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Journal of Affective Disorders 107 (2008) 299 305 www.elsevier.

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Preliminary communication

Family environment patterns in families with bipolar children


Cecilia Belardinelli a,b,, John P. Hatch a,c , Rene L. Olvera a , Manoela Fonseca a,d , Sheila C. Caetano a,d , Mark Nicoletti a , Steven Pliszka a , Jair C. Soares e
MOOD-CNS Program, Division of Mood and Anxiety Disorders, Department of Psychiatry, The University of Texas Health Science Center at San Antonio, San Antonio, TX, USA b University of Buenos Aires, School of Medicine, Buenos Aires, Argentina c Department of Orthodontics, The University of Texas Health Science Center at San Antonio, San Antonio, TX, USA d Department of Psychiatry, University of Sao Paulo School of Medicine, Sao Paulo, Brazil Yeargan Distinguished Professor of Psychiatry Director, UNC Center of Excellence for Research and Treatment of Bipolar Disorders (CERT-BD), Department of Psychiatry, UNC School of Medicine, USA Received 27 June 2007; received in revised form 10 August 2007; accepted 14 August 2007 Available online 1 October 2007
a

Abstract Background: We studied the characteristics of family functioning in bipolar children and healthy comparison children. We hypothesized that the family environment of bipolar children would show greater levels of dysfunction as measured by the Family Environment Scale (FES). Methods: We compared the family functioning of 36 families that included a child with DSM-IV bipolar disorder versus 29 comparison families that included only healthy children. All subjects and their parents were assessed with the K-SADS-PL interview. The parents completed the FES to assess their current family functioning. Multivariate analysis of variance was used to compare the family environment of families with and without offspring with bipolar disorder. Results: Parents of bipolar children reported lower levels of family cohesion ( p b 0.001), expressiveness ( p = 0.005), activerecreational orientation ( p b 0.001), intellectualcultural orientation ( p = 0.04) and higher levels of conflict ( p b 0.001) compared to parents with no bipolar children. Secondary analyses within the bipolar group revealed lower levels of organization ( p = 0.031) and cohesion ( p = 0.014) in families where a parent had a history of mood disorders compared to families where parents had no history of mood disorders. Length of illness in the affected child was inversely associated with family cohesion (r = 0.47, p = 0.004). Limitations: Due to the case-control design of the study, we cannot comment on the development of these family problems or attribute their cause specifically to child bipolar disorder. Conclusion: Families with bipolar children show dysfunctional patterns related to interpersonal interactions and personal growth. A distressed family environment should be addressed when treating children with bipolar disorder. 2007 Elsevier B.V. All rights reserved.
Keywords: Family environment; Bipolar disorder; Children

1. Introduction Bipolar disorder is a familial disorder, which is influenced by genetic and environmental factors (Althoff et al., 2005). Among the environmental factors considered most important is the family environment. Previous

Corresponding author. 14227 Sage Trail, San Antonio, Texas 78231, USA. Tel.: +1 210 492 3019. E-mail address: cecibel2000@hotmail.com (C. Belardinelli). 0165-0327/$ - see front matter 2007 Elsevier B.V. All rights reserved. doi:10.1016/j.jad.2007.08.011

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studies link disordered family environment with poor prognosis in bipolar disorder. A number of studies have noted that exposure to family members who exhibit high expressed emotion (high criticism, emotional over-involvement and negative affective style) may increase the risk of relapse (Johnson et al., 2003; Miklowitz et al., 2004), prolong episode duration (Johnson et al., 1999) and predict higher levels of mania and depression at follow-up (Kim and Miklowitz, 2004). In a retrospective rating of their families of origin, no significant differences in family functioning were observed between adult bipolar patients and control subjects when assessed using the Family Environment Scale (FES). Nevertheless, within the adult bipolar patients, those with a history of dysthymia reported lower levels of family Expressiveness, while those with a history of suicide attempts reported lower ratings of family Cohesiveness compared to the control sample (Cooke et al., 1999). Characteristics like insecure attachment and more hostility have been described in relation with a higher incidence of relapse after recovery (Geller et al., 2000). Other psychosocial stressors interact with genetic predisposition to produce neurobiological changes that could lead to affective episodes as well as create vulnerability for future episodes (Post, 1992; Caspi et al., 2003). A history of traumatic experiences is related to suicide attempts in bipolar adults (Leverich et al., 2002), suggesting that early adverse experiences create a vulnerability for subsequent affective episodes (Goldstein et al., 2005). Johnson et al. (2000) postulated that even low intensity stress can precipitate mood episodes in those patients with high genetic loading. The family environment provides the most important resource for supporting the child's growth and development. Considering that pediatric bipolar disorder is associated with severe, chronic impairment in functioning, further exploration is needed to identify family environment characteristics that could be targets of useful interventions. Previous studies evaluated the family environment in adult bipolar patients and their offspring (Chang et al., 2001; Romero et al., 2005) and in affected children (Petti et al., 2004) and adolescents (Robertson et al., 2001). Chang et al. (2001) found that families that include a bipolar parent have less cohesion and organization and more conflict compared to the US normative data for the FES (Moos and Moos, 2002). Romero et al. (2005) compared the family environments of families with at least one parent suffering bipolar disorder to families with healthy parents. They found significantly lower scores on cohesion and expressiveness in bipolar families compared with healthy families. While the two studies cited here, Chang et al.

(2001) and Romero et al. (2005), suggest that bipolar disorder among adult family members affects the family environment, they do not address the possible effects of bipolar disorder among juvenile family members. Petti et al. (2004) studied the home environment and the importance of life stress events in families where the offspring had bipolar disorder but the parents did not have an affective disorder, noting psychosocial markers of risk such as need for more discipline and more negative life events in the families with affected offspring. Robertson et al. (2001) studied family interactions from the perspective of stabilized bipolar I and unipolar probands, and did not find significant differences in perceptions of family dynamics compared to a control group. Our current study used the FES scale administered to parents of bipolar children to test the hypothesis that the family environment of these children would show a dysfunctional profile, namely high conflict and low cohesion, compared with healthy comparison families. Our secondary hypothesis was that children whose parents also had a mood disorder would have a profile suggestive of greater pathology. Lastly we explored clinical characteristics (comorbidity, symptom scores, ethnicity) to identify possible associations with a dysfunctional profile. 2. Methods 2.1. Subjects The sample comprised 36 children and adolescents between 8 and 17 years of age who met DSM IV criteria (American Psychiatric Association, 2002) for bipolar disorder and 29 healthy comparison children and adolescents without current or past psychiatric or neurological disorders. These children were participants in various brain imaging studies. No subjects had current serious medical problems, and no healthy children had a positive history of any psychiatric disorder in any of their relatives who shared the home environment. This study was approved by the local Institutional Review Board. Written informed consent was obtained from parents and written assent was obtained from the children after a detailed explanation of the study requirements was provided. 2.2. Subject assessment Demographic information comprising age, gender, race, parental occupation and level of education was collected. Socioeconomic status (SES) was assessed using Hollingshead's Two Factor Index of Social Position (Miller, 1991). The diagnosis was confirmed using

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the Kiddie-SADS-Present and Lifetime Version (KSADS PL) (Kaufman et al., 1997) interview for Axis I diagnosis supplemented by the Washington University Kiddie Schedule for Affective Disorders and Schizophrenia (WASH-U-KSADS) (Geller et al., 2001) administered both to the affected children and one of their parents. Prior to conducting K-SADS-PL interviews, M.D. or Ph.D. level clinicians showed 100% agreement with a board certified faculty child and adolescent psychiatrist (R.L.O.) for the diagnoses of bipolar disorder type I or type II. Final diagnoses were assigned via consensus of our diagnostic team after integrating the child and parent interview and all other available records. Additional rating scales, including the Children's Depression Rating Scale Revised (CDRS-R) (Poznanski et al., 1985), the Hamilton Rating Scale for Depression (HAM-D) (Hamilton, 1960) and the Young Mania Rating Scale (YMRS) (Young et al., 1978) were completed by the interviewer for each subject. The Children Global Assessment Scale (CGAS) (Shaffer et al., 1983) was administered to quantify social and academic functioning. Family functioning was assessed using the FES, a 90item true false scale, the validity and reliability of which have been described (Moos and Moos, 2002). The FES was developed to evaluate family functioning in 10 areas: cohesion, expressiveness, conflict, independence, achievement orientation, intellectualcultural orientation, active-recreational orientation, moralreligious emphasis, organization and control. The first three subscales assess interpersonal relationships, the next five assess personal growth and the last two assess a system maintenance dimension. We used the Real Form (Form R) administered to one of the parents. FES subscale scores are reported as Standard Scores (Mean = 50, sd = 10) referenced to the Form I normative sample reported in the FES Manual (Moos and Moos, 2002). 2.3. Statistical analysis Data analysis was performed using SPSS for Windows software version 14.0 (SPSS, Inc., Chicago, IL). We used multivariate analysis of variance to test the hypothesis that the FES profiles of bipolar and healthy comparison families were similar. Following rejection of this hypothesis, we used univariate analysis of variance to analyze each FES subscale. In addition, we performed exploratory analyses using independent samples t-tests to compare bipolar subjects with respect to bipolar disorder subtype (Type I vs. II), medication status, presence or absence of family history of psychiatric and mood disorders, age of bipolar disorder onset (younger than

10 years vs. 10 years or older), presence or absence of comorbidities and level of functioning (CGAS score 51 vs. b 51). One-way analysis of variance was employed to analyze hospitalizations (none vs. one vs. more than one). Pearson and Spearman correlations were used to assess the association between the FES subscale scores and age of onset, length of illness, and CDRS-R, HAM-D, YMRS and CGAS scores. We adopted a twosided statistical significance of p b 0.05 for all hypothesis testing. For the primary hypothesis comparing bipolar and control families we report p-values after Bonferroni correction for multiple comparisons involving the 10 FES subscales. Bonferroni adjustments for the exploratory hypotheses were not done. 3. Results 3.1. Patient characteristics The demographic characteristics of the healthy and bipolar subjects are presented in Table 1 There were no statistically significant differences in age or SES. Most of the subjects belonged to Social Class III (Hollingshead), meaning their parents were technicians, sales workers and owners of small businesses who have completed some college. The bipolar group contained a slightly higher proportion of Caucasian males, while the healthy group contained more Hispanics, but the difference was not significant. 3.2. Clinical characteristics of bipolar patients There was a high incidence of comorbid psychiatric disorders (81%) among the bipolar subjects, most of them externalizing disorders (Table 2). Ten (27.7%) of the bipolar subjects presented a lifetime history of psychotic symptoms. Thirty-one (86%) of the patients had at least one first or second degree relative with a positive history of one or more mood disorders: Eleven
Table 1 Demographic characteristics Healthy control (n = 29) Age in years (mean sd) Male gender (%) Ethnicity African American (%) Hispanic (%) Caucasian (%) Other (%) Mean Hollingshead socioeconomic status sd 12.6 3.8 15 (51) 4 (14) 19 (66) 6 (21) 0 45.3 15.1 Bipolar (n = 36) 12.9 3.6 23 (64) 0 13 (36) 21 (58) 2 (6) 40.8 14.8

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Table 2 Clinical characteristics of bipolar subjects Characteristic Age of onset in years, median (range) Bipolar type (%) I II NOS On psychiatric medication (%) Most recent mood episode (%) Manic Hypomanic Mixed Depressive No data Presence of suicide attempts (%) Presence psychiatric comorbidities (%) ADHD ODD/CD Any anxiety disorder Hospitalizations (%) CGAS Score (%) 51 b 51 Family history of psychiatric disorders (%) Number (n = 36) 8 (316) 20 12 4 24 6 2 8 13 5 7 29 23 17 33 17 (56) (33) (11) (67) (17) (6) (22) (36) (14) (19) (81) (64) (47) (92) (47)

10 (28) 26 (72) 35 (97)

(30%) patients had a parent with a diagnosis of bipolar disorder, and 14 (39%) had a parent with major depressive disorder. Nine patients (25%) had a grandparent with a history of bipolar disorder, and 9 (25%) had a grandparent with major depressive disorder. Other diagnoses found in relatives were ADHD (28%) and alcohol and drug abuse (67%). Psychiatric conditions in siblings was not assessed. 3.3. Family environment of bipolar and healthy subjects The family environment profile of families with bipolar patients was significantly different from that of

the comparison families. The multivariate analysis produced a highly significant main effect for diagnosis (Wilks' lambda = 0.51, F = 5.1, df = 10, 54, p b 0.001). Post hoc univariate analysis showed that families with bipolar children scored significantly lower on cohesion ( p b 0.001), expressiveness ( p = 0.005), intellectual cultural orientation ( p = 0.04) and active-recreational orientation ( p b 0.001), and they scored significantly higher on conflict ( p b 0.001) compared to the comparison families with no mentally ill family members (refer to Table 3 and Fig. 1). Families of bipolar children in which one or both parents also had a diagnosis of any mood disorder scored significantly lower on: cohesion (mean sd = 36.1 20.6 vs. 53.2 10.1, p = 0.014) and organization (mean sd = 46.1 11.0 vs. 58.0 10.1, p = 0.013) compared with families where the patients had no family history of mood disorders. Spearman's rho correlation coefficients indicated a significant inverse correlation between length of illness and cohesion (rS = 0.47; p = 0.004) but not any other subscale. Age of onset was not significantly related to FES scale scores. Pearson correlations indicated no significant relation between FES subscale scores and other measures of illness severity, including CGAS, HAMD, CDRS-R and YMRS scores. There was no significant difference between bipolar types I and II with regard to any of the FES scores. There also were no significant differences on FES scores between subgroups of bipolar subjects with and without the most frequent comorbidities, including attention deficit hyperactivity disorder (ADHD), oppositional defiant disorder (ODD), conduct disorder (CD), generalized anxiety disorder (GAD), separation anxiety and phobias. T-test analyses further indicated that use of psychotropic medications and the presence of psychotic

Table 3 Mean sd FES scores of families with a bipolar child and healthy comparison families Healthy (n = 29) Mean sd FES subscale Cohesion Expressiveness Conflict Independence Achievement Orientation Intellectual-cultural Orientation Active-recreational Orientation Moral-religious emphasis Organization Control 59.5 9.0 57.5 7.7 43.7 7.9 45.5 12.1 50.6 8.3 55.0 10.0 54.0 7.0 62.0 8.3 50.2 13.0 54.0 8.4 Range 2565 4071 3360 2969 3566 1969 4369 3671 3269 2765 Bipolar (n = 36) Mean sd 38.4 20.3 51.1 9.5 58.9 14.8 41.8 12.1 47.4 9.9 49.1 11.0 43.1 11.6 57.7 9.3 47.7 11.6 58.1 8.6 Range 465 3471 3380 1361 2266 1963 2364 3271 2669 4376 F 26.9 8.6 25.1 1.5 2.0 4.5 18.5 3.8 0.6 3.6 P b 0.001 0.005 b 0.001 0.23 0.16 0.04 b 0.001 0.06 0.42 0.06

Univariate analysis of variance with df = 1, 63. Significance after Bonferroni correction.

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Fig. 1. FES profiles of bipolar and comparison families. Mean standard scores for each subscale are shown. Bipolar families scored significantly lower on C, EX, ICO and ARO and higher on CON than comparison families. C cohesion, EX expressiveness, CON conflict, IND independence, AO achievement orientation, ICO intellectualcultural orientation, ARO active-recreational orientation, MRE moral-religious emphasis, ORG organization, CTL control.

symptoms were not significantly related to the FES subscale scores. We found no significant association between history of suicide attempts or antecedent hospitalizations with any of the FES subscales scores. We used multivariate analysis of variance to compare the FES profiles of Hispanic and Caucasian families. For both healthy and bipolar families the Hispanic families scored significantly lower on independence. The healthy Hispanic families had a lower mean independence score (42.5 11.0 vs. 55.7 12.0) compared to the healthy Caucasian families ( p = 0.02). The bipolar Hispanic families had a lower mean independence score of 35.2 14.3 vs. 45.4 9.3 compared to the bipolar Caucasian families ( p = 0.016). 4. Discussion We found significant differences between the family environments of bipolar children and demographically matched comparison children. Our results show that the families of bipolar children are characterized by low levels of cohesion, expressiveness, intellectualcultural orientation, active-recreational orientation and by high levels of conflict. Low cohesion (the support and help that family members give to each other) and high conflict are consistent findings among previous studies that evaluated families where one or both parents had ever received a diagnosis of bipolar disorder (Chang et al., 2001; Romero et al., 2005). Low levels of expressiveness (the extent to which family members are encouraged to express their feelings directly) also have been reported in families that include a bipolar parent (Romero et al., 2005). Our exploratory analysis showed further that scores on the

cohesion and organization subscales were significantly lower in those bipolar families where parents had a history of mood disorders. A cross-sectional study such as ours cannot determine whether these factors contribute to the illness or are the result of the mood instability within a family member. Considering the high incidence of mood disorders in the parents, this sample of bipolar children may share characteristics of bipolar disorder described in studies of offspring of bipolar parents. Diminished support from mentally ill parents could influence the personal growth, development and individual achievement of their children. Cohesion, expressiveness and intellectualcultural orientation are closely associated with children's cognitive and social development (Bullock and Pennington, 1988; Gottfried and Gottfried, 1984).It is also important to keep in mind the dynamic nature of the family unit. As described in the literature, offspring of bipolar parents tend to develop a severe phenotype of the disorder (Lewinsohn et al., 2003; Perlis et al., 2004; Carlson et al., 2002). This appears to be the case in our sample, where subjects had early age of onset, high incidence of manic and mixed episodes, presence of psychotic symptoms, histories of hospitalizations, numerous comorbidities, and low global functioning. The literature shows that problems in the family environment are associated with ADHD (Presman et al., 2006) as well as conduct disorder and oppositional defiant disorder (Rey et al., 2000). The stress of having a child with emotional and behavioral problems implies changes in family organization in order to respond adaptively. The problem, however, could be compounded by the high levels of psychiatric comorbidity in the parents. As reviewed by Moffit (1993), children with challenging emotional and behavioral problems are not always born into ideal family circumstances. Low levels of family cohesion, intellectual and recreational orientation could, therefore, result from parental as well as child psychopathology. It would be useful for future studies to determine whether the impairment in individual and family functioning is related more to the child's bipolar diagnosis or is a consequence of the bipolar parent who may model inappropriate coping skills and fail to develop appropriate parental skills. The literature has described characteristics like less maternal warmth (Geller et al., 2000) and less attachment in parental relations among bipolar parents (Alloy et al., 2005). Our results support the importance of considering the family context in the therapeutic approach taken in treating the bipolar child. Working on the dynamics of family interactions could be an appropriate target for psychosocial intervention. A good example of a psychosocial intervention that can yield positive results is the

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C. Belardinelli et al. / Journal of Affective Disorders 107 (2008) 299305 Althoff, R.R., Faraone, S.V., Rettew, D.C., Morley, C.P., Hudziak, J.J., 2005. Family, twin, adoption, and molecular genetic studies of juvenile bipolar disorder. Bipolar Disord. 7, 598609. American Psychiatric Association, 2002. Multiaxial assessment. American Psychiatric Association DSM IV-TR: Diagnostic and Statistical Manual of Mental Disorders. American Psychiatric Association, Washington, DC. Bullock, J.R., Pennington, D., 1988. The relationship between parental perceptions of the family environment and children's perceived competence. Child Study J. 18, 1731. Carlson, G.A., Bromet, E.J., Driessens, C., Mojtabai, R., Schwartz, J.E., 2002. Age at onset, childhood psychopathology, and 2-year outcome in psychotic bipolar disorder. Am. J .Psychiatry 159, 307309. Caspi, A., Sudgen, K., Moffitt, T.E., Taylor, A., Craig, I.W., Harrington, H., McClay, J., Mill, J., Martin, J., Braithwaite, A., Poulton, R., 2003. Influence of life stress on depression: moderation by a polymorphism in the 5-HTT gene. Science 301, 386389. Chang, K.D., Blasey, C., Ketter, T.A., Steiner, H., 2001. Family environment of children and adolescents with bipolar parents. Bipolar Disord 3, 7378. Cooke, R.G., Young, L.T., Mohri, L., Blake, P., Joffe, R.T., 1999. Family-of-origin characteristics in bipolar disorder: a controlled study. Can. J. Psychiatry 44, 379381. Geller, B., Bolhofner, K., Craney, J.L., Williams, M., DelBello, M.P., Gundersen, K., 2000. Psychosocial functioning in prepuberal and early adolescent bipolar disorder phenotype. J. Am. Acad. Child Adolesc. Psychiatry 39, 15431548. Geller, B., Zimerman, B., Williams, M., Bolhofner, K., Craney, J.L., DelBello, M.P., Soutullo, C., 2001. Reliability of the Washington University in St. Louis kiddie schedule for affective disorders and schizophrenia (WASH-U-KSADS) mania and rapid cycling sections. J. Am. Acad. Child Adolesc. Psychiatry. 40 (4), 450455. Goldstein, T.R., Birmaher, B., Axelson, D., Ryan, N.D., Strober, M.A., Gill, M.K., Valeri, S., Chiappetta, L., Leonard, H., Hunt, J., Bridge, J.A., Brent, D.A., Keller, M., 2005. History of suicide attempts in pediatric bipolar disorder: factors associated with increased risk. Bipolar Disord 7, 525535. Gottfried, A.W., Gottfried, A.E., 1984. Home environment and cognitive development in young children of middle socioeconomic states families. In: Gottfried, A.W. (Ed.), Home Environment and Early Cognitive Development. Academic Press, New York, pp. 57115. Hamilton, M., 1960. A rating scale for depression. J. Neurol. Neurosurg. Psychiatry 23, 3662. Johnson, L., Andersson-Lundman, G., berg-Wistedt, A., Math'e, A.A., 2000. Age of onset in affective disorder: its correlation with hereditary and psychosocial factors. J. Affect. Disord. 59, 139148. Johnson, L., Lundstrom, O., Aberg-Wistedt, A., Mathe, A.A., 2003. Social Support in bipolar disorder : its relevance to remission and relapse. Bipolar Disord 5 (2), 129137. Johnson, S.L., Winett, C.A., Meyer, B., Greenhouse, W.J., Miller, I., 1999. Social support and the course of bipolar disorder. J. Abnorm. Psychol. 108 (4), 558566. Kaufman, J., Birmaher, B., Brent, D., Rao, U., Flynn, C., Moreci, P., Williamson, D., Ryan, N., 1997. Schedule for affective disorders and schizophrenia for school-age children-present and lifetime version (K-SADS-PL): initial reliability and validity data. J. Am. Acad. Child Adolesc. Psychiatry 36, 980988. Kim, E.Y., Miklowitz, D.J., 2004. Expressed emotion as a predictor of outcome among bipolar patients undergoing family therapy. J. Affect. Disord. 82 (3), 343352.

Multi Family Psychoeducation Group and Individual Family Psychoeducation Programs developed by Lofthouse and Fristad (2004). These programs are applied in bipolar disorder with early onset, a condition conceptualized by the authors as a biopsychological entity, where caregivers experience their parenting role as highly stressful. These interventions consider general objectives like increasing knowledge of the disorder, symptoms, and medications, increasing coping and problem-solving skills, and improving peer and family relationships. Child-and family-focused cognitive behavioral therapy, specifically designed for pediatric bipolar patients by Pavuluri et al. (2004), and Miklowitz's family focused therapy for adolescents with bipolar disorder (Miklowitz et al., 2004) are other examples of interventions that employ an adjunctive psychosocial approach. Our study noted cultural differences of the sample, as Hispanic families on the whole had lower scores on the independence subscale. Cultural attitudes regarding independence are complex and beyond the scope of this paper (Oyserman et al., 2002). FES items that contribute to high independence scores include doing things on our own and relying on themselves (Moos and Moos, 2002). In our context, lower independence seems to be a cultural characteristic rather than a marker of pathology. Moos and Moos (2002) described a profile of Latin families with higher scores on achievement orientation, moralreligious emphasis and organization and lower score on expressiveness. Due to its case-control design, our study cannot determine whether the observed indicators of dysfunction persist or continue to evolve over time. Future longitudinal studies without these limitations may clarify the specific role and impact of child psychopathology on the family dynamic as well as determine those factors from the environment that may influence the evolution and prognosis of the disorder.
Role of funding source This work was partly supported by MH 01736, MH 069774, RR 020571, K23-MH068280,UTHSCSA GCRC (M01-RR-01346), the Krus Endowed Chair in Psychiatry (UTHSCSA), and Capes Foundation (Brazil). Conflict of Interest Dr. Olvera is on the speaker's bureau for McNeil Pediatrics, Janssen Pharmaceuticals, Shire Pharmaceuticals, and AztraZeneca.

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