquire, in healthy developing children, negative valence. Because of their impairment in the response to the sadness and fear of other individuals and in the formation of aversive stimulus–reinforcement associations, individuals with psychopathy are less able to take advantage of this “moral” social referencing. They should be, and are (Oxford, Cavell, & Hughes, 2003; Wootton, Frick, Shelton et al., 1997), more difficult to socialize through standard parenting techniques. They will not learn to avoid using instrumental antisocial behavior to achieve their goals. This is because of relative in-difference to the “punishment” of the victim’s distress and impairment in learning the association between this “punishment” and the representation of the action that caused the victim’s distress. If confirmed, this observation should have fundamental implications for treatment. In the future, the nature of interventions directed to children with severe conduct problems could come to vary based on the degree to which the specific child exhibits the emotional features of psychopathy. Treatment of Psychopathy Critics express concern about labeling children as having psychopathy. This concern emerges mainly because adult psychopaths are presumed to be untreatable (see chapter 50; Hare, 2003). Clinicians rightly wish to avoid applying a label to children that implies they cannot be treated. However, identifying the emotional dysfunction early on can offer an important opportunity for prevention (for a discussion of this issue see Lynam, Caspi, Moffitt et al., 2007). The concern about labeling should not curb clinical research that may lead to treatment options for this vulnerable group of youngsters. The behavioral manifestation of psychopathy is not always resistant to change. Most clinicians familiar with individuals with psychopathy will have had experience with individuals substantially modifying their behavior to achieve particular goals in institutional settings. This change in behavioral manifestation does not necessarily imply modification of the disorder’s core features, the emotional dysfunction (for a review see Harris & Rice, 2006). However, socially desirable responding could be achieved in individuals with psychopathy even if the core emotional dysfunction is resistant to complete amelioration. Individuals with psychopathy value status, power and dominance and are strong on self-interest and short-term rewards. They are poor at acting on long-term interest and low on empathy and altruism. Despite the demonstrated amygdala dysfunction in this population, several other brain areas, including most of frontal cortex, appear to function normally in psychopaths. It is thus possible to devise cognitive–behavioral intervention programs that capitalize on what is spared in terms of brain function and behavioral goal setting. Intervention programs for at-risk, generally impoverished children, even those not developed specifically to address childhood aggression and antisocial behaviors, have had longterm effects on reducing the occurrence of conduct problems (New et al., 2002; Zigler, Taussig, & Black, 1992). These programs have provided a mix of child care, family support, parental educational and job training, parent–child education and parent management training, and preschool education to high-risk families with children 0–5 years old (Connor, Carlson, Chang et al., 2006). Only one study to date has investigated 9781405145497_4_051.qxd 29/03/2008 02:55 PM Page 859

CHAPTER 51 860 how the emotion dysfunction component of psychopathy mediates the success of parent training (Hawes & Dadds, 2005). This study found that the emotional dysfunction is associated with poorer treatment success. Frick (2001) has suggested that many of the current intervention programs do not incorporate elements that would be particularly important for treating psychopathic antisocial behavior. As such, it is possible to speculate that at least some of the current treatment success statistics may be marred by not accounting for the presence of children who have psychopathy and who may dilute the estimated effect of overall treatment success. Categorizing children with CD into those with and without psychopathy is highly desirable in developing appropriate intervention approaches. Studies suggest that it is predominantly reactive, impulsive, affective aggression that responds to medication (for a review see Connor, Carlson, Chang et al., 2006). However, most studies do not separate reactive from instrumental forms of aggression and no studies as yet have examined pharmacological interventions on the emotional dysfunction (and the potential consequence of instrumental aggression) seen in psychopathy. In addition to the development of new cognitive–behavioral treatment approaches, this is an area in urgent need of research. Conclusions In conclusion, current definitions of psychopathy categorize individuals with psychopathy as a subset of individuals with CD/ASPD. It is thought that their pathological development is related to a specific form of emotional dysfunction: callous and unemotional traits. Behaviorally, individuals with psychopathy are marked by an increased risk for both reactive and instrumental aggression. Importantly, the psychopathy construct has been extremely successful in predicting long-term prognosis, particularly in adult samples. Perhaps the greatest current importance of this construct for child psychiatrists is that it identifies a sample of children who are unlikely to benefit from standard treatment strategies for children with CD/ODD (Hawes & Dadds, 2005). Behavioral genetic work indicates that callous and unemotional traits are strongly heritable and that antisocial behavior of callous–unemotional children is under considerable genetic influence. However, the corresponding molecular work remains in its infancy. What can be suggested is that the genetic contribution influences at the very minimum the functional integrity of the amygdala and the related circuitry. Because of the amygdala’s important roles in responding to distress cues, fear expressions and the formation of stimulus–reinforcement associations, the capacity of the developing child to acquire morality is severely compromised. The child is able to learn the rewards associated with antisocial behavior but is less sensitive to the punishments (the distress of the victims), increasing the risk that antisocial behavior will be used instrumentally by the child to achieve goals. Currently, there are no adequate treatment options for this disorder. This should be an area of urgent concern and translational research should focus on capitalizing on the emerging research findings from the cognitive neuroscience of psychopathy. Acknowledgments The authors’ research into psychopathy is supported by the Intramural Research Program of the National Institutes of Health: National Institute of Mental Health (R.J.R.B.), UK Department of Health and Medical Research Council (E.V.). Further Reading and Sources of Additional Information Blair, R. J. R., Mitchell, D. G. V., & Blair, K. S. (2005). The psychopath: Emotion and the brain. Oxford: Blackwell. Patrick, C. J. (2006). Handbook of psychopathy. New York: Guilford Press. Robert Hare’s home page: http://www.hare.org/ Rutter, M. (2005). Commentary: What is the meaning and utility of the psychopathy concept? Journal of Abnormal Child Psychology, 33, 499–503. References Adolphs, R. (2002). Neural systems for recognizing emotion. Current Opinion in Neurobiology, 12, 169–177. Adolphs, R., Sears, L., & Piven, J. (2001). Abnormal processing of social information from faces in autism. Journal of Cognitive Neuroscience, 13, 232–240. Ambrogi Lorenzini, C. G., Baldi, E., Bucherelli, C., Sacchetti, B., & Tassoni, G. (1999). Neural topography and chronology of memory consolidation: A review of functional inactivation findings. Neurobiology of Learning and Memory, 71, 1–18. Anderson, S. W., Bechara, A., Damasio, H., Tranel, D., & Damasio, A. R. (1999). Impairment of social and moral behaviour related to early damage in human prefrontal cortex. Nature Neuroscience, 2, 1032–1037. Angrilli, A., Mauri, A., Palomba, D., Flor, H., Birhaumer, N., Sartori, G., et al. (1996). Startle reflex and emotion modulation impairment after a right amygdala lesion. Brain, 119, 1991–2000. Bachevalier, J., & Loveland, K. A. (2006). The orbitofrontal–amygdala circuit and self-regulation of social-emotional behavior in autism. Neuroscience and Biobehavioural Reviews, 30, 97–117. Baron-Cohen, S., Leslie, A. M., & Frith, U. (1985). Does the autistic child have a “theory of mind”? Cognition, 21, 37–46. Barry, C. T., Frick, P. J., DeShazo, T. M., McCoy, M. G., Ellis, M., & Loney, B. R. (2000). The importance of callous–unemotional traits for extending the concept of psychopathy to children. Journal of Abnormal Psychology, 109, 335–340. Baxter, M. G., & Murray, E. A. (2002). The amygdala and reward. Nature Reviews Neuroscience, 3, 563–573. Bechara, A., Tranel, D., Damasio, H., Adolphs, R., Rockland, C., & Damasio, A. R. (1995). Double dissociation of conditioning and declarative knowledge relative to the amygdala and hippocampus in humans. Science, 269, 1115–1118. 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864 This chapter consists of two parts. First, we provide an overview on gender identity disorder (GID) in children and adolescents; second, we provide an overview on the paraphilias, with an emphasis on pedophilia because this diagnostic category is common among adolescents with sexual misconduct. Gender Identity Disorder Phenomenology Boys and girls diagnosed with GID display an array of sextyped behavior signaling a strong psychological identification with the opposite sex (Cohen-Kettenis & Pfäfflin, 2003; Green, 1974; Zucker & Bradley, 1995). Most of the behaviors that characterize GID begin during the preschool years (2–4 years), i.e., during the same period that more typical sex-dimorphic behaviors can be observed in young children (Ruble, Martin, & Berenbaum, 2006). Epidemiology Prevalence Contemporary epidemiological studies on the prevalence of psychiatric disorders in children and youth have not examined GID. Accordingly, estimates of prevalence have had to rely on less sophisticated approaches. It has been suggested that prevalence might be inferred from the number of persons attending clinics for adults that serve as gateways for hormonal and surgical sex reassignment. Because not all gender-dysphoric adults may attend such clinics, this method may well underestimate the prevalence of GID; in any case, the number of adult transsexuals is small – one estimate from the Netherlands suggested a prevalence of 1 in 11,000 men and 1 in 30,400 women (Bakker, van Kesteren, Gooren, & Bezemer, 1993). More liberal estimates of prevalence can be judged from studies of children in whom specific cross-gender behaviors have been assessed. For example, the standardization study of the Child Behavior Checklist (CBCL; Achenbach & Edelbrock, 1983), a parent-report questionnaire of behavior problems, includes two items that pertain to cross-gender identification: “behaves like opposite sex” and “wishes to be of opposite sex.” In the standardization study, endorsement of both items was more common for girls than for boys, regardless of age and clinical status (referred versus non-referred). Among non-referred boys (ages 4–11 years), 3.8% received a rating of 1 (somewhat true) and 1.0% received a rating of 2 (very true) for the item “behaves like opposite sex,” but only 1.0% received a rating of 1 and 0.0% received a rating of 2 for the item “wishes to be of opposite sex” (Zucker, Bradley, & Sanikhani, 1997a). The comparable percentages among nonreferred girls were 8.3%, 2.3%, 2.5% and 1.0%, respectively. Thus, there appears to be a sex difference in the occurrence of mild displays of cross-gender behavior, but not with regard to more extreme cross-gender behavior. These findings were largely replicated in a large-scale study of Dutch twins at ages 7 and 10 (van Beijsterveldt, Hudziak, & Boomsma, 2006). Sex Differences in Referral Rates Among children between the ages of 3 and 12 years, boys are referred clinically more often than girls for concerns regarding gender identity. From one specialty clinic in Toronto, Canada, Cohen-Kettenis, Owen, Kaijser, Bradley, and Zucker (2003) reported a sex ratio of 5.75:1 (n = 358) of boys to girls based on consecutive referrals 1975–2000. Comparative data from the sole gender identity clinic for children in Utrecht, the Netherlands, also favored referral of boys over girls (sex ratio of 2.93:1, n = 130). The skewed sex ratio favoring boys may reflect bona fide sex differences in prevalence, but social factors may also have a role. Parents, teachers and peers are less tolerant of crossgender behavior in boys than in girls, which might result in a sex-differential in clinical referral (Zucker & Bradley, 1995). In support of this viewpoint, two studies found that girls may need to display more cross-gender behavior than boys before a referral is initiated (Cohen-Kettenis, Owen, Kaijser et al., 2003; Zucker, Bradley, & Sanikhani, 1997a). In both the Toronto and Utrecht clinics, girls were referred at a slightly older age than boys (means 8.1 versus 7.3 years, respectively), despite the fact that the girls showed, on average, higher levels of cross-gender behavior than the boys (CohenKettenis, Owen, Kaijser et al., 2003). However, the sexes did not differ in the percentage who met the complete DSM criteria for GID; thus, there was no evidence for a sex difference in false positive referrals. Another factor that could affect sex differences in referral rates pertains to the relative salience of cross-gender behavior Gender Identity and Sexual Disorders 52 Kenneth J. Zucker and Michael C. Seto 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 864 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

in boys versus girls. For behaviors showing significant betweensex variation, it is almost always the case that girls are more likely to engage in masculine behaviors than boys are likely to engage in feminine behaviors (Zucker, 2005a). Thus, the base rates for cross-gender behavior, at least within the range of normative variation, may well differ between the sexes. Diagnosis and Assessment DSM-IV Diagnosis of Gender Identity Disorder In DSM-IV-TR (American Psychiatric Association, 2000) there were some changes in the conceptualization of GID and in the diagnostic criteria. The DSM-IV Subcommittee on Gender Identity Disorders (Bradley, Blanchard, Coates et al., 1991) took the position that the DSM-III-R diagnoses of gender identity disorder of childhood, transsexualism and gender identity disorder of adolescence or adulthood, non-transsexual type were not qualitatively distinct disorders, but reflected differences in both developmental and severity parameters. As a result, the DSM-IV Subcommittee recommended one overarching diagnosis, gender identity disorder, that could be used, with appropriate variations in criteria, across the life cycle. In the DSM-IV, there are three criteria required for the diagnosis and there is also one exclusion criterion. The Point A criteria reflect the child’s cross-gender identification, indexed by five behavioral characteristics, of which at least four must be present. The Point B criteria reflect the child’s rejection of his or her anatomic status and/or rejection of same-sex stereotypical activities and behaviors. Point D specifies that the “disturbance . . . causes clinically significant distress or impairment in social, occupational, or other important areas of functioning.” Point C is an exclusion criterion and pertains to the presence of a physical intersex condition (when the clinical signs of GID are present in individuals with a physical intersex condition, the DSM-IV specifies that the residual diagnosis, Gender Identity Disorder Not Otherwise Specified, can be used). Reliability and Validity The clinical research literature has paid little attention to reliability of diagnosis for GID. One study showed that, for children, clinicians can reliably make the diagnosis (Zucker, Finegan, Doering, & Bradley, 1984) but, to our knowledge, no study has evaluated the reliability of the diagnosis for adolescents (for further discussion see Zucker, 2006a). There is a much more extensive literature on the discriminant validity of the GID diagnosis. Over the past 30-plus years, a variety of measurement approaches have been developed to assess the sex-typed behavior in children referred clinically for GID: observation of sex-typed behavior in free play tasks, on semi-projective or projective tasks and on a structured Gender Identity Interview schedule. In addition, several parent-report questionnaires pertaining to sex-typed behavior have been developed. In this line of research, several comparison groups have typically been utilized: siblings of GID probands, clinical controls and non-referred (or “normal”) controls (for a summary and review of measures see Zucker, 1992, 2005a). These studies have demonstrated strong evidence for the discriminant validity of the various measures, with good evidence for sensitivity and specificity. Many of these measures can be used to complement the ordinary clinical assessment involving children and their parents. Gender Identity Disorder as a Disorder? In psychiatric nosology, it is common for there to be debate regarding the number of symptoms required for a diagnosis, the exact wording of indicators, validational evidence and so on. Although this has occurred for GID (Langer & Martin, 2004; Wilson, Griffin, & Wren, 2002; cf. Zucker, 2006b), the more common debate is over the general legitimacy of GID as a disorder (Isay, 1997). Critics of the GID diagnosis have made several claims: 1 GID is nothing more than normal variation, albeit extreme, in gender-related behavior (Hill, Rozanski, Carfagnini, & Willoughby, 2007); 2 Children with GID show little evidence of distress and/or impairment and, if they do, it is not inherent to the condition, but merely a reaction to social disapproval (Bartlett, Vasey, & Bukowski, 2000); and 3 Because GID in children is strongly predictive of a homosexual sexual orientation in adulthood, its inclusion in DSM-III was nothing more than a veiled backdoor maneuver to prevent later homosexuality. As this last claim has been addressed elsewhere (Zucker & Spitzer, 2005), we will appraise here only the first two criticisms and suggest ways in which the diagnostic criteria for GID might be reformed in DSM-V. GID as Normal Variation in Gender-Related Behavior Because of the putative conflation of gender identity dysphoria and gender role behavior, particularly in the Point A criterion, one could argue that reform of the criteria is called for. One relatively simple reform would be to raise the threshold for the Point A criterion and require the presence of all five indicators. Thus, it would be necessary for the child to systematically verbalize the wish to be of the opposite sex in order for the Point A criterion to be met. A more radical reform would be to relegate the indicators of extreme cross-gender role behavior (A2–A5) to the text description of GID, with an explanation that they may not be sufficient, on their own, to indicate the presence of gender dysphoria. The diagnostic criteria could then be modified by the inclusion of multiple indicators of gender dysphoria. To explore this, new psychometric studies would be required: factor analysis can be used to establish the coherence of these putative indicators of gender dysphoria; this would be followed by tests of discriminant validity; and then an empirical determination of the appropriate cutpoint for high sensitivity and specificity. If such an enterprise proved successful, perhaps this would allay concerns that children with extreme gender non-conformity, but who are not truly gender dysphoric, are being inappropriately diagnosed. GENDER IDENTITY AND SEXUAL DISORDERS 865 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 865

Distress and Impairment The DSM-III-R did not provide guidelines regarding the assessment of distress in the Point A criterion (“persistent and intense distress” about being a boy or a girl) or the ways in which it might be distinct from other operationalized components in the criteria (i.e., the “desire” to be of the other sex; Zucker, 1992). In the DSM-IV, this problem persists, except that it is now located in Point D, and there is the additional problem of defining impairment. Some critics have argued that the distress and/or impairment that clinicians observe in people with GID are merely a response to the reaction of others (Bartlett, Vasey, & Bukowski, 2000). Consistent with this critique, Stoller (1968) argued that GID in boys was ego-syntonic and that they became distressed only when their cross-gender fantasies and behaviors were interfered with. However, Stoller’s conceptualization was a psychodynamic formulation: he believed that GID was ego-syntonic because the familial conditions that produced it were systemically syntonic. Thus, it is likely that Stoller would have argued that the DSM’s conceptualization of distress was, and is, too narrow. Other theorists have argued otherwise. For example, Coates and Person (1985) claimed that GID was a “solution” to specific forms of psychopathology in the child, particularly separation anxiety and “annihilation” anxiety, which were induced by familial psychopathology. It is conceivable that both the “primary” and “secondary” views are correct or that one or the other better fits individual cases. If one considers the developmental adolescent or adult “endstate” of GID (i.e., the strong desire to align the body via contrasex hormones and sex-reassignment surgery: in females, mastectomy, phalloplasty; in males, penectomy/castration, vaginoplasty), it is difficult to argue that cross-gender feelings and behaviors simply constitute normative variation or do not constitute an example of impairment. The required physical interventions are simply too radical to be thought about otherwise. One indication of impairment is that children with GID have trouble with basic cognitive concepts concerning gender. They are more likely than other children to misclassify their own gender (Zucker, Bradley, Lowry Sullivan et al., 1993), and they appear to have a “developmental lag” in the acquisition of gender constancy (Zucker, Bradley, Kuksis et al., 1999). Given the ubiquity of gender as a social category, this may well lead to affective confusion in self-representation and in social interactions. As noted in the next section, there is evidence that children with GID have poor peer relations and more general behavioral problems, which are possible indices of impairment. Associated Psychopathology The most systematic information on general behavior problems in children with GID comes from parent-report data using the CBCL. Clinic-referred boys and girls with GID show, on average, significantly more general behavior problems than do their siblings and non-referred children (Cohen-Kettenis, Owen, Kaijser et al., 2003; Zucker & Bradley, 1995). On the CBCL, boys with GID have a predominance of emotional difficulties whereas girls with GID do not (Cohen-Kettenis, Owen, Kaijser et al., 2003; Zucker & Bradley, 1995). Psychopathological problems increase with age in boys with GID (Cohen-Kettenis, Owen, Kaijser et al., 2003; Zucker & Bradley, 1995). This could be influenced by peer ostracism. Children with GID experience significant difficulties within the peer group (Cohen-Kettenis, Owen, Kaijser et al., 2003; Zucker, Bradley, & Sanikhani et al., 1997a). Cohen-Kettenis, Owen, Kaijser et al. (2003) found that poor peer relations were the strongest predictor of CBCL behavior problems in both boys and girls with GID, accounting for 32% and 24% of the variance, respectively. Thus, social ostracism may be a potential mediator between cross-gender behavior and behavior problems. It seems likely that marked cross-gender behavior is particularly salient in eliciting negative reactions from peers. Although we have shown that poor peer relations is an important correlate of general behavior problems in children with GID, this is not meant to imply that it is the only source of these difficulties. Other research has shown that CBCLmeasured problems in boys with GID are associated with a composite index of maternal psychopathology, which may reflect generic non-specific familial risk factors in producing psychopathology in general (Zucker & Bradley, 1995) and the predominance of emotional psychopathology may reflect familial risk for affective disorders and temperamental features of the boys (Marantz & Coates, 1991). Thus, it is likely that there are both specific and general risk factors involved in the general psychological problems of children with GID. Etiological Influences Biological Mechanisms The role of prenatal sex hormones in causing the postnatal display of various sex-dimorphic behaviors has been the focus of detailed experimental animal research for almost 50 years (Baum, 2006). In humans, the influence of normative sex differences in prenatal sex hormone exposure is hard to disentangle from normative sex-of-rearing effects; accordingly, researchers have relied on children born with various disorders of sex development (DSDs), previously referred to as physical intersex conditions, to separate the relative influence of biological and psychosocial processes on psychosexual differentiation. This research provides considerable evidence for an influence of prenatal sex hormones on postnatal sex-dimorphic behavior. For example, chromosomal females with congenital adrenal hyperplasia (CAH), who are exposed to sex-atypical levels of prenatal androgen, show more masculinized gender role behavior in childhood and adolescence and a higher rate of a bisexual and/or homosexual sexual orientation in adulthood (for review see Cohen-Bendahan, van de Beek, & Berenbaum, 2005; Hines, 2004; Zucker, 1999). Nevertheless, most females with CAH show a sex-typical gender identity (e.g., Dessens, Slijper, & Drop, 2005; Meyer-Bahlburg, Dolezal, Baker et al., 2004). It appears therefore that prenatal sex hormones may have a relatively stronger impact on some components CHAPTER 52 866 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 866

of psychosexual differentiation (e.g., gender role behavior and sexual orientation) than on others (e.g., gender identity). One of the most dramatic cases in this field was of a biologically normal male (one of a pair of identical twins), whose penis was accidentally ablated during a circumcision at the age of 7 months. Known in the literature as the John-Joan case (and then as David Reimer, the patient’s name), this infant was gender-reassigned to female at the age of 17 months and gonadectomy was performed at age 22 months. Early reports indicated that the patient had differentiated a female gender identity (suggesting a crucial role for socialization influences on gender identity), although she was described as tomboyish in comportment (Money, 1975). However, a longer-term follow-up reported that the patient experienced profound conflict as a girl and, in early adolescence, “returned” to living as a boy and, in adulthood, married a woman and adopted her three children (Colapinto, 2000; Diamond & Sigmundson, 1997). Sadly, Mr. Reimer committed suicide in 2004 at the age of 38 (Agrell, 2004). His death followed the suicide of his cotwin 2 years earlier (Jang & Smith, 2004). The David Reimer case has been used to highlight the importance of biological factors on psychosexual differentiation. Indeed, some have argued that a “normal” male sexual biology cannot be countered by socialization efforts to raise a male as a girl (even with gonadectomy, the use of female sex hormones to induce a feminizing puberty, feminizing surgery on the genitalia, etc.) and that the Reimer case was “proof” for the role of prenatal androgen “imprinting” of a male gender identity. However, a counter to this position comes from another case of penile ablation reported on by Bradley, Oliver, Chernick, and Zucker (1998). In this case, the circumcision accident occurred at the age of 2 months and the patient was genderreassigned, along with gonadectomy, at the age of 7 months. In adolescence, the patient began feminizing hormonal and surgical treatment. A follow-up at the age of 26 years showed that the patient had a female-typical gender identity; however, her gender role interests were conventionally masculine and she reported a bisexual sexual orientation in both fantasy and behavior. Although the Reimer case has had a profound impact on the field, some authors have cautioned against extrapolating too much from it (Meyer-Bahlburg, 2005) and noted that the case has raised as many questions as answers. When it comes to children with GID, one must appreciate that they invariably do not show signs of a DSD, which would appear to rule out an etiology that involves a marked prenatal hormonal anomaly (Meyer-Bahlburg, 2005). Thus, the search for biological influences on the development of GID must focus on factors that do not affect the configuration of the external genitalia. Prenatal hormonal influences may have a role, but there would have to be some kind of brain–genital dissociation in their impact (e.g., perhaps resulting from timing effects; Gooren, 2006; Goy, Bercovitch, & McBrair, 1988). Given that there are no postmortem studies on brain neuroanatomy in GID children or youth, research has focused either on indirect markers of putative prenatal hormonal influences or on other biological factors. A selective review of relevant studies is provided here. Molecular and Behavior Genetics There have been no molecular genetic studies of GID, but several behavioral genetic studies have shown that the liability for cross-gender behavior in the general population has a strong heritable component (van Beijsterveldt, Hudziak, & Boomsma et al., 2006). Another twin study also identified strong shared environmental influences (Knafo, Iervolino, & Plomin, 2005). Such environmental influences could, of course, pertain to non-genetic biological factors but could also involve postnatal psychosocial factors, some of which may be specific to twins. In any case, it should be recognized that these studies have not identified the specific genetic and environmental factors, or the gene × environmental interactions, underlying the liability to cross-gender behavior. That genetic factors do not account for all of the variance in the liability to cross-gender behavior is demonstrated quite clearly from detailed clinical case reports of identical twins discordant for GID (Segal, 2006). Activity Level Activity level (AL) is a dimension of temperament with some evidence for a genetic basis and possibly prenatal hormonal influences (Campbell & Eaton, 1999). In general, boys have a higher AL than girls. Parents report lower levels of AL in boys with GID compared to controls (Bates, Bentler, & Thompson, 1979; Zucker & Bradley, 1995). Zucker and Bradley also found that girls with GID had a higher AL than control girls. It is possible that a sex-atypical AL is a temperamental factor that predisposes to the development of GID. For example, a low-active boy with GID may find the typical play behavior of other boys to be incompatible with his own behavioral style, which might make it difficult for him to integrate successfully into a male peer group. Sibling Sex Ratio and Birth Order Boys with GID have an excess of brothers to sisters (sibling sex ratio) and a later birth order. Some additional evidence shows that boys with GID are born later primarily in relation to the number of older brothers, but not sisters (Blanchard, Zucker, Bradley, & Hume, 1995; Zucker, Green, Coates et al., 1997b). In the Blanchard et al. study, clinical control boys showed no evidence for an altered sibling sex ratio or a late birth order. These findings mesh nicely with studies of adult males with GID with a homosexual sexual orientation, who also have an excess of brothers to sisters and a later birth order (Blanchard, 2001). One biological explanation that has been offered to account for these results pertains to maternal immune reactions during pregnancy. The male fetus is experienced by the mother as more “foreign” (antigenic) than the female fetus. Accordingly, it has been suggested that one consequence of this is that the mother produces antibodies that have the consequence of demasculinizing or feminizing the male fetus, but no GENDER IDENTITY AND SEXUAL DISORDERS 867 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 867

corresponding masculinizing or defeminizing of the female fetus (Blanchard, 2001). This model would predict that males born later in a sibship might be more affected, because the mother’s antigenicity increases with each successive male pregnancy, which is consistent with the empirical evidence on sibling sex ratio and birth order among GID probands. At present, this proposed mechanism has not been formally tested in humans. Psychosocial Mechanisms Psychosocial factors, to truly merit causal status, must be shown to influence the emergence of marked cross-gender behavior in the first few years of life. Otherwise, such factors would be better conceptualized as perpetuating rather than predisposing. Sex Assignment at Birth In some DSDs, sex assignment is delayed and, on occasion, changed from the initial sex assignment. It has been argued that prolonged delay or uncertainty about the child’s “true” sex can contribute to gender identity conflict in affected individuals (Meyer-Bahlburg, Gruen, New et al., 1996). Because most children with GID do not have a co-occurring DSD, this does not appear to be a relevant psychosocial factor. Prenatal Gender Preference It is common for parents to express a prenatal preference for the gender of the expected baby. However, there is no evidence that parents of children with GID are more likely than control parents to report having had a desire for a child of the opposite sex (Zucker, Green, Garofano et al., 1994). Social Reinforcement of Cross-Gender Behavior The parental response to early cross-gender behavior in children with GID is typically neutral (tolerance) or even encouraging (Mitchell, 1991; Roberts, Green, Williams, & Goodman, 1987; Zucker & Bradley, 1995). Green (1974) concluded that “What comes closest so far to being a necessary variable is that, as any feminine behavior begins to emerge, there is no discouragement of that behavior by the child’s principal caretaker” (p. 238, italics in original). The reasons why parents might tolerate, if not encourage, early cross-gender behaviors appear to be quite diverse. Some report being influenced by ideas regarding non-sexist child rearing. In others, the antecedents seem to be rooted in pervasive conflict that revolves around gender issues, including what Zucker and Bradley (1995) characterized as pathologic gender mourning. General Psychopathology The role of maternal psychopathology in the genesis and perpetuation of GID has received a great deal of clinical and theoretical attention but, unfortunately, only limited empirical evaluation. All the available empirical studies have been confined to the mothers of boys with GID – comparable studies are not available regarding the mothers of girls with GID. Marantz and Coates (1991) found that the mothers of boys with GID showed more signs of psychopathology than did the mothers of demographically matched normal boys, including more pathologic ratings on the Diagnostic Interview for Borderline Patients (Gunderson, Kolb, & Austin, 1981) and more symptoms of depression on the Beck Depression Inventory. In a similarly designed study, Zucker (2005b) reported that almost half of mothers of boys with GID (n = 245) met criteria for two or more diagnoses on the Diagnostic Interview Schedule, of whom over 30% had three or more diagnoses. The most common diagnoses were major depressive episode and recurrent major depression. Although rates of emotional distress and psychiatric impairment are elevated in the mothers of boys with GID, these maternal characteristics are not unique to the mothers of boys with GID, but common to the mothers of clinic-referred boys in general. Accordingly, maternal emotional distress and/or impairment functions, at most, only as a non-specific risk factor in the development of GID. The role of paternal influences in the genesis and perpetuation of GID has also received a great deal of clinical and theoretical attention but, again, only limited empirical evaluation and only for boys with GID. One account implicates the father’s role by virtue of his absence from the family matrix. Across 10 samples of boys with GID, the rate of father absence was 34.5% (Zucker & Bradley, 1995). It is unlikely that this rate would differ significantly from the rate found in clinical populations in general, if not the general population. Green (1987) found that paternal separations occurred earlier in the families of boys with GID than normal control boys; therefore it is possible that timing is an additional variable to consider. Green also found that the fathers of boys with GID (both father-present and father-absent) recalled spending less time with their sons than did the fathers of control boys during preschool years and at the time of assessment. The inclusion of a clinical control group would be helpful in gauging the specificity of this finding. Long-Term Follow-Up The “natural history” of GID in children has been confined to clinic-referred samples. Because there are no natural history studies of such children taken from epidemiological samples, one has to be cautious in drawing any broad generalizations from this literature. Follow-up Studies of Boys Green’s (1987) study constitutes the most comprehensive longterm follow-up of behaviorally feminine boys, the majority of whom would likely have met DSM criteria for GID. His study contained 66 feminine and 56 control boys, assessed initially at a mean age of 7.1 years (range, 4–12). Forty-four feminine boys and 30 control boys were available for followup at a mean age of 18.9 years (range, 14–24). The majority of the boys were not in therapy between assessment and follow-up. Sexual orientation in fantasy and behavior was assessed by means of a semi-structured interview. Of the previously feminine boys, 75–80% were either bisexual or homosexual at follow-up versus 0–4% of the control boys. Only one CHAPTER 52 868 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 868

youth, at the age of 18 years, was gender-dysphoric to the extent of considering sex-reassignment surgery. Data from six other follow-up reports on 55 boys with GID were summarized by Zucker and Bradley (1995). At followup (range, 13–26 years), if one excludes 13 uncertain cases, then 27 (64.3%) of the remaining 42 cases had “atypical” (i.e., homosexual, transsexual or transvestitic) outcomes. In these studies, the percentage of boys who showed persistent GID was higher than that reported by Green (11.9% vs. 2.2%, respectively), but the percentage who were homosexual (62.1%) was somewhat lower. Zucker and Bradley (1995) reported preliminary follow-up data, at mean age 16 years, on their own sample of boys first seen in childhood (mean age at assessment, 8.2 years; range, 3–12). Of the 40 boys, 8 (20%) were classified as genderdysphoric at follow-up. The remaining 80% had a “normal” gender identity. Regarding sexual orientation in fantasy, 20 (50%) were classified as heterosexual, 17 (42.5%) were classified as bisexual/homosexual and 3 (7.5%) were classified as “asexual” (i.e., they did not report any sexual fantasies). Regarding sexual orientation in behavior, 9 (22.5%) were classified as heterosexual, 11 (27.5%) were classified as bisexual/ homosexual and 20 (50.0%) were classified as “asexual” (i.e., they did not report any interpersonal sexual experiences). Cohen-Kettenis (2001) reported preliminary data on a sample of 56 boys first seen in childhood (mean age at assessment, 9 years; range, 6–12) and who had now reached adolescence. Of these, 9 (16.1%) requested sex-reassignment, and all 9 had a homosexual sexual orientation (P. T. CohenKettenis, personal communication, February 1, 2003). Thus, the rate of GID persistence, at least into adolescence, was higher than that reported by Green (1987) and comparable with the rate obtained by Zucker and Bradley (1995). In taking stock of these outcome data, Green’s study shows that boys with GID were disproportionately, and substantially, more likely than the control boys to differentiate a bisexual/ homosexual sexual orientation. The other follow-up studies yielded somewhat lower estimates of a bisexual/homosexual sexual orientation. In this regard, at least one caveat is in order. In the Zucker and Bradley follow-up, the boys were somewhat younger than were the boys followed-up by Green, so their lower rate of a bisexual/homosexual sexual orientation outcome should be interpreted cautiously, especially as one would expect that social desirability considerations would lead to underreporting of atypical sexual orientation. However, even these lower rates of a bisexual/homosexual sexual orientation are substantially higher than the currently accepted base rate of about 2–3% of a homosexual sexual orientation in men (Laumann, Gagnon, Michael, & Michaels, 1994). A more substantive difference between Green’s study and the other follow-up reports pertains to the persistence of gender dysphoria. Both Zucker and Bradley, and CohenKettenis, for example, found higher rates of persistence than Green. At present, the explanations for this are unclear. One possibility pertains to sampling differences. Green’s study was carried out in the context of an advertised research study whereas the Zucker and Bradley and Cohen-Kettenis samples were clinic-referred. Thus, it is conceivable that their samples may have included more extreme cases of childhood GID than the sample ascertained by Green. Because the Toronto and Dutch groups now have substantially larger samples on which to complete outcome studies, it should be possible to carry out within-group analyses to identify predictor variables (e.g., with regard to persistent GID versus desistent GID). Follow-up Studies of Girls Unfortunately, the long-term follow-up of girls with GID remains very patchy. In part, this reflects the comparatively lower rate of referred girls than referred boys with GID in child samples (Cohen-Kettenis, Owen, Kaijser et al., 2003). Since the last edition of this volume, the first systematic follow-up of girls with GID has become available. Drummond, Bradley, Badali-Peterson, and Zucker (in press) evaluated 25 girls, originally assessed in our clinic at a mean age of 8.8 years (range, 3–12), at a follow-up mean age of 23.2 years (range, 15–36). Of these 25 girls, three (12%) had persistent GID (at follow-up ages of 17, 21 and 23 years, respectively), two of whom had a homosexual sexual orientation and the third was asexual. The remaining 22 (88%) girls had a “normal” gender identity. Regarding sexual orientation in fantasy (Kinsey ratings) for the 12 months preceding the follow-up assessment, 15 (60%) girls were classified as exclusively heterosexual, 8 (32%) were classified as bisexual/homosexual and 2 (8%) were classified as “asexual” (i.e., they did not report any sexual fantasies). Regarding sexual orientation in behavior, 11 (44%) girls were classified as exclusively heterosexual, 6 (24%) were classified as bisexual/homosexual and 8 (32%) were classified as “asexual” (i.e., they did not report any interpersonal sexual experiences). Although these data are preliminary, it appears that there is a range of outcomes, but it is clear that the rates of GID and a bisexual/homosexual sexual orientation without cooccurring gender dysphoria are likely to be higher than the base rates of these two aspects of psychosexual differentiation in an unselected population of women. Using Bakker, van Kesteren, Gooren et al.s’ (1993) estimation of GID prevalence in females, the odds of persistent gender dysphoria in Drummond, Bradley, Badali-Peterson et al. was 4084 times the odds of gender dysphoria in the general population. Using prevalence estimates of bisexuality/homosexuality in fantasy among biological females (anywhere between 2% and 5%), the odds of reporting bisexuality/homosexuality in fantasy were 8.9–23.1 times higher than in the general population. The odds of reporting bisexuality/homosexuality in behavior were 6.7–15.5 times higher than in the general population. In another study, Cohen-Kettenis (2001) reported preliminary data on a sample of 18 girls first seen in childhood (mean age at assessment, 9 years; range, 6–12) and who had now reached adolescence. Of these, 8 (44.4%) requested sex-reassignment and all had a homosexual sexual orientation (P. T. CohenKettenis, personal communication, February 1, 2003). Thus, the GENDER IDENTITY AND SEXUAL DISORDERS 869 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 869

rate of GID persistence, at least into adolescence, was high (and much higher than the rate of persistence for her boys with GID). Persistence and Desistance in a Comparative-Developmental Perspective A key challenge for developmental theories of psychosexual differentiation is to account for the disjunction between retrospective and prospective data with regard to GID persistence: it is clear that only a minority of children followed prospectively show a persistence of GID into adolescence and young adulthood. Regarding children with GID then, we need to understand why, for the majority, the disorder apparently remits by adolescence, if not earlier. One possible explanation concerns referral bias. Green (1974) argued that children with GID who are referred for clinical assessment (and then, in some cases, therapy) may come from families in which there is more concern than is the case for adolescents and adults, the majority of whom did not receive a clinical evaluation and treatment during childhood. Thus, a clinical evaluation and subsequent therapeutic intervention during childhood may alter the natural history of GID. Of course, this is only one account of the disjunction and there may well be additional factors that might distinguish those children who are more strongly at risk for the disorder’s continuation from those who are not. One such explanation pertains to the concepts of developmental malleability and plasticity. It is possible that gender identity shows relative malleability during childhood, with a gradual narrowing of plasticity as the gendered sense of self consolidates as one approaches adolescence. Some support for this idea comes from follow-up studies of adolescents with GID, who appear to show a much higher rate of GID persistence as they are followed into young adulthood. The best data on long-term outcome on adolescents come from the Dutch group. Cohen-Kettenis and van Goozen (1997) reported that 22 (66.7%) of 33 adolescents went on to receive sexual reassignment surgery (SRS). At initial assessment, the mean age of the 22 adolescents who received SRS was 17.5 years (range, 15–20). Of the 11 who did not receive SRS, 8 were not recommended for it because they were not diagnosed with transsexualism (presumably the DSM-IV diagnosis of GID); the three remaining patients were given a diagnosis of transsexualism but the “real-life test” (i.e., living for a time as the opposite sex prior to the institution of contrasex hormonal treatment and surgery) was postponed because of severe concurrent psychopathology and/or adverse social circumstances. Similar findings were reported by Smith, van Goozen, and Cohen-Kettenis (2002). These data suggest a very high rate of GID persistence, which is eventually treated by SRS. Therapeutics Ethical Considerations Consider the following clinical scenarios: 1 A mother of a 4-year-old boy contacts a well-known clinic that specializes in sexuality throughout the lifespan. She describes behaviors consistent with the DSM diagnosis of GID. A clinician tells the mother: “Well, we don’t consider ‘it’ to be a problem.” The mother then contacts a specialty clinic for children and adolescents and asks “What should I do?” 2 A mother of a 4-year-old boy contacts a well-known clinic that specializes in gender identity problems. She describes behaviors consistent with the DSM diagnosis of GID. She says that she would like her child treated so he does not grow up to be gay. She also worries that her child will be ostracized within the peer group because of his pervasive cross-gender behavior. What should the clinician say to this mother? 3 The parents of a 6-year-old boy (somatically male) conclude that their son is really a girl, so they seek the help of an attorney to institute a legal name change (from Zachary to Aurora) and inform the school principal that their son will attend school as a girl. The local child protection agency is notified and the child removed from his parents’ care (Cloud, 2000). If a clinician was asked to evaluate the situation, what would be in the best interests of the child and family? 4 A 15-year-old adolescent girl with GID asks to be seen for treatment. She would like cross-sex hormones and a mastectomy immediately. If refused, the patient threatens to kill herself. What should the clinician do? 5 A 16-year-old adolescent boy with GID asks for SRS. He states that he is sexually attracted to biological males and wants a sex-change because that will make his sexual feelings for males “normal.” He states that homosexuality is against his religious beliefs and is taboo in his culture of origin. What should the clinician do? Any contemporary developmental clinician responsible for the therapeutic care of children and adolescents with GID will quickly be introduced to these kinds of complex social and ethical scenarios and will have to think them through carefully in formulating a therapeutic plan. Treatment of Children In summarizing the treatment literature on GID, one will find not a single randomized controlled treatment trial. However, there have been some treatment effectiveness studies, although much is lacking in these investigations. In general, the practitioner must rely largely on the “clinical wisdom” that has accumulated in the case report literature and the conceptual underpinnings that inform the various approaches to intervention. In an era that emphasizes evidence-based therapeutics, the relatively meager empirical database is a source of frustration for many clinicians. Behavioral Therapy There are 13 published single-case reports that employed a behavioral therapy approach to the treatment of GID in children (for review see Zucker, 1985). One type of intervention employed has been termed differential social attention or social reinforcement. This type of intervention has been applied in clinic settings, particularly to sex-typed play behaviors. The therapist first establishes with baseline measures that the child (either when alone or in the presence of a non-interacting adult) prefers playing with cross-sex toys or dress-up apparel rather CHAPTER 52 870 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 870

than same-sex toys or dress-up apparel. A parent or stranger is then introduced into the playroom and instructed to attend to the child’s same-sex play (e.g., by looking, smiling and verbal praise) and to ignore the child’s cross-sex play (e.g., by looking away and pretending to read). Such adult responses seem to elicit rather sharp changes in play behavior. There have been two main limitations to the use of social attention or reinforcement in treating cross-gender behavior. First, at least some of the children studied reverted to cross-sex play patterns in the adult’s absence or in other environments, such as the home; second, there was little generalization to untreated cross-sex behaviors (Rekers, 1975). Such problems have led behavior therapists to seek more effective strategies of promoting generalization. One such strategy, self-regulation or self-monitoring, has the child reinforce himself or herself when engaging in a sex-typical behavior. Although self-monitoring also resulted in substantial decreases in cross-sex play, the evidence is weak that generalization is better promoted by self-regulation than by social attention (Zucker, 1985). The behavior-therapy literature has produced no new case reports for over 20 years although its principles are often used in broader treatment approaches that involve the parents. This void is rather curious, because contemporary behavioral approaches, such as cognitive–behavioral therapy, are now used so widely with other disorders. Behavior therapy with an emphasis on the child’s cognitive structures regarding gender could be an interesting and novel approach to treatment. There is now a fairly large literature on the development of cognitive gender-schemas in typically developing children (Martin, Ruble, & Szkrybalo, 2002). It is possible that children with GID have more elaborately developed cross-gender schemas than same-gender schemas and that more positive affective appraisals are differentiated for the former than for the latter (e.g., in boys, “girls get to wear prettier clothes” versus “boys are too rough”). A cognitive approach to treatment might help children with GID to develop more flexible and realistic notions about gender-related traits (e.g., “boys can wear pretty cool clothes too” or “there are lots of boys who don’t like to be rough”), which may result in more positive gender feelings about being a boy or being a girl. Psychotherapy There is a large case report literature on the treatment of children with GID using psychoanalysis, psychoanalytic psychotherapy or psychotherapy (for references see Zucker, 2001, 2007). The psychoanalytic treatment literature is more diverse than the behavior therapy literature, including varied theoretical approaches to understanding the putative etiology of GID (e.g., classic object relations and self-psychology). Unfortunately, this literature has not developed very far in providing systematic documentation of therapeutic effectiveness. Apart from the general developmental perspective inherent in a psychoanalytic understanding of psychopathology, one might add to this a gender-specific perspective on development (Martin, Ruble, & Szkrybalo, 2002). Many developmentalists note that the first signs of normative gender development appear during the toddler years, including the ability to correctly self-label oneself as a boy or a girl. Money, Hampson, and Hampson (1957) postulated a sensitive period for gender identity formation, which suggests a period of time in which there is a greater malleability or plasticity in the direction that gender identity can move. Thus, early gender identity formation intersects quite neatly with analytic views on the early development of the sense of self in more global terms. It is likely therefore that the putative pathogenic mechanisms identified in the development of GID are likely to have a greater impact only if they occur during the alleged sensitive period for gender identity formation. Treatment of the Parents Two rationales have been offered for parental involvement in treatment. The first emphasizes the hypothesized role of parental dynamics and psychopathology in the genesis or maintenance of the disorder. From this perspective, individual therapy with the child will probably proceed more smoothly and quickly if the parents are able to gain some insight into their own contribution to their child’s difficulties. Many clinicians who have worked extensively with gender-disturbed children subscribe to this rationale (Newman, 1976; Zucker, 2001, 2007). In this context, a treatment plan requires as much of an assessment of the parents as it does of the child, as is the case with many child psychiatric disorders. Assessment of psychopathology and the marital relationship in the parents of children with GID reveals great variability in adaptive functioning, which may well prove to be a prognostic factor. The second rationale is that parents will benefit from regular formalized contact with the therapist to discuss day-today management issues that arise in carrying out the overall therapeutic plan. Work with parents can focus on the setting of limits with regard to cross-gender behavior, such as crossdressing, cross-gender role and fantasy play, and cross-gender toy play and, at the same time, attempting to provide alternative activities (e.g., encouragement of same-sex peer relations and involvement in more gender-typical and neutral activities). In addition, parents can work on conveying to their child that they are trying to help him or her feel better about being a boy or a girl and that they want their child to be happier in this regard. Although many contemporary clinicians have stressed the important role of working with the parents of children with GID, there is scanty empirical evidence on efficacy. The most relevant study found some evidence that parental involvement in therapy was significantly correlated with a greater degree of behavioral change in the child at a 1-year follow-up, but this study did not make random assignment to different treatment protocols, so one has to interpret the findings with caution (Zucker, Bradley, Doering, & Lozinski, 1985). Supportive Treatments In the past few years, clinicians critical of conceptualizing marked cross-gender behavior in children as a disorder have GENDER IDENTITY AND SEXUAL DISORDERS 871 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 871

provided a dissenting perspective to the treatment approaches described so far (Menvielle & Tuerk, 2002; Menvielle, Tuerk, & Perrin, 2005). These clinicians conceptualize GID or pervasive gender-variant behavior from an essentialist perspective (i.e., that it is fully constitutional or congenital in origin) and are skeptical about the role of psychosocial or psychodynamic factors. For example, Menvielle and Tuerk (2002) noted that, while it might be helpful to set limits on pervasive cross-gender behaviors that may contribute to social ostracism, their primary treatment goal was “not at changing the children’s behavior, but helping parents to be supportive and to maximize opportunities for the children’s adjustment” (p. 2002). This perspective therefore appears to encourage some parents to take an approach that is rather different from the more “traditional” therapeutic models: there are now several examples reported on in the media in which parents of very young children enforce a social gender change (e.g., registering a biological male child in school as a girl; Brown, 2006; Santiago, 2006). Because comparative treatment approaches are not available, it is not possible to say whether or not this supportive or “crossgender affirming” approach will result in both short-term and long-term outcomes any different from the more traditional approaches to treatment. Treatment of Adolescents In adolescents with GID, there are three broad clinical issues that require evaluation: 1 Phenomenology pertaining to the GID itself; 2 Sexual orientation; and 3 Psychiatric comorbidity. Apart from the GID itself, gender-dysphoric adolescents with a childhood onset of cross-gender behavior typically have a homosexual orientation (i.e., they are attracted to members of their own “birth sex”). Some such adolescents may not report any sexual feelings, but follow-up will typically find the emergence of same-sex attractions. Thus, the clinician must evaluate simultaneously two dimensions of the patient’s psychosexual development: current gender identity and current sexual orientation. The psychotherapy treatment literature on adolescents with GID has been very poorly developed and is confined to a few case reports (Zucker, 2006a; Zucker & Bradley, 1995). In general, the prognosis for adolescents resolving the GID is more guarded than it is for children. From a clinical management point of view, two key issues need to be considered: 1 Some adolescents with GID are not particularly good candidates for therapy because of comorbid disorders and general life circumstances; 2 Some adolescents with GID have little interest in psychologically oriented treatment and are quite adamant about proceeding with hormonal and surgical sex reassignment. Prior to recommending hormonal and surgical interventions, many clinicians encourage adolescents with GID to consider alternatives to this invasive and expensive treatment. One area of inquiry can therefore explore the meaning behind the adolescent’s desire for sex reassignment and if there are viable alternative lifestyle adaptations. The most common area of exploration in this regard pertains to the patient’s sexual orientation. Some adolescents with GID recall that they always felt uncomfortable growing up as boys or as girls, but that the idea of “sex change” did not occur until they became aware of homoerotic attractions. For some of these youngsters, the idea that they might be gay or homosexual is abhorrent. For adolescents in whom the gender dysphoria appears chronic, there is considerable evidence that it interferes with general social adaptation, including general psychiatric impairment, conflicted family relations and dropping out of school. For these youngsters, the treating clinician can consider two main options: 1 Management until the adolescent turns 18 and can be referred to an adult gender identity clinic; or 2 “Early” institution of contrasex hormonal treatment. Gooren and Delemarre-van de Waal (1996) and CohenKettenis and van Goozen (1998) recommended that one option with gender-dysphoric adolescents is to prescribe pubertyblocking luteinizing hormone-release agonists (e.g., depot leuprolide or triptorelin) that make it easier to pass as the opposite sex. Thus, in male adolescents such medication can suppress the development of secondary sex characteristics, such as facial hair growth and voice deepening, which make it more difficult to pass in the female social role. Cohen-Kettenis and van Goozen (1997) and Smith, van Goozen, & CohenKettenis et al. (2002) reported that early cross-sex hormone treatment for adolescents under the age of 18 years, who were free of gross psychiatric comorbidity, facilitated the complex psychosexual and psychosocial transition to living as a member of the opposite sex and resulted in a lessening of the gender dysphoria. Although such early hormonal treatment is controversial (Beh & Diamond, 2005), it may well be the treatment of choice once the clinician is confident that other options have been exhausted (Cohen-Kettenis, 2005). Paraphilic Sexual Disorders Diagnosis Paraphilias can broadly be classified into those that involve atypical sexual targets or those that involve atypical sexual activities. Atypical sexual targets include sexually immature persons (pedophilia), non-human animals (zoophilia) and inanimate objects (fetishism); atypical activities include the infliction of pain or humiliation on another (sadism), experiencing pain or humiliation (masochism) or exposing one’s genitals to unsuspecting strangers (exhibitionism). A wide variety of paraphilias have been described in the clinical and research literatures, but most are rare. The most commonly seen paraphilias in clinical or correctional settings are those that involve illegal behavior if they are acted upon, such as pedophilia and the commission of sexual offences against children, or sadism and the commission of violent sexual offenses. The DSM-IV-TR lists a number of the more commonly known paraphilias, including pedophilia, sadism and fetishism; CHAPTER 52 872 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 872

the paraphilias listed in the 10th revision of the ICD-10 are quite similar in content (World Health Organization, 1997). For example, the ICD-10 defines pedophilia as “a sexual preference for children, usually of prepubertal or early pubertal age” while the DSM-IV-TR elaborates by defining sexual preference in terms of timeline and manifestation, and adding criteria regarding the age difference between the person and the child or children (the person is at least age 16 years and at least 5 years older than the child or children). Epidemiology Neither the incidence nor prevalence of paraphilias is known. Epidemiological surveys of the general population, with the specific questions that are needed to identify paraphilias – particularly questions about the persistence and intensity of sexual thoughts, fantasies, urges, arousal or behavior involving the paraphilic target or activity – have not yet been conducted. Almost all of the research on paraphilias has been conducted with clinical or correctional samples, and most of that work has involved adult sex offenders rather than adolescent sex offenders, or other minors. Surveys of convenience samples of young adults suggest that paraphilias are rare (Templeman & Stinnett, 1991). Researchers have not asked similar questions of adolescents. Assessment Interviews and Questionnaires In order to diagnose a paraphilic sexual disorder, a thorough sexual history is needed. Such histories are typically obtained through clinical interview. Respondents are asked questions about their sexual thoughts, interests and behaviors, especially with regard to atypical sexual targets or activities. Comprehensive interviews also include questions intended to help clinicians rule out other explanations for atypical sexual thoughts, urges, fantasies or behavior. For example, some individuals with obsessive-compulsive disorder may report being disturbed by thoughts about molesting a child (see, e.g., Gordon, 2002). The differential diagnosis is made by determining if the thoughts are associated with sexual arousal or pleasure and by inquiring about other symptoms of obsessivecompulsive disorder. Interviews can be very informative, but there are potential problems with recall and other biases in gathering data on sexual behavior in this way (Wiederman, 2002). Questions about sexual history are sensitive and many interviewees may minimize or deny certain sexual interests or behaviors, especially those that could result in social ostracism. For example, a study found that male adolescents were more willing to disclose criminal acts they have committed than the fact that they have masturbated, even though many of them had engaged in both behaviors (Halpern, Udry, Suchindran, & Campbell, 2000). The validity of self-report may be increased if a rapport is established, as can occur after involvement in treatment (Worling & Curwen, 2000). One way to reduce the reluctance of individuals to disclose sexual interests or behavior in interviews is to administer questionnaires. For example, Koss and Gidycz (1985) found that male respondents were more likely to admit engaging in sexually coercive acts on a questionnaire than in an interview. Using questionnaires also addresses the potential problem of interviewers who skip or forget important questions. An example of a measure that has been developed for adolescents is the Sexual Fantasy Questionnaire, which contains items about atypical sexual fantasies (Daleidin, Kaufman, Hilliker, & O’Neil, 1998). Other Sources of Information Sexual history information can also be obtained from other sources, including interviews with an adolescent’s present sexual partner, if available, and a parent or guardian of children or younger adolescents. However, such information is usually limited because the minor will often be reluctant to disclose sexual urges, fantasies or thoughts to others, even with intimate others and even when such urges involve conventional targets or activities. A review of clinical or criminal justice records can also be very helpful. Clinicians can use information about sexual victim characteristics to make the diagnosis of pedophilia, because pedophilia is more likely to be present among sex offenders who have very young victims, boy victims and victims outside the offender’s immediate family (Seto, Murphy, Page, & Ennis, 2003). Data on the sexual behavior problems of children can be collected from maternal observations through the Child Sexual Behavior Inventory, which lists a variety of both typical and atypical sexual behaviors and asks mothers or other female caregivers to rate their frequency (Friedrich, 1997; Friedrich, Fisher, Dittner et al., 2001). Childhood sexual behavior problems are positively correlated with non-sexual behavior problems as measured by the CBCL. Some sexual behaviors are common among young children (e.g., touching sex parts at home, undressing in front of others), but aggressive sexual behaviors and more intrusive behaviors are rare. Phallometry and Viewing Time Phallometry involves the measurement of penile responses to sexual stimuli that systematically vary on the dimensions of interest, such as the age and sex of the figures in a set of slides depicting female children, adolescents and adults, and male children, adolescents and adults. Phallometry was developed as an assessment method by Freund (1963). Phallometric responses are recorded as increases in either penile circumference or penile volume; bigger increases in circumference or volume are interpreted as evidence of greater sexual arousal to the presented stimulus. Erectile response (except for erections that occur during sleep) is a specifically sexual measure, unlike other psychophysiological responses, such as pupillary dilation, heart rate, viewing time and skin conductance (Zuckerman, 1971). Phallometric indices of responding discriminate sex offenders from men who have not committed sexual offences and predict sexual recidivism. Other investigators have shown that phallometry can similarly distinguish men who admit to sadistic fantasies, cross-dress or expose their genitals in public from GENDER IDENTITY AND SEXUAL DISORDERS 873 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 873

men who do not (Freund, Seto, & Kuban, 1996; Marshall, Payne, Barbaree, & Eccles, 1991; Seto & Kuban, 1996). Seto, Lalumière, and Blanchard (2000) evaluated the use of phallometric testing for adolescent sex offenders with child victims. Because an age-matched comparison group was not available, these adolescents were compared with young adults between the ages of 18 and 21 who had not committed sexual offences involving children. As a group, adolescents with male victims had relatively higher responses to pictures of children than the young adult comparison participants. Adolescents with both male and female children as victims responded more to pictures of children than to pictures of adults. Further evidence for the validity of phallometry in adolescent sex offenders comes from Blanchard and Barbaree (2005), Gretton, McBride, O’Shaughnessy, and Kumka (2001) and Robinson, Rouleau, and Madrigano (1997). Another objective measure that is increasingly used with adolescent sex offenders is viewing time. The procedure involves showing a series of pictures depicting girls, boys, women or men; these pictures can depict clothed, semi-clothed or nude figures. Respondents are either asked to examine the pictures to answer later questions or they are asked to rate each picture (e.g., how attractive the depicted person is). Respondents are instructed to proceed to the next picture at their own pace and are expected to be unaware that the key dependent measure is the amount of time they spend looking at each picture. Viewing time is correlated with self-reported sexual interest and phallometric responding among non-offending volunteers recruited from the community (Quinsey, Ketsetsis, Earls, & Karamanoukian, 1996). Several studies have shown that adult sex offenders with child victims can be distinguished from other men by the amount of time they spend looking at pictures of children relative to pictures of adults (Harris, Rice, Quinsey, & Chaplin, 1996) or by a combination of viewing time and additional questionnaire responses (Abel, Jordan, Hand, Holland, & Phipps, 2001). However, Smith and Fischer (1999) were not able to demonstrate discriminative validity in a study of adolescent sex offenders and non-offenders using viewing time without additional questionnaire responses. No published studies have yet demonstrated that scores on viewing time measures, whether alone or in combination with self-report, predict recidivism among adolescent (or adult) sex offenders. Another potential problem for viewing time measures is that they may become vulnerable to faking once the client finds out that viewing time is the key variable of interest. Associated Psychopathology Kafka (1997, 2003) noted that paraphilias and related sexual disorders are often comorbid with mood disorders. In conjunction with evidence on the impact of antidepressant medications on sexual behavior, Kafka (1997) suggested that there is evidence that paraphilias and mood disorders share a common serotonergic diathesis. Consistent with this idea, we recently completed a meta-analysis of studies that compared adolescent sex offenders with adolescents who committed other kinds of offences (Seto & Lalumière, 2004). We found that adolescent sex offenders reported significantly more anxiety or depression than other offenders, suggesting variables in this domain might have a unique role in explaining sexual offending. However, the timing of symptoms was not clearly distinguished in the studies that we reviewed. The symptoms could have preceded the sexual offences, but they could also be a consequence of being identified as a sex offender (e.g., becoming more anxious and depressed after being arrested and charged for a sexual offence), given the extremely negative social views about sexual offending, even when compared to other forms of crime. Research on the association between paraphilias and mood disorders in samples outside of clinical or correctional settings is needed to determine if this link is an artifact of where the research has been conducted. There is also evidence that sexual behavior problems are frequently comorbid with non-sexual behavior problems (Friedrich, Grambsch, Broughton et al., 1991; Friedrich, Fisher, Dittner et al., 2001). Thus, children with sexual behavior problems and adolescent sex offenders may meet diagnostic criteria for attention deficit/hyperactivity disorder, oppositional defiant disorder or conduct disorder (Seto & Lalumière, 2004). France and Hudson (1993) reviewed studies on this topic and concluded that up to half of adolescent sex offenders would meet diagnostic criteria for conduct disorder. Paraphilias are often comorbid, such that engaging in one form of paraphilic behavior greatly increases the likelihood of engaging in another (Abel, Becker, Cunningham-Rathner, Mittelman, & Rouleau, 1988; Bradford, Boulet, & Pawlak, 1992). We have recently demonstrated that exhibitionistic and voyeuristic behavior also co-occurred more often than expected by chance in a nationally representative sample of young men between the ages of 18 and 21 from Sweden (Långström & Seto, 2006). The co-occurrence of paraphilic behaviors suggests there may be a common cause to the paraphilias (which does not rule out specific factors). There are no data of this kind for adolescents. Etiology Different theories have been proposed to explain the etiology of paraphilias. We focus here on theories that posit conditioning has a role, and theories that suggest paraphilias are the result of neurological perturbations. We then discuss the implications of recent findings on the association between childhood sexual abuse and pedophilia. Conditioning It has been proposed that masturbatory conditioning has a role in the development of paraphilias (McGuire, Carlisle, & Young, 1965). Some individuals, for unspecified reasons, pair cues of unusual initial sexual experiences with the sexual pleasure elicited by these early experiences, and learn to associate these cues with the powerful reinforcement of orgasm. However, there is equivocal evidence about the ability to condition increases in sexual arousal in humans. Rachman and Hodgson (1968) reported significantly increased sexual arousal CHAPTER 52 874 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 874

to pictures of boots among normal volunteers in a set of frequently cited experimental studies examining the development of boot fetishism, but these studies did not have control conditions. Lalumière and Quinsey (1998) and Hoffmann, Janssen, and Turner (2004) found a significant effect of conditioning on sexual arousal among male volunteers, while Plaud and Martini (1999) reported mixed results in a conditioning study of nine male volunteers. The efficacy of conditioning approaches for changing sexual arousal patterns has been reviewed elsewhere (Barbaree & Seto, 1997). Overall, this research suggests that conditioning can have an effect on the suppression of sexual arousal of adolescent or adult sex offenders, but it is unclear how long these changes are maintained, what mechanisms are actually responsible for the changes in sexual arousal patterns and whether the changes reflect an actual shift in sexual interests or greater voluntary control over sexual arousal when assessed in the laboratory (Lalumière & Earls, 1992). Conditioning might have a role in maintaining rather than initiating paraphilic sexual arousal. Dandescu and Wolfe (2003) found that atypical sexual fantasies preceded and accompanied the first sexual offence reported by a sample of 57 sex offenders with child victims; the sexual fantasies then increased in frequency after the first sexual offence. Approximately two-thirds of the offenders reported having any “deviant sexual fantasies” prior to their first contact with a child, while 81% reported having such fantasies afterwards. This finding suggests that the sexual arousal and gratification experienced during the first sexual offence increased the frequency of sexual fantasies among men who already had such fantasies. These research findings suggest that conditioning is unlikely to have a significant role in the onset of paraphilias. Experimental efforts to increase sexual arousal have produced small effects, and it is unclear how long conditioned changes in sexual responding (e.g., suppression of sexual arousal to child stimuli through aversive conditioning) can be maintained. If conditioning does have a role in the development of paraphilia, as suggested by Dandescu and Wolfe’s (2003) study, then there is probably an interaction between conditioning experiences and other predisposing factors. Neurological Perturbations Brain abnormalities have long been suspected as a cause of paraphilias. Information on brain functioning can be obtained both directly and indirectly. Indirectly, investigators have compared the performance of sex offenders with child victims with other groups of men on measures of intelligence and other aspects of cognitive functioning. Cantor, Blanchard, Robichaud, and Christensen (2005) conducted a meta-analysis of 165 adult samples and 71 adolescent samples. For the adults, there was a significant difference between sex offenders and others, with sex offenders scoring lower on measures of intelligence than other offenders, who, in turn, scored lower than non-offending controls. For the adolescents, there was a significant difference between sex offenders and other offenders. Among the sex offenders, Cantor et al. found that intelligence scores appeared to be related to the proportion of the sample who were pedophilic, because sex offenders with child victims scored lower than sex offenders without any child victims, and there was a positive relationship between the age cut-off used to define child victims and mean intelligence score; in other words, samples composed of men who offended against younger children tended to produce lower mean IQ scores than samples composed of men who offended against older children. In earlier studies, this same research group also found that pedophilic sex offenders were more likely to report experiencing head injuries before age 13, but did not differ in reports of head injuries after age 13 (Blanchard, Christensen, Strong et al., 2002; Blanchard, Kuban, Klassen et al., 2003). The fact that there was no difference in reports of head injuries after age 13 counters the alternative explanation that pedophiles have a retrospective reporting bias in recalling head injuries, because there would be no reason to think that head injuries before the age of 13 are more socially acceptable than head injuries later in life. The fact that there was no difference after age 13 also suggests there is a critical age window in terms of the impact of head injury on the development of pedophilia (and perhaps other paraphilias). This is an intriguing possibility because of other work that suggests the time before and during puberty is critical in the development of sexual preferences. Of course, this assumes that the relationship between head injury and pedophilia is causal. It is possible that a third variable explains both the higher incidence of head injury and pedophilia; for example, prenatal neurological perturbations may both increase accident-proneness, resulting in a higher risk for head injury, as well as increase the likelihood that an individual will be pedophilic. Researchers have also attempted to assess brain structure directly, especially in the frontal (associated with executive control) and temporal regions (associated with emotional processing and regulation of sexual behavior). Recently, Cantor, Kabani, Christensen et al. (2007) discovered a significant difference in white matter, such that pedophiles have less white matter than non-pedophilic men, particularly in two tracts: the right superior occipitofrontal fasciculus and the right arcuate fasciculus. These white matter tracts connect areas that are involved in determining if a stimulus is sexually relevant, suggesting that pedophiles are somehow deficient in how they process and integrate sexual cues. Cycle of Sexual Abuse The most frequently discussed factor in explanations of adolescent sexual offending is sexual abuse history (Knight & Sims-Knight, 2003). Seto and Lalumière (2004) conducted a meta-analysis of studies that compared adolescent sex offenders with other adolescent offenders, and found that adolescent sex offenders were almost five times as likely to have a history of sexual abuse. This was found whether the analysis involved studies that relied on self-report or on other sources of information (e.g., clinical files, official records). Moreover, adolescent GENDER IDENTITY AND SEXUAL DISORDERS 875 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 875

sex offenders who have been sexually abused show relatively greater sexual arousal to children or coercive sex than those who have not been abused (Becker, Hunter, Stein, & Kaplan, 1989). Among children, Friedrich, Grambsch, Damon et al. (1992) showed that scores on the Child Sexual Behavior Inventory could discriminate children who had been sexually abused from those who had not. Hall, Matthews, and Pearce (1998) found, in a sample of 99 sexually abused boys and girls between the ages of 3 and 7, that those who became sexually aroused during the sexual abuse were more likely to engage in sexual behavior problems involving others (n = 62) than those who did not (n = 37). The sexually abused–sexual abuser hypothesis suggests that male children who are sexually abused are more likely to engage in sexual offending later in life. Burton (2003) described plausible mechanisms linking sexual abuse and later sexual offending: modeling of the perpetrator’s behavior, conditioning as a result of pairing any sexual stimulation caused by the sexual abuse with cues such as the type of acts that occurred, and changing cognitions about the acceptability of adult–child sex. The mechanisms underlying the association between childhood sexual abuse and sexual behavior problems among children and adolescent sexual offending are not known (Widom & Ames, 1994). A number of investigators have suggested that aspects of the sexual abuse such as the victim–perpetrator relationship, nature of sexual abuse, duration and timing of the sexual abuse are important (Burton, 2003). There must be individual characteristics that increase both vulnerability to childhood sexual abuse and the likelihood of adolescent sexual offending, because many sexually abused children do not go on to commit sexual offenses later in life, and many adolescent sex offenders have no known history of sexual abuse. Developmental Course A developmental perspective is important because there are myriad differences between children, adolescents and adults, and because there is both persistence and desistance in sexual offending across the lifespan. Retrospective studies suggest approximately half of adult sex offenders report that their first sexual offense was committed as a juvenile (Abel, Osborn, & Twigg, 1993), and up to half of adolescent sex offenders have engaged in sexual misconduct under the age of 12 (Burton, 2000; Ryan, Miyoshi, Metzner, Krugman, & Fryer, 1996; Zolondek, Abel, Northey, & Jordan, 2001). In prospective studies, however, only a minority (perhaps 10–15%) of adolescent sex offenders commit another sexual offense over 5 years of opportunity (for review see Caldwell, 2002). The proportion of children with sexual behavior problems who persist and commit a sexual offense as an adolescent is also relatively small (Carpentier, Silovsky, & Chaffin, 2006). Taken together, these data indicate that many children with sexual behavior problems and many adolescent sex offenders desist. For many children, their sexual behavior problems may represent a reaction to sexual abuse they have experienced, sexual precocity and normative sexual play. For many adolescents, their sexual offenses may represent opportunistic and relatively transient antisocial behavior. A small group of children and adolescents, however, will persist in committing sexual offenses into adulthood. The adolescents who will develop paraphilias are more likely to persist. Of particular relevance is a study reported by Zolondek, Abel, Northey et al. (2001) of 485 adolescent males between the ages of 11 and 17, referred for assessment or treatment of possibly paraphilic interests or behavior. The youths completed a questionnaire with questions regarding sexual interests and experiences as part of their assessment. Twenty-six percent acknowledged engaging in fetishistic behavior, 17% acknowledged voyeuristic acts and 12% acknowledged exhibitionistic behavior. The average age of onset across paraphilic behaviors was between 10 and 12 years, which is younger than the average age of onset found in retrospective studies of adults (Abel, Becker, Mittelman et al., 1987). These data are consistent with the idea that the onset of paraphilias is likely around the time of puberty, just as many people are becoming aware of their sexual preference for males or females. Treatment Pharmacological Treatment The common aim of drug therapies in the treatment of pedophilia is to suppress sexual response involving children. Much of the initial interest was in antiandrogens to reduce sexual response, but clinicians and researchers have more recently focused on serotonergic agents. Because of the impact of reducing androgens on physical development, antiandrogens are very rarely prescribed for minors, except in cases where the sexual behavior is very problematic (e.g., very violent sexual assaults on young children) and cannot be controlled by other means. It is a logical hypothesis that antiandrogens would have an impact on paraphilic sexual responses, because testosterone has a critical role in male mammalian sexuality (Davidson, Smith, & Damassa, 1977). The earliest clinical study was reported by Laschet and Laschet (1971), who treated more than 100 paraphilic men; most were pedophiles or exhibitionists. Today, the most commonly prescribed agents are cyproterone acetate (CPA) or medroxyprogesterone acetate (MPA), both of which interfere with the action of testosterone. CPA blocks intracellular testosterone uptake and thus reduces plasma testosterone, while MPA reduces gonadotropin secretion. The use of either drug in the treatment of paraphilias is off-label, meaning it is not specifically approved by regulatory bodies for this purpose. Side effects of antiandrogens include headaches, dizziness, nausea, gynecomastia, depression and osteoporosis. As a result of these potentially serious side effects, and the fact that some patients do not want to experience a massive reduction in their sex drive, treatment attrition and compliance are significant issues in antiandrogen treatment. For example, in Hucker, Langevin, and Bain (1988), only 11 of 18 men completed the 12-week trial. There is some support for the efficacy of antiandrogens in reducing the frequency or intensity of sexual urges and CHAPTER 52 876 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 876

arousal, but there has been a dearth of larger, better controlled outcome studies. Gijs and Gooren (1996) reviewed the literature evaluating the effects of CPA and MPA, focusing on methodologically stronger studies that included features such as random assignment, a placebo condition and doubleblinding. They identified four controlled studies of CPA and six such studies of MPA. All four studies of CPA reported that treated men had a significant reduction in sexual response, while only one of the six MPA studies showed a significant difference between men in the treatment and comparison conditions. Serotonin is involved in the regulation of human sexual behavior, and antidepressant medications that have a serotonergic effect have long been known to reduce sexual desire and delay ejaculation in males (for a review see Meston & Gorzalka, 1992). Some clinical investigators have gone further and suggested, with little evidence, that selective serotonin reuptake inhibitors (SSRIs), such as fluoxetine and buspirone, can specifically affect sexual arousal to children without affecting sexual arousal to adults (Kafka, 1991). The interest in serotonergic agents for the treatment of paraphilias appears to be almost entirely based on uncontrolled case studies or open trials (Gijs & Gooren, 1996); there has been only one experimental study that compared desipramine and chloripramine in a double-blind crossover trial that was preceded by a single-blind placebo condition (Kruesi, Fine, Valladares, Phillips, & Rapoport, 1992). Kruesi, Fine, Valladares et al. reported a significant reduction of selfreported paraphilic behavior (predominantly exhibitionism, transvestic fetishism, telephone scatalogia and fetishism) with either drug, but their result was difficult to interpret because only 8 of 15 paraphilic men completed the trial; four patients were dropped because they responded to the placebo and three did not complete the drug trial. Including the patients who responded to placebo would have attenuated, and perhaps eliminated, the apparent effect of the drugs on self-reported paraphilic behavior. Despite the intuitive appeal of pharmacological interventions to reduce sexual drive and thus the likelihood of (illegal) paraphilic behavior, reviews of outcome studies suggest that there is no strong empirical support for the idea that such interventions can reduce sexual recidivism. Psychosocial Aversive conditioning techniques were used as early as the 1950s and 1960s for paraphilic interests such as fetishism and transvestic fetishism (Raymond, 1956). In the behavioral treatment of paraphilias, aversion techniques are used to suppress sexual arousal to the atypical target or activity, while masturbatory reconditioning techniques are used to increase sexual arousal to acceptable targets (peers or adults) and activities (consenting sexual activities). In aversion procedures, unpleasant stimuli such as mild electric shock or ammonia odors are paired with repeated presentations of sexual stimuli depicting the paraphilic target or activity. In a variation called covert sensitization, the aversive stimulus is imagined (e.g., being discovered by family, friends or coworkers while engaging in paraphilic behavior). Overall, the existing research suggests that behavioral techniques can have an effect on sexual arousal patterns, but it is unclear how long these changes are maintained and whether they result in actual changes in interests, as opposed to greater voluntary control over pedophilic sexual arousal (Lalumière & Earls, 1992). Cognitive–behavioral treatments (CBT) aim to teach individuals how to manage their sexual urges. Published evaluations have reported promising results. Carpentier, Silovsky, & Chaffin (2006) reported on a long-term evaluation of 135 children with sexual behavior problems, compared with 156 children from the same out-patient clinic. The children with sexual behavior problems were randomly assigned to a play therapy group or a CBT group. Both treatments were manualized and ran for 12 hour-long sessions. The CBT group was highly structured and focused on education and behavior management. The play therapy group was less structured and more reflective, although it focused on many of the same themes: sexual behavior problems, boundaries, parenting skills and sex education. Children assigned to the CBT group were significantly less likely than children assigned to the play therapy group to sexually offend (defined as an arrest for a sexual offence or a child welfare report of sexual abuse perpetration) during the 10-year follow-up period. In fact, the rate of sexual offending among children in the CBT group was not significantly different from the comparison group without any sexual behavior problems. Borduin, Henggeler, Blaske, and Stein (1990) reported a significant effect of treatment in a small sample of adolescent sex offenders, using a multisystemic treatment that targeted factors associated with criminal behavior, including antisocial attitudes and beliefs, associations with antisocial peers and substance abuse (for a review see Andrews & Bonta, 2002). Multisystemic treatment is also distinguished by being skillsfocused, involving both the adolescent and his or her family, and paying careful attention to program fidelity and individualization. Multiple studies have shown that multisystemic treatment can significantly reduce recidivism and other negative outcomes among serious adolescent offenders (Henggeler, Schoenwald, Borduin, Rowland, & Cunningham, 1998). Borduin and Schaeffer (2001) replicated the Borduin, Henggeler, Blaske et al. (1990) study by showing that this multisystemic treatment had a large impact on sexual recidivism in a larger sample of 48 juvenile sex offenders, even though it was not designed specifically for this group. Conclusions There are many unanswered questions about the onset, development and prognosis of paraphilias. There has been relatively little research conducted on adolescents, perhaps reflecting a discomfort with the idea that some adolescents and older children engage in problematic sexual behavior as a result of atypical sexual interests that are very likely to be stable. As GENDER IDENTITY AND SEXUAL DISORDERS 877 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 877

a result, this chapter has drawn upon relevant studies of adult sex offenders. This is not to suggest that children with sexual behavior problems or adolescent sex offenders are likely to have paraphilias. In fact, it is likely that most do not, given the high rate of desistance from childhood to adolescence, and from adolescence to adulthood. A small minority do persist, however, and are likely to continue engaging in problematic sexual behavior without intervention. There is emerging evidence to support the idea that there may be neurodevelopmental or neurological risk factors for pedophilia. Given the high comorbidity of paraphilias, it is likely that other paraphilias are similarly influenced. As for many other disorders, we know more about the assessment than the treatment of paraphilias. Further research on paraphilias will be expedited by the development of ageappropriate measures of sexual interests. Viewing time is a particularly promising technology. Unlike the adult literature, however, there are some promising results in the treatment of sexual behavior problems in children and sexual offending among adolescents. These treatments share a common focus on skills, behavior and problemsolving, and involve parents or other caregivers. Further advances in treatment will benefit from a greater undestanding of the etiology and developmental course of paraphilias. Further Reading Barbaree, H. E., & Marshall, W. L. (Eds.). (2006). The juvenile sex offender (2nd edn.). New York: Guilford Press. Cohen-Kettenis, P. T., & Pfäfflin, F. (2003). Transgenderism and intersexuality in childhood and adolescence: Making choices. Thousand Oaks, CA: Sage Publications. Laws, D. R., & O’Donohue, W. T. (Eds.). (1997). Sexual deviance: Theory, assessment and treatment. New York: Guilford Press. Zucker, K. J., & Bradley, S. J. (2005). Gender identity disorder and psychosexual problems in children and adolescents. New York: Guilford Press. 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Sex differences in referral rates of children with gender identity disorder: Some hypotheses. Journal of Abnormal Child Psychology, 25, 217– 227. Zucker, K. J., Finegan, J. K., Doering, R. W., & Bradley, S. J. (1984). Two subgroups of gender-problem children. Archives of Sexual Behavior, 13, 27–39. Zucker, K. J., Green, R., Coates, S., Zuger, B., Cohen-Kettenis, P. T., Zecca, G. M., et al. (1997b). Sibling sex ratio of boys with gender identity disorder. Journal of Child Psychology and Psychiatry, 38, 543–551. Zucker, K. J., Green, R., Garofano, C., Bradley, S. J., Williams, K., Rebach, H. M., et al. (1994). Prenatal gender preference of mothers of feminine and masculine boys: Relation to sibling sex composition and birth order. Journal of Abnormal Child Psychology, 22, 1–13. Zucker, K. J., & Spitzer, R. L. (2005). Was the Gender Identity Disorder of Childhood diagnosis introduced into DSM-III as a backdoor maneuver to replace homosexuality? An historical note. Journal of Sex and Marital Therapy, 31, 31–42. Zuckerman, M. (1971). Physiological measures of sexual arousal in the human. Psychological Bulletin, 75, 297–329. GENDER IDENTITY AND SEXUAL DISORDERS 881 9781405145497_4_052.qxd 29/03/2008 02:55 PM Page 881

882 This chapter covers disruptive behavior problems, including oppositional and attentional difficulties; emotional problems such as fears, phobias and depression; and feeding problems in the age period 0–5 years. It will begin by considering common developmental aspects and causal explanations of early behavioral problems, followed by describing, for each type of disorder, classification, prevalence and stability. Finally, we discuss treatment. Other problems common in the early years are discussed in chapters on sleep (see chapter 54), attachment disorder (see chapter 55) and developmental disorders (see chapters 3, 34, 46 and 47). Cross-reference is also made to chapters on similar disorders in older children. The term “disorder” is used cautiously in this age group, with some skepticism about its validity (Campbell, 2002). Evidence for stability and prognostic significance of preschool problems is not particularly strong, especially in the underthrees. Comorbidity is very high, and there are also concerns about distinguishing normal from abnormal behavior in this period of rapid developmental change, and about labeling very young children with disorders. However, there may also be disadvantages of not defining disorders in young children, including failure to recognize distress and provide appropriate help (Carter, Briggs-Gowan, & Davis, 2004; Egger & Angold, 2006). The latter is particularly important, given that there is a good deal of evidence about effective interventions for this age group, particularly for disruptive behavior problems. In view of these uncertainties about defining disorders, most studies in the 0–5 age group use dimensional approaches to defining and measuring problem behavior. Accordingly, we use the term “disruptive problems” to refer to a constellation of oppositional or attentional symptoms, and the term “emotional problems” to refer to depressive and anxious-type symptoms. Studies based on dimensional approaches include children with severe or milder problems, often defined by clinical cutoff on symptom questionnaires. Where studies use diagnostic criteria, we use DSM and/or ICD disorder terminology. The period of infancy refers to 0–1 years, toddlerhood 1–3 years and preschool 3–5 years. Developmental Perspectives on Problem Behavior in Early Childhood It is important to recognize the rapid pace of growth and maturation that takes place from birth to age 5, which has multiple implications for assessment and treatment. For the primary types of problem behavior discussed in this chapter (i.e., disruptive and emotional problems), identification and diagnosis based purely on child behavior have been shown to have limited stability and prognostic implications until at least 24 months of age (Campbell, Shaw, & Gilliom, 2000; Keenan, Shaw, Walsh, Delliquadri, & Giovannelli, 1997). For assessing risk of clinically significant child problem behavior in the 0–2 age group, greater emphasis has been placed on parenting and factors that might compromise parental functioning (e.g., depression, social support, teen parenthood; Shaw, Dishion Supplee et al., 2006), as well as factors that might compromise the developing brain (e.g., prematurity; prenatal drug use). Child problem behavior shows increasing stability beginning in the toddler period. Whereas a substantial percentage of children will “outgrow” these problems, longitudinal studies suggest that 50–60% of children showing high rates of disruptive behavior at age 3–4 will continue to show these problems at school age (Campbell, Szumowski, Ewing et al., 1982; Campbell, Pierce, Moore et al., 1996; Campbell, Shaw, & Gilliom, 2000; Richman, Stevenson, & Graham, 1982). In a recent study of low-income boys, among those identified above the 90th percentile on disruptive symptoms at age 2, 60% remained above the 90th percentile and 100% (all 18) remained above the median at age 6 (Shaw, Gilliom, & Giovannelli, 2000; Shaw, Gilliom, Ingoldsby et al., 2003). The low stability of behavior problems between toddlerhood and later childhood has implications for using the term “disorder” in referring to these problems in very early childhood (Campbell, 2002; Egger & Angold, 2006). It may be somewhat less problematic in the preschool range. There is currently much debate about the appropriateness of using traditional diagnostic categories, particularly for infants and toddlers. Rapid developmental change within the period 0–5 makes it particularly hard to distinguish normal from abnormal behavior, and to set age-appropriate criteria for classification systems. Thus, having six temper tantrums a day might be bothersome but normal at 22 months, but could contribute to a diagnosis of oppositional defiant disorder (ODD) in a 4-year-old. Similarly, Behavioral Problems of Infancy and Preschool Children (0–5) Frances Gardner and Daniel S. Shaw 53 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 882 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

a 2-year-old showing difficulty separating from a parent in the first week of daycare would be less worrisome than a 5-year-old showing similar reactions over many weeks. Clearly, diagnostic systems need to be able to take these developmental variations into account. Systems for assessing preschoolers go some way to solving this problem, using criteria that are more age-appropriate, and basing assessments on observations and reports of child behavior in multiple settings (i.e., home and preschool, from parents and teachers). Recent advances in the field include the Research Diagnostic Criteria–Preschool Age (RDC-PA) which has attempted to modify DSM criteria to be suitable for preschoolers (Task Force on Research Diagnostic Criteria, 2003), and the Preschool Age Psychiatric Assessment (PAPA; Egger & Angold, 2004), which is a structured parent-interview schedule for ages 2–5. However, these systems cannot easily solve the problem that meaning and significance of problem behavior shift rapidly even within this period, and within a given cultural or family setting. Systems for very young children, such as the diagnostic classification: 0–3 (DC: 0–3) (Zero to Three, 1994), tend to place more emphasis on aspects of the parent–child relationship, rather that focusing solely on child behavior, with an increasing focus on behavior as children approach school age. However, evidence for validity of 0–3 diagnostic categories, including stability and, in some cases, links to later disorders of the same name (e.g., infantile “anorexia”; Chatoor & Ganiban, 2004) is not strong. Based on the relative instability of problem behavior during early childhood, it should not be surprising to learn that many children demonstrate multiple types of problem behavior, including co-occurring disruptive and emotional problems (e.g., oppositionality coupled with depression or attention deficit/hyperactivity disorder [ADHD]). Egger and Angold (2006) reported that 50% of preschoolers showing a disorder in their community sample also showed one or more cooccurring disorders. Similar rates of comorbidity were found by Lavigne, Gibbons, Christoffel et al. (1996) and Keenan, Shaw, Walsh et al. (1997). Arguably, very high rates of comorbidity could be seen as a reason for caution in using diagnostic systems in this age group. However, this problem is not specific to the 0–5 range, as high rates of comorbidity are typical of childhood disorders in general (see chapter 2; Angold, Costello, & Erkanli, 1999). Because of these rapid changes in development, and instability across time and context, problems may manifest themselves in different ways in under-fives compared to older children. For example, toddler fears and worries may manifest as irritable or oppositional behavior (e.g., tantrums). These may be harder to detect than in older children because of young children’s limited ability to communicate emotions to adults. Equally, superficially similar behaviors in toddlers may reflect quite different underlying problems. Thus, if a toddler is isolated from peers in the nursery, it is important to assess whether this is a temporary reaction to a new environment, or related to aggression, social anxiety or to a more profound problem in communication (e.g., severe learning disability, autism spectrum disorder; see chapters 46 and 49). In a school-aged child, pervasive learning problems would be more apparent and, in most cases, already identified. Theoretical perspectives Preschool behavior problems are influenced by both biological and environmental factors, as manifest in individual differences in child characteristics (e.g., temperamental dimensions of activity, sociability, attention) and the quality of the caregiving environment. Genetic and prenatal environmental factors are influential in this age period (see chapters 23 and 30). We distinguish between risk factors, in the presence of which the probability of showing a disorder is raised; precursors, where there is continuity between an early problem (e.g., preschool disruptive problems) and a later one (e.g., conduct disorder); and the presence of formal disorder. Extreme difficult temperament is often viewed as a risk factor for later behavior problems (Hill, 2002), although at moderate levels of difficulty and without other indicators of child or family risk, such individual differences are likely to reflect developmentally normative patterns rather than necessarily implying risk for disorder. In recent years, there has been increasing recognition of the multiple interacting factors that contribute to divergence in outcomes of infants who demonstrate early problems in feeding, emotionality or disruptive behavior (Campbell, Shaw, & Gilliom, 2000). This change in focus can be traced to the pioneering efforts of Thomas, Chess, and Birch (1968), who emphasized the goodness-of-fit between parent and child temperament, to Bell’s (1968) work on children’s effects on parents, and Sameroff and Chandler’s (1975) transactional model of parent–child interaction. Thus, assessment and intervention efforts across problem behavior types have focused on changing child behavior, parent behavior and resources, and the quality of parent–child interaction. As children under 5 years are so dependent on their caregiving environment, there is an emphasis on identifying risk factors in the family and the wider caregiving context (e.g., quality of daycare or nonparental caregiving) which moderate the course of early problem behavior. In early childhood intervention, similar parent management strategies are often used to manage apparently dissimilar problems (e.g., infant feeding or sleeping problems, preschool disruptive behavior). That similar intervention strategies are used to treat these various problem behaviors should not be surprising, as self-regulation skills develop rapidly during this age period. Self-regulation involves the ability to control impulses and expressions of emotion; thus, children with difficulties in self-regulation might show a range of problems, including higher rates of tantrums, irritable mood and oppositionality, and disturbances in sleep, eating, activity or attention. Despite the range of symptoms, there may be common maintaining mechanisms. All of these problems might elicit similar levels of frustration in parents, their responses leading to a worsening of child symptoms, in a cycle of coercive interaction (Patterson, 1982; Shaw & Bell, 1993). BEHAVIORAL PROBLEMS 883 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 883

In recent decades, there has been much speculation about the need to ensure that children’s early years are not marked by environmental adversity, because of the fear of irreversible harm, as evidenced in animal studies. Unfortunately, relevant human data are limited. There is some evidence to suggest that exposure to psychosocial adversity during the toddler period shows stronger predictions to later outcomes than exposure in later periods (Appleyard, Egeland, van Dulmen, & Sroufe, 2005; Shaw, Gilliom, & Giovannelli, 2000). Thus, in the study by Appleyard et al. (2005) of children from low-income families, a cumulative index of contextual adversity in early childhood continued to be associated with adolescent antisocial behavior after accounting for the effects of contextual risk in middle childhood. However, in most cases, study designs have not permitted examination of whether there are critical periods for the operation of risk factors, where, based on timing of exposure, there might be differential effects on child adjustment. Clinical Problems Disruptive Behavior Disruptive behavior, sometimes referred to as externalizing or “acting-out” problems, includes attentional and oppositional problems and their corresponding disorders, ADHD and ODD (Lahey, Loeber, Quay, Frick, & Grimm, 1992). As conduct disorder (CD) includes more serious forms of aggression, property destruction and theft, it is rarely applied to preschoolers. Disruptive problems represent the most frequent type of problem behavior in early childhood, particularly after the second year when parental expectations for children to comply with rules and contain aggressive behavior increase. Tremblay (1998) reported that by age 17 months, 70% of children take toys away from other children, 46% push others to obtain what they want and 21–27% engage in one or more of the following with peers: biting, kicking, fighting or physically attacking. Tremblay also reported that aggression occurs more frequently for infants with siblings, especially for girls, providing daily opportunities for conflicts over possessions. Relative to emotional problems, much more research has been conducted on the stability, course and predictors of disruptive problems in early childhood and about 50% of oppositional 3-year-olds continue to have these problems in the school years (Campbell, Shaw, & Gilliom, 2000). Using DSM-IV criteria, generated from a structured interview with parents, the prevalence of preschool ODD in a US sample was estimated at 7%, and ADHD at 3% (Egger & Angold, 2006). Other studies broadly concur, although some questionnaire studies arrive at higher estimates, depending, not surprisingly, on the choice of definition, cut-off and informant (Koot, 1993; Richman, Stevenson, & Graham, 1982). In the toddler and preschool years, sex differences are not as marked as in older children. Boys’ higher rates of disruptive behavior seem to emerge during the later preschool period, with studies documenting absence of sex differences from ages 1 to 3 (Achenbach, 1992; Keenan & Shaw, 1994; Richman, Stevenson, & Graham, 1982), followed by increasing sex differences from age 4 to 5 (Lavigne, Gibbons, Christoffel et al., 1996; Rose, Rose, & Feldman, 1989), although this was not found for ODD by Egger and Angold (2006). Attention Deficit/Hyperactivity Disorder Core features include inattention, impulsivity and hyperactivity (Barkley, 1998). In terms of etiology, models tend to focus on different biological factors, including activity, impulse and attention control (see chapter 34; Barkley, 1997; Rothbart, Ellis, Rueda, & Posner, 2003). As with older children, most preschoolers with ADHD show other difficulties, most typically conduct problems (Barkley, 1997; Shaw, LaCourse, & Nagin, 2005). This co-occurrence carries risk for more severe and chronic adjustment problems in adolescence and adulthood (Moffitt, 1990; Weiss & Hechtman, 1993). Oppositional Problems These include defiant, angry, annoying, non-compliant and sometimes aggressive behaviors. For a diagnosis of ODD, behaviors need to occur more commonly than is typical for that age, persist for more than 6 months, and impair the child’s functioning (American Psychiatric Association, 2000; Campbell, 2002). During toddlerhood, despite the high frequency of aggressive-like and oppositional behavior, many of these behaviors tend to be tolerated by parents because of children’s limited ability to understand the consequences of their actions. However, with emerging cognitive maturation during this period, parental tolerance for disruptive behavior decreases, particularly when children cannot refrain from showing this behavior with peers and adults outside of the home (e.g., in daycare). Causes and Correlates of Disruptive Behavior Problems Causal models emphasize the dynamic interplay of child, parenting and contextual factors (Sameroff & Chandler, 1975), with individual differences in disruptive behavior often being magnified by ineffective parent management strategies (Campbell, Pierce, Moore et al., 1996; Shaw, Gilliom, Ingoldsby et al., 2003). Most of this research has focused on early predictors of conduct problems rather than ADHD, but one recent study found similar types of predictors for both conduct problems and ADHD (Shaw, Lacourse, & Nagin, 2005). Predictive validity for early child markers of more serious disruptive behavior begins to emerge around age 2 (Keenan, Shaw, Walsh et al., 1997; Shaw, Gilliom, Ingoldsby et al., 2003), with clearer predictions from age 3 to later, more serious forms of antisocial behavior (Caspi, Henry, McGee, Moffitt, & Silva, 1995; Henry, Caspi, Moffitt, & Silva, 1996). Parenting models have been developed from attachment and social learning perspectives, with attachment models emphasizing unresponsive caregiving during infancy (Greenberg, Speltz, & DeKlyen, 1993; Sroufe, 1997), and social learning models focusing on the development of coercive cycles in which parents unwittingly reinforce and maintain child disruptive behavior by using inconsistent and harsh management strategies CHAPTER 53 884 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 884

(Patterson, 1982; Shaw & Bell, 1993). Research has consistently validated associations among low-quality parent–child relationships in the early years, the use of unresponsive or harsh parenting practices, and disruptive behavior in preschool and later years (Loeber & Dishion, 1983; Lyons-Ruth, 1996; Shaw, Keenan, & Vondra, 1994; Shaw, Winslow, Owens et al., 1998). Positive parenting skills (e.g., anticipating the child’s troublesome moments, providing joint play activities) may be particularly important in the toddler years, as they can help to divert children from problem behavior at an age when children have limited skills for self-management of boredom and tempting impulses (Gardner, 1994; Gardner, Sonuga-Barke, & Sayal, 1999; Gardner, Ward, Burton et al., 2003; Gardner, Shaw, Dishion et al., 2007; Martin, 1981). Rather different parenting skills become important in middle childhood, where increasing independence requires greater monitoring of children across multiple settings, and planning for preventing problems that may occur in the parent’s absence. The wider family and caregiving environment can also influence the development of early emotional and behavioral problems. The nature of sibling relationships, for example, appears to be influenced by factors such as the temperament of each child, and the quality of other relationships in the family, such as parent–child and marital relationships (Dunn, 1993). Sibling relationships often encompass a complex mix of supportive and conflictual dimensions, and appear to be important for development. For example, Garcia, Shaw, Winslow, and Yaggi (2000) found that sibling conflict at age 5 was predictive of later disruptive problems at home and school, after accounting for earlier parenting and child behavior. As young children spend more time outside the home, researchers have addressed whether and how daycare might affect early problem behavior. Early studies of daycare were mixed in quality of the methods used, quality of the daycare studied and the results found. A recent US study of daycare effects (over 1000 children) found that more hours spent in any kind of non-maternal care from infancy to age 54 months was associated with higher ratings of oppositional problems, more so for ratings made by alternative caregivers and teachers than parents (NICHD Early Child Care Research Network, 2003). These effects, albeit statistically significant, tended to be modest in magnitude and did not predict trajectories of clinically meaningful problem behavior. The quality of early child care has also bee found to discriminate conduct problems in a large sample of children from low-income families (Votruba–Drzal, Coley, & Chase-Landsdale, 2004). Results indicated that many hours of low-quality care was associated with higher oppositional problems at school entry, whereas high-quality care served a protective function. Fewer behavior problems have also been found in children in highquality day care, compared to home care, in Sweden, even at 10-year follow-up (Andersson, 1992). Overall, quality and, to some extent, amount of care, have been found to influence the course of early disruptive problems. In general, risk factors for early disruptive behavior are similar to those for later CD (see chapter 35), including parental attributes (e.g., mental illness; see chapter 27) and contextual factors (e.g., socioeconomic status; Shaw, Keenan, & Vondra, 1994; Shaw, Winslow, Owens et al., 1998; Tremblay, Nagin, Seguin et al., 2004). Cumulatively, these data suggest that several targets have been established for prevention and intervention studies (see chapter 61; Olds, 2002), some of which might be appropriate before a child is born. Emotional Problems Emotional problems such as anxiety, depression and posttraumatic stress in preschoolers have been much less studied than disruptive problems. There are a number of reasons for this, including the inability of young children easily to communicate about their emotions, or for adults to notice them as problematic. Furthermore, there are difficulties in distinguishing developmentally normal emotions (e.g., fears, crying) from more severe and prolonged anxiety or misery that might constitute a disorder. This is especially difficult in the early years, when children undergo rapid changes in the development of emotions, and in their ability to communicate these to others. There is probably insufficient evidence at present to decide whether diagnosing emotional disorders is valid and useful in the 0–5 year range. Classification Systems Many DSM categories, including social phobia, generalized anxiety and depression apply across the range of child to adulthood, and are therefore not designed to take preschool developmental factors into account. One exception is Separation Anxiety Disorder, which applies only to children. It is defined as “developmentally inappropriate and excessive anxiety concerning separation from home or from those the child is attached to” (APA, 2000). However, there are still problems in applying this diagnosis to very young children because normative expectations of responses to separation from caregivers change so much across the first 3 years. Thus, in the 0–3 system, this constellation is not considered abnormal and therefore is not included as a disorder. In all cases it is important to take into account the child’s developmental stage in assessing whether a problem is of concern. The majority of children show fears at some stage in development, and typical fears change with age. Thus, fear of strangers is very common in late infancy, and fear of animals in toddlerhood. Whether they constitute a clinical problem (Campbell, 2002) depends also on factors such as persistence, and how much they impair the child’s and family’s well-being, for example if the child is persistently unable to engage in normal activities (e.g., going to daycare or the park). Stability Many emotional problems in this age range are thought to represent transient reactions to stressful life events, rather than disorders per se. Relative to studies of disruptive problems, there is a paucity of research on the stability of early emotional problems for young children. In one study, stability of preschool depression over 6 months was high among 3- to BEHAVIORAL PROBLEMS 885 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 885

5-year-olds (Luby, Heffelfinger, Mrakotsky et al., 2002), but there are no data on the long-term prognosis for these children. Both predictive validity and functional impairment are important in validating a diagnostic category, and in the case of depressed preschoolers, there is preliminary evidence that these children show moderate to high levels of impairment in their daily functioning (Egger & Angold, 2006). Prevalence Studies of preschool children using DSM-III and DSM-IV criteria have estimated prevalence of emotional disorder diagnoses; these tend to be low for each category: separation anxiety (0.3–5%), social phobia (2–4%), specific phobia (0–2%) and depression (0–2%; Egger & Angold, 2006; Lavigne, Gibbons, Christoffel et al., 1996). However, in Egger and Angold’s study, which included multiple types of problem behavior, rates of any emotional disorder were as high as for disruptive disorders, both around 10%. As found in middle childhood, prevalence of depression and anxiety did not differ by gender (Keenan, Shaw, Walsh et al., 1997). Sex differences tend to emerge around puberty. Comorbidity between depression and ODD was very high in one study of 2- to 5-year-olds (Egger & Angold, 2006), raising questions about whether these are separate disorders. Causes and Correlates of Emotional Problems As with disruptive behaviors, causes are likely to be multifaceted, and to reflect the interplay between child and parent genetic factors, parenting and the wider social environment around the child. As with other aspects of emotional problems, there have been few studies of multiple etiological factors, especially in preschool children. Studies have suggested quite high heritability for some preschool emotional problems (Eley, Bolton, O’Connor et al., 2003). Where risk factors have been studied in preschoolers, broadly these appear similar to those found in studies of older children (Luby, Heffelfinger, Mrakotsky et al., 2003). Maternal transmission of anxiety also seems to be an important factor, not only via genetic factors, but also through processes such as modeling and an anxious style of parenting (see chapter 27). Individual differences in children’s temperament (see chapter 14) also have been linked to early emotional problems, most notably for children who are behaviorally inhibited (Hirshfeld-Becker, Biederman, & Rosenbaum, 2004). Behavioral inhibition is defined as the tendency to react to novelty with unusual fear, cautiousness and withdrawal, and appears to be moderately stable across early childhood (Kagan, 1999; Kagan, Reznick, & Snidman, 1988). Biederman, Hirshfeld-Becker, Rosenbaum et al. (2001) found that behavioral inhibition assessed during early childhood was associated with increased risk for anxiety disorders 5 years later. Another recent study examined predictors of trajectories of boys’ anxiety from ages 2 to 10 years, and subsequent emotional disorders in early adolescence (Feng, Shaw, Lane, Alarcon, & Skuban, 2006). Consistent with Kagan’s work, shy temperament at age 2 tended to differentiate between initial high- and low-anxiety trajectory groups, whereas early maternal negative control and maternal depression were associated with increasing trajectories and elevated anxiety symptoms in middle childhood. Follow-up to adolescence indicated that child factors such as temperament contributed strongly to diagnoses of anxiety disorders, whereas both child and parent factors (e.g., negative control) contributed to boys’ depression. Eating and Feeding Problems Normal patterns of feeding change greatly between individuals and across time in the developmental period 0–5. The same can be said for disorders of eating and feeding, which present in a wide variety of ways (Stein & Barnes, 2002). Thus, in the early months, babies may fail to thrive for a number of reasons, or show signs of infantile colic. In the preschool years, food refusal or excess fussiness may be linked to other toddler oppositional behaviors, and intervention may need to consider parental management of child behavior more broadly. Feeding problems are common, but are more prevalent in children with developmental disabilities, pointing to the combination of physical health, family factors and developmental delay that are often involved in feeding problems. Their multifactorial origins can make it hard to define which problems belong within the realm of child psychiatry, rather than pediatrics, as many feeding problems that are primarily physical (e.g., failure to thrive [FTT] as a result of oral–motor dysfunction in cerebral palsy) may or may not have a substantial component exacerbated by family stress or poor parenting skills. The picture is further complicated by the fact that etiology is often unknown (e.g., in FTT, colic). While bearing in mind this caution, we will focus on problems thought to have a primarily psychosocial cause. Classification and Prevalence There is no standard system for classifying all types of feeding problems in the 0–5 period. DSM-IV includes the categories pica, rumination and “feeding disorder of infancy and early childhood,” defined as persistent failure to eat adequately, with weight loss or failure to gain weight that is not caused by physical illness, and onset before age 6. Various systems of classification have been proposed, but some make unwarranted assumptions about etiology, for example Chatoor & Ganiban (2004), who classify disorders by psychogenic or other cause (e.g., “feeding disorder of caregiver–infant reciprocity,” or “post-traumatic feeding disorder”). This chapter covers the most common problems in this age group, including colic, FTT and food refusal. Feeding problems have been estimated to affect up to 30% of infants (Jenkins, Bax, & Hart, 1980). However, given the lack of standard criteria for definition, figures vary quite widely across studies. Similarly, investigation of prognosis and causes of feeding problems is hampered by lack of a clear classification system. Infant Colic The causes of colic are unclear and, as a result, it has been classified variously as a feeding, regulatory or crying problem. Wessel, Cobb, Jackson, Harris, and Detweiler’s (1954) classic “rule of three” definition involves “paroxysms of irritability, CHAPTER 53 886 9781405145497_4_053.qxd 29/03/2008 02:55 PM Page 886