Familial Aggregation of Adolescent Personality Disorders

Familial Aggregation of Adolescent Personality Disorders BARBARA A. JOHNSON, M.D., DAVID A. BRENT, M.D., JOHN CONNOLLY, M.A., JEFF BRIDGE, B.A., B.S., JAMES MATTA, M.A., DOREEN CONSTANTINE, M.ED., CHRIS RATHER, AND TINA WHITE

ABSTRACT Objective: A family study of DSM-III-R personality disorders was conducted in the families of 66 clinically referred

adolescents to examine the validity of personality disorder diagnoses in adolescents. Method: Semistructured interviews of Axis I and II disorders, including the Structured Clinical Interview for DSM-III-R Personality Disorders, were used to directly interview 66 clinically referred adolescents and their adult first-degree family members, combining family study and family history data. Results: The relatives of adolescents with avoidant personality disorder had an increased prevalence of avoidant and cluster A (schizoid, schizotypal, and paranoid) personality disorders. The relatives of adolescents with borderline personality disorder demonstrated increased rates of borderline and avoidant personality disorders, even after adjusting for comorbidity. Conclusions: The results of this study support the validity of Axis II diagnoses, particularly avoidant and borderline disorders, in adolescents. J. Am. Acad. Child Ado/esc. Psychiatry, 1995,

34, 6:798-804. Key Words: personality disorder, borderline personality disorder, avoidant personality disorder, family study, SCID-II.

There has been a gradual acceptance that personality disorders can be diagnosed reliably in adolescence (Brent et al., 1990; Kutcher et al., 1990; Marton et al., 1989; McManus et al., 1984). However, there have been few investigations into the validity of adolescent personality disorder diagnoses. Extant studies support the convergent validity of adolescent personality disorders which have been correlated with increased cognitive distortion, suicide, and suicidal behavior; poor treatment response; and dimensional measures of personality trait (Brent et al., 1993, 1994; Marton et al., 1989; Marttunen et al., 1991). However, the standard approach for the establishment of the validity for psychiatric diagnostic entities (Robins and Guze, 1970)

Accepted November 15, 1994. From the University ofPittsburgh, Western PsychiatricInstitute and Clinic. This work was supported by NIMH grant MH43366. A previous version of this paper was presented as a poster at the Annual Meeting of the Society for Research in Child and Adolescent Psychopathology. Albuquerque, NM, February 19, 1993. The assistance ofSylvia Bartle, MS. W, in the interviewing and Carol Kostek in the preparation of this manuscript is gratefully acknowledged. Reprint requests to Dr. Johnson, University ofPittsburgh, WesternPsychiatric Institute and Clinic, 3811 O'Hara Street, Pittsburgh, PA 15213-2593; telephone: (412) 624-1240. 0890-8567/95/3406-0798$03.00/0©1995 by the American Academy of Child and Adolescent Psychiatry.

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has not been applied to the study of adolescent personality disorders. This standard validation consists of four elements: the demonstration of a consistent course, response to treatment, biological markers, and familial aggregation. In this article, we focus on the issue of familial aggregation as a validator for adolescent personality disorders. This approach has been used to validate other entities in childhood and adolescence, namely early-onset affectiveillness and anxiety disorders (e.g., Last et al., 1991; Puig-Antich et al., 1989). The heritability of personality traits has been supported by twin studies, in which personality traits of adult monozygotic and dizygotic twins showed substantial similarities in proportion to amount of shared genetic material, regardless of whether they were reared together or apart (Kendler et al., 1993; Tellegen et al., 1988). However, the generalizability of these findings to the heritability of personality disorder, while plausible, has not been established, since diagnostic entities of personality disorder were not investigated in the abovenoted samples. Studies of the familial aggregation of personality disorders in adult patients have been suggestive, but not definitive. Soloff and Milward (1983), in a chart review, found an increased prevalence of "eccentric or peculiar behavior" in the first-degree relatives of the

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borderline subjects, compared with the relatives of depressive or schizophrenic patients. However, they did not report directly on the prevalence of personality disorders. Pope et al. (1983) performed a chart review and found that adult subjects with borderline personality disorder, compared with subjects with bipolar or schizophrenic disorders, had higher rates of histrionic, borderline, and antisocial personality disorders in their first-degree relatives. However, they did not directly assess the family members for personality disorder. In a family history study, Pfohl et al. (1984) found that adult probands with major depression and comorbid personality disorders had more relatives with antisocial personality disorder than those probands with major depressive disorder who did not have comorbid personality disorders. However, the prevalence of other personality disorder diagnoses in the relatives were not reported. There is also evidence supporting the familial aggregation of dependent personality disorder and avoidant and dependent traits using a standardized family history method including most but not all symptoms of avoidant and dependent personality disorders (Reich, 1991a,b). However, the rates of these personality traits and disorders were equally elevated in the relatives of patients with affective and anxiety disorders who did not have personality disorders, suggesting that dependent and avoidant personality disorders may be part of a group of affective and anxiety "spectrum" disorders. Alternately, these data suggest that avoidant and dependent traits may predispose to the development of these Axis I disorders. In light of the extant literature, we wish to report on a family study of personality disorders in the adult relatives of adolescent inpatients with and without personality disorder. This study, in distinction to many studies in the literature, uses a family study methodology involving structured direct assessment of family members by clinicians blind to proband diagnoses. We examined the following questions: (1) Is personality disorder in adolescent probands associated with higher rates of personality disorder in adult first-degree relatives? (2) More specifically, do cluster A, B, and C personality disorders show familial aggregation? (3) Finally, when examining individual personality disorders, is there any evidence of more specific patterns of familial transmission?

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METHOD Sample The probands for this family study were 37 suicide attempters and 29 never-suicidal psychiatric controls previously described (Brent et al., 1993). Probands were between the ages of 13 and 19 years and inpatients at Western Psychiatric Institute and Clinic. The suicide attemprers had made a suicide attempt within 1 year of admission, had shown actual suicidal intent, and had engaged in self-destructive behavior of clear lethality. Therefore, excluded from the suicidal group were adolescents whose behavior was only self-mutilative in nature. The never-suicidal group consisted of adolescent patients who had never engaged in suicidal behavior nor had suicidal ideation with a plan. To be considered for the study, both groups had to have at least one parent who was available for direct interview and willing to participate in the study. Therefore, patients in protective custody, foster placement, and, in many instances, those referred from residential facilities were ineligible for study. Other exclusionary criteria were IQ less than 70, inability to cooperate with the interview because of delirium or psychosis, diagnosis of a chronic medical illness, or an eating disorder. Of 98 eligible adolescents and families approached, 66 agreed, for an overall compliance rate of 68%. The group who agreed to the study, compared with those who refused, did not differ in age, ethniciry, gender, or admitting diagnosis. On average, four family members were directly interviewed per adolescent proband (mean = 3.9, SD = 1.6). There were no significant differences in the number of family members interviewed for attempter versus control families (4.0 [1.7] versus 3.8 [1.5]). There was also no difference in the mean number of family members interviewed for the adolescents with personality disorder versus the adolescents with no personality disorder (4.0 [1.7] versus 3.7 [1.4]), nor were there differences in the age or gender of the relatives on the basis of either suicidality or personality disorder status of the adolescent proband. Comparing the rate of direct interview of the living first-degree relatives of the adolescents with personality disorder versus that of the adolescents with no personality disorder, there were no significant differences: 96% versus 94% of me mothers, respectively, were directly interviewed, 49% versus 65% of fathers, and 71% versus 42% of the adult siblings.

Assessment Instruments The probands were all assessed for the presence of current and past psychiatric disorders using the Schedule for Affective Disorders and Schizophrenia for School-Age Children-Epidemiologic (Orvaschel et al., 1982) and Present Episode (Chambers et aI., 1985) versions (K-SADS-E and K-SADS-P). All adult first-degree relatives were assessedfor the presence ofpast and current psychiatric disorder by use of the Schedule for Affective Disorders and SchizophreniaLifetime Version (SADS-L) (Endicott and Spitzer, 1978). Both the K-SADS-E/P and the SADS-L provide a detailed description of past and current psychopathology including age at onset, number of episodes and severity, and DSM-III diagnoses. The SADS-L was supplemented with portions of the K-SADS-E to ascertain the presence and age at onset of childhood psychiatric disorders not included in the SADS-L. In the present study, interrater agreement was assessed for 47 adult and 8 adolescent subjects; a high level of agreement was found: for the SADS-L the overall mean 1C = .95 (SE = .03), for the K-SADS the overall mean 1C = .99 (SE =

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JOHNSON ET AL.

.01). An important part of the study design was to perform family interviews blind to proband diagnosis and vice versa. Consequently, the number of interrater reliability interviews among the adolescents was limited because of the need to maintain blindness to proband diagnoses. The Structured Clinical Interview for the DSM-III-R diagnosis of personality disorder (SCID-II) (Spitzer et aI., 1989) was used for the assessment of Axis II disorders in both the adolescent probands and their adult first-degree relatives. This instrument is organized according to DSM-III-R criteria for each personality disorder. Each symptom can be rated as absent (0), present (1), and present and clearly functionally impairing (2). To fulfill definite criteria for a disorder, a subject must have the requisite number of symptoms at a "2" level with documentation by the interviewer of examples of such behavior. Subjects were given a diagnosis of a probable personality disorder if the total count of symptoms per disorder at a "2" level was one short of the requisite number necessaty for the definite diagnosis, and functional impairment was evident. For example, for a patient to fulfill criteria for borderline disorder, five symptoms at a "2" level are required. Therefore, four symptoms at a "2" level would be necessaty to be considered probable borderline personality disorder. The SCID-II interview was modified slightly to adapt the language to adolescents, and screening questions were developed so that the entire interview did not necessarily have to be administered. For the first 20 interviews, both the screening questions and the entire interview were administered. It was determined that the screening questions were both sensitive and specific. Thereafter, an entire interview section was administered only if the subject endorsed one or more of the screening questions. Previous studies have found this instrument to be reliable in the diagnosis of personality disorder in adults (Spitzer et al., 1989). In the present study, interrater agreement was assessed for 29 adult and 8 adolescent subjects, and there was a high level of agreement: for the SCID-II (overall mean 1C = .91 [SE = .07] for the adults; overall mean 1C = .97 [SE = .03] for the adolescents). Multiple researchers have reviewed the issues related to the classification of personality disorders (Frances, 1982; Frances and Widiger, 1986; Klein, 1993). Frances and Widiger (1986) view personality disorders as dimensional constructs; therefore, they advocate the identification of a cutoff point that is "clinically meaningfuL" Our choice of cutoff point for "probable disorder" is clinically relevant to the construct of personality disorder, insofar as the same criteria for chronicity and severity were required, i.e., duration greater than 2 years and association with significant functional impairment. As preliminary validation of these constructs, in related studies, we have shown that both definite and probable personality disorder were associated with adolescent suicide attempts and completions (Brent et al., 1993, 1994).

Diagnostic Process Interviews were conducted by rnaster's-level clinicians using semistructured interviews. The caretaking parent, as well as the adolescent proband, served as the informants for the adolescent probands. Adolescents were interviewed about themselves, and in a separate interview their parents were also interviewed about the adolescents. Any discrepancies between informants were resolved by reinterview or by additional informants. The same interviewer assessed for the presence of Axis I and Axis II psychopathology in all probands. For the most part, this procedure was followed in interviews with relatives as well. In the few cases in which the interviews of the relatives were completed by two different people,

800

the SCID-II interviewer reviewed the SADS-L in detail (often with the other interviewer) before conducting the Axis II interview. Information from different informants was combined in diagnostic conferences chaired by either of the senior authors (B.A.]. or D.A.B.). The importance given to an informant was based on three factors: likelihood that informant would actually have knowledge of the information conveyed, internal consistency in informant's report, and evidence of concrete examples to support this diagnostic viewpoint. In addition, first- and second-degree relativesserved as informants for the first-degree relatives, using the family history method. Interviews and diagnoses on family members were rendered by clinicians blind to the diagnoses of the adolescent proband. Diagnoses were rendered by consensus in diagnostic conferences, preserving blindness between proband and relative diagnosis. Most importantly, Axis II diagnoses were made only when it was clear that the personality symptoms were independent of the Axis I disorder. DSM-III criteria were used for Axis I disorders and DSM-III-R criteria for Axis II disorders (American Psychiatric Association, 1980, 1987).

Data Analysis Although there is substantial evidence for assortative mating among psychiatric patients (Merikangas et al., 1988), we found no evidence of assortative mating for any of the personality disorders in question between the mothers and the fathers. Log-linear analyses were used to learn whether the relationship between proband and relative personality disorder was affected by group membership (atternpter versus control). There was no evidence of three-way interactions among suicide attempt, proband personality disorder, and relative personality disorder. Therefore, proband suicide attempt was not controlled for in our subsequent analyses. The raw rates of personality disorder among adult firstdegree relatives were compared between relatives with and without any personality disorder. Additional comparisons involving specific personality disorders were also performed. Raw rates were used because age of onset has been viewed in the past as fixed at 18 years of age. There was no evidence of assortative mating between parents for personality disorder, either in the broadest or most specific categories. Moreover, when the data were analyzed per family unit, rather than using raw rates among first-degree relatives, the results were unchanged. Those who met criteria for personality disorder ("disorder" hereafter includes all diagnoses that meet the criteria for definite or probable personality disorder, as described above) within a certain cluster or a specific personality disorder were compared, using the X2 test, with those who had no personality disorder. Because the majority of avoidant and borderline adolescents (63% and 88%, respectively) had more than one personality disorder, we conducted two separate analyses,comparing relatives of probands having avoidant and borderline personality disorders with (1) the relatives of probands with no personality disorder, and (2) the relatives of probands who did not have avoidant or borderline personality disorders, respectively. The Fisher's Exact Test was used when any cell value was zero. Log-linear analyses were then used to explore associations between relative and proband personality disorder, while controlling for other interactions in the data.

RESULTS

The adolescents with personality disorder did not differ significantly from the adolescents without personality disorder with respect to age, gender, ethnicity,

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TABLE 1 Demographic Characteristics of the Adolescent Probands with Personality Disorder versus Those with No Personality Disorder (n = 66) Any Axis II (n = 49) Age: mean (SD) yr Sex (% female) Ethnicity (% white/black/other)

16 (1.6)

15 (1.9)

41

35

90/6W

88/12/0

SESb I II III IV V County (% Allegheny) Family lives with Both biological parents One biological parent Other

Proband No Axis II (n = 17)

o

10 27 31 22 10 61

18 6 77

43

35

53

59 6

4

47

29

Note: There were no significant differences between the two groups on the above characteristics. a

b

This group includes one Korean-American and one Vietnamese-American proband. Socioeconomic status (Hollingshead, 1975).

socioeconomic status, family constellation, or rurality of the county in which the adolescent resided (Allegheny county was the most urban) (Table 1). The adolescents with personality disorder had a higher rate of suicide attempts than those without a personality disorder (65% versus 35%, X2 = 4.01, P = .05); however, there were no significant differences in the rates of major psychiatric disorders between the two groups (Table 2). Table 3 shows the relationship between adolescent personality disorder diagnosis and first-degree relative personality disorder diagnosis. Adolescents with Any Personality Disorder

There was no significant difference in the frequency of personality disorder in the adult relatives of probands with personality disorders compared with relatives of probands without personality disorder (41% versus 26%). Cluster A, S, and C Personality Disorders

To examine the specificity of association of Axis II disorder clusters and specific Axis II disorders within families, we subdivided the adolescent proband group by the presence or absence of each of the following: cluster A (schizoid, schizotypal, or paranoid), cluster B (borderline, narcissistic, histrionic, or antisocial),

J. AM. ACAD. CHILD ADOLESC. PSYCHIATRY, 34:6, JUNE 1995

and cluster C (passive-aggressive, avoidant, compulsive, or dependent). Cluster A. There was no specific association between cluster A in the adolescent probands and their relatives (7% versus 13%). Cluster B. Among the relatives of adolescents with cluster B disorders, cluster B disorders were not significantly more prevalent than in the relatives of the adolescents with no personality disorder (17% versus 7%). Cluster C. Relatives of the adolescents with cluster C disorders had a higher prevalence of any Axis II disorder in their adult first-degree relatives (43% versus 26%, X2 = 3.89, P = .05) than those of adolescents with no disorder. Individual Personality Disorders

The familial aggregation of avoidant and borderline personality disorders was informative and is examined in detail below. In contrast, no relationship was observed in this sample between paranoid, schizoid, schizotypal, histrionic, passive-aggressive, dependent, or self-defeating personality disorder and the same or other individual personality disorders or clusters. Avoidant Personality Disorder

AvoidantAdolescents versus Adolescents without Personality Disorder. The first-degree adult relatives of avoid-

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JOHNSON ET AL.

TABLE 2 Axis I Characteristics of the Adolescent Probands with Personality Disorder versus Those with No Personality Disorder

Mfective disorder Major depression Bipolar spectrum Affective with nonaffective comorbidity Substance abuse Attention deficit disorder Any anxiety disorder Any conduct disorder Suicide attempt

*p

=

Any Axis II

Proband No Axis II

78

59 47

69 25

47 35

18

35 29

18

18

35

24

57 63*

71

35

.05.

ant adolescents demonstrated a higher prevalence of any personality disorder (55% versus 26%, X2 = 8.93, P = .003), cluster C (40% versus 15%, X2 = 7.98, P = .005), and obsessive-compulsive personality disorder (13% versus 2%, X2 = 3.98, P = .05) than those of adolescents without a personality disorder. Avoidant disorder showed a nonsignificant trend toward a higher rate in the adult first-degree relatives of the adolescents with avoidant disorder (18% versus 7%). There was a higher lifetime prevalence of affective disorder (96% versus 59%, X2 = 8.68, P = .003) and anxiety disorder (54% versus 24%, X2 = 3.85, P = .05) and lower rates of conduct disorder (38% versus 71%, X2 = 4.36, P = .04) in the adolescents with avoidant disorder than in the adolescents with no personality disorder; therefore, these variables were incorporated into our log-linear analyses and no relationship was found between proband and relative avoidant personality disorder. Avoidant Adolescents Compared with Nonavoidant Adolescents. The relatives of the avoidant probands showed higher rates of any personality disorder (55% versus 28%, X2 = 12.58, P = .0004), cluster A (19% versus 8%, X2 = 4.82, P = .03), cluster C (40% versus 17%, X2 = 11.53, P = .0007), and avoidant disorder (18% versus 7%, X2 = 4.61, P = .03). Borderline Personality Disorder

Borderline Adolescents Compared with Adolescents with No Personality Disorder. The first-degree adult relatives of adolescents with borderline personality disorder demonstrated a higher prevalence of any personality disorder (59% versus 26%, X2 = 9.43, P = .002), cluster 802

C (44% versus 15%, X2 = 8.39, P = .004), and borderline disorder (10% versus 0%, Fisher's Exact Test, p = .04) than those of adolescents without a personality disorder. Borderline Adolescents Compared with Nonborderline Adolescents. In comparing probands with and without borderline personality disorder, the relatives of probands with borderline disorder showed higher rates of any personality disorder (59% versus 31%, X2 = 10.28, P = .001). There was a higher prevalence, but not significantly so, of borderline disorder in the first-. degree relatives of the borderline adolescents (10% versus 3%) than in the relatives of the adolescents without borderline personality disorder.

DISCUSSION

Many of our initial hypotheses were supported. As expected, adolescents with avoidant and borderline personality disorder had first-degree adult relatives with increased prevalence of avoidant and borderline personality disorders, respectively. While our results are consistent with the familial aggregation studies of personality disorder in the adult literature (Pope et al., 1983; Reich, 1991a,b; Soloff and Milward, 1983), we emphasize that this is the first study to our knowledge that examined the family aggregation of personality disorders using a family study methodology, therefore, allowing one to make stronger inferences about the familial nature of certain personality disorders than was possible from previous studies (Pope et al., 1983; Reich, 1991a,b; Soloff and Milward, 1983). In addition to the methodological strength of the family study method, there are some limitations to this particular study. As is often the case in family studies, fathers were less likely to be the custodial parent after a divorce or separation and, therefore, were less likely to be directly interviewed than were other family members. However, by design our study supplemented direct interview information with structured family history interviews to generate best-estimate diagnoses. Our modest sample size may have resulted in our ability to identify only the most robust associations. However, our small sample size does not increase the chance of making a type II error, because small samples require a larger difference between the two groups to be significant at the same p values.

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TABLE 3 Relationships between Adolescent Proband Axis II Diagnosis and First-Degree Adult Relative Axis II Diagnosis Total No . of Adult First-Degree Relatives With With With With With With

cluster A PD cluster B PD cluster C PD avoidant PD borderline PD any PD

Relatives Relatives Relatives Relatives Relatives Relatives of Probands with of Probands with of Probands with of Probands with of Probands with of Probands with Cluster A PD Cluster B PD Cluster C PD Avoidant PD Borderline PD No Axis II PD (n = 42) (n = 64) (n = 112) (n = 62) (n = 39) (n = 46) 3 5 11 4 1 16

(7)

(12) (26) (10) (2) (38)

8 11 21 9 5 29

(13) (17) (33)* (14) (8) (45)*

14 16 33 15 4 48

(13) (14) (30) (13) (4) (43)*

12 9 25 11 2 34

(19) (15) (40)** (18) (3) (55)**

5 8 17 8 4 23

(13) (21) (44)** (21) (10)* (59)**

6 (13) 3 (7) 7 (15) 3 (7) 0 (0) 12 (26)

Note: Values represent number (percent). PD = personality disorder. Cluster A includes paranoid, schizoid, schizotypal PDs and traits. Cluster B includes borderline, narcissistic, histrionic, and antisocial (for adults only) PDs and traits. Cluster C includes avoidant, obsessivecompulsive, dependent, and passive-aggressive PDs and traits. Any PD or trait includes all of the above plus self-defeating PD . Significance of comparisons to the relatives of the probands with no personality disorders: * p :$ .05; **P :$ .01.

The findings of the study provide firm support for the validity of certain personality disorders in adolescents. These results also provide evidence for the specific familial aggregation of borderline , avoidant , and cluster C disorders. We did not find such evidence for the other personality disorders, but this may be attributable in part to a lack of power because of the low frequency of the other disorders in our sample. For example, while we found no evidence of familial aggregation of schizotypallschizoid personality disorders, othe r much larger studies have found evidence that schizotypall schizoid personality disorders are familial and are genetically related to schizophrenia (Baron et al., 1983, 1985; Kendler et al., 198 1, 1984). Alternately, some personality disorders may not show familial aggregation , or they may not be valid categories at all. If some of these personality disorders are familial, what are the possible mechanisms that could account for their familial aggregation? The relationship between proband and relative personality disorder could be the result of genetic transm ission or of the impact of parental personality disorder on offspring. The latter could be mediated via the impact of parent-child hostility, which has been a part icular feature of the parenting style of adults with a personality disorder (Rutter and Quinton, 1984). These results do not appear to be solely att ributable to assortative mat ing for personality disorder among the parents, for in this sample it was not evident. The absence of assortative mating for personality disorder is consistent with the findings of Heun and Maier (1993), who examined the risk for Axis I and II disorders among spouses of

psychiatric patients and community controls. Clearly, the next step in th is area is to replicate these findings in different populations. High-risk and longitudinal designs will be important types of studies to disentangle possible mechanisms. We have used familial aggregation studies as one method to validate the diagnosis of personality disorders in adolescents. The presence of familial aggregation does not explain the origin or path of the influences; therefore, other validation methods should also be explored. These includ e longitudinal studies of ph enomenology and outcome, longitudinal high-risk studies of siblings or offspring of individuals with personality disorders, studies of the convergent validity of Axis II disorders, and identification of psychobiological correlates of adolescent personality disorder.

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