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Psychosocial adjustment of children treated for anorectal anomalies
Psychosocial Adjustment of Children Treated for Anorectal Anomalies By L. L u d m a n and L. Spitz
London, England • The psychosocial adjustment of 160 children with anorectal malformations was assessed at 6 to 17 years of age in relation to levels of continence (Kelly score), Five measures of emotional and behavioural adjustment were used to assess a number of domains of child/adolescent functioning and to include measures from multiple perspectives. The psychiatric assessment of the child identified a disorder in 29% of the group overall. Based on parental assessments, behavioural maladjustment was shown in 27% of the children, and on the basis of a self-report questionnaire 24% of the children were depressed. Behavioural adjustment as rated by teachers was similar to the norms. The level of continence achieved (total Kelly score) did not appear to influence psychological adjustment, with the exception of the incontinent young girls (6 to 11 years) who were shown to be less well adjusted than the continent young girls. Differences between children showing positive versus negative adjustment were dependent on the perspective of the respondent and were also related to the child's age and gender, age of achieving continence, frequency of accidents, and parental factors.
mentally delayed and those with multiple handicaps, of which the anorectal malformations were only one component. Associated anomalies were identified in 51 (32%) of patients reviewed. There were 90 boys and 70 girls (mean age at assessment 11.3 _ 3.42 years). Eighty-one were between ages 6 and 11 years (designated as children) and 79 were aged between 12 and 17 (designated as adolescents).
Copyright © 1995 by W.B, Saunders Company
Child/Adolescent
INDEX WORDS: Anorectal anomalies, psychosocial adjustment, imperforate anus, chronic disorder, neonatal surgery.
The interview with the child/adolescent included a standardized form of psychiatric interview modelled on the Child Assessment Schedule (CAS) 6 and semistructured interviews.7 Self-report questionnaires (excluded children/adolescents with borderline intellectual functioning). (1) The Depression SelfRating Scale (DSRS) s is an 18-item self-report screening measure for affective disorders in childhood and adolescence. (2) Harter Self-Perception Profiles9 are self-completed developmentally appropriate questionnaires for assessing the children's perception of themselves, and for providing a global measure of self-worth as well as separate subscales such as social acceptance and physical appearance.
NUMEROUS technical advances in D ESPITE the surgical repair of anorectal malformations, a significant proportion of children have long-term problems with faecal continence. Little is known about the psychosocial consequences of this chronic disability, although there is extensive anecdotal evidence that faecal incontinence is the cause of distress to both the child and the family. Only three studies have previously addressed these issues. Ditesheim and Templeton 1 assessed quality of life in relation to the success or failure of attaining faecal continence, and Ginn-Pease et al2 and Diseth et aP reported on the psychological adjustment of small groups of children with imperforate anus. The purpose of this study was to assess the emotional and social adjustment of children with anorectal malformations in relation to the level of continence achieved, and to assess whether the prevalence of behavioural and emotional disturbance increases during adolescence. MATERIALS AND METHODS Of 223 patients who had their primary definitive surgery for anorectal anomalies performed at Great Ormond Street Children's Hospital between 1974 and 1987, 160 (72%) agreed to participate in the study, 52 (23%) could not he traced, and only 11 (5%) refused. Excluded were children who were known to be developJournal of Pediatric Surgery, Vo130, No 3 (March), 1995: pp 495-499
Clinical Assessment The details of each patient were reviewed and the child/ adolescent fully evaluated clinically. The Kelly scoring scale4 was used to provide a level of faecal continence score, and the level of the lesion was classified according to the Wingspread classification system. 5 Table 1 illustrates the relationship between the level of the lesion and total Kelly scores.
Psychological Assessments In addition to individual interviews with each child and his or her parents, the participants completed standardized questionnaires.
Parents This interview included detailed questions about the child's and family's methods of managing and coping with continence problems at home, at school, and socially; about the child's social and personal relationships; and about psychological and emotional symptoms in the child.
From the Department of Paediatric Surgery, Institute of Child Health and Great Ormond Street Hospital for Children NHS Trust, London, England. Presented at the 41st Annual International Congress of the British Association of Paediatric Surgeons, Rotterdam, The Netherlands, June 29-July 1, 1994. This research was funded by a grant from the Leverhulme Trust. Address reprint requests to Dr L. Ludman, Department of Paediatric Surgery, Institute of Child Health, and Great Ormond Street Hospital for Children NHS Trust, 30 Guilford St, London WCIN 1EH, England. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3003-0030503.00/0 495
496
LUDMAN AND SPITZ
Table 1. Relationship Between Level of Lesion and TKS
Psychiatric Interview
Level of Lesion: Number (%) TKS
Low
Intermediate
High
5 or 6 _<4
45 (30) 17 (12)
5 (3) 12 (8)
6 (4) 63 (43)
Total
62 (42)
17 (11)
69 (47)
NOTE. Total sample with Kelly score 148 (no TKS for 8 cases; 4 cases had a permanent colostomy).
Standardized questionnaires. The parents completed the newly revised Child Behaviour Checklist (CBCL), 1° which yields a Total Behaviour Problem score (TPS) and two subscales, referred to as Internalizing (social withdrawal, depression, anxiety) and Externalizing Symptom (antisocial, aggressive, delinquency) scales. A T score of >_60 was used as the cut-off point for defining levels of clinical significance. Self-report questionnaires. Both parents completed the 30-item general health questionnaire 11 for assessing psychiatric morbidity, and the mothers completed a marital rating measure. 12
Teachers Teachers completed the Teacher's Report Form (TRF), t3 which is modelled on the CBCL with additional items appropriate for teachers.
RESULTS
Analyses of the data were conducted to examine the psychological adjustment of (1) The study group overall; (2) Groups based on the total Kelly score (TKS) (continent: score of 5 or 6, n = 56, incontinent: score < 4, n = 92). (A total Kelly score was not available for eight children, and four had permanent colostomies); (3) The age at which continence had been achieved: Group 1 (control group) continent before the age of 4 (n = 40), Group 2 (late continent) continent between 5 and 17 years (n = 18), and Group 3 (incontinent, n = 102); (4) Children/ adolescents with a total Kelly score of 6 (n = 38) and children/adolescents having frequent accidents
(n
=
33).
Forty-six of the 157 children/adolescents interviewed (29%) were judged as having some psychiatric disorder based on the diagnostic interview, ranging in severity from mild in 16 (10%) to moderate in 30 (19%) (a disorder severe enough to influence their daily lives). This prevalence of 19% is significantly higher when compared with a rate of 10% in the general child population ( < .001), 14 but is similar to that found in children with chronic disorders. The proportion deemed to have a disorder tended to be higher among the adolescents (35%) than among the children (24%). There were no significant differences between the continence groups based either on their total Kelly score or age of achieving continence. However, those with persistent incontinence (frequent accidents) were more likely to receive a psychiatric diagnosis when compared with those with good control (total Kelly score of 6) (z = 1.98, P < .05).
CBCL Questionnaire Data The mean Total Problem T scores (Table 2) and the internalizing mean T scores of the young boys, both continent [2 59 (SD 13)] and incontinent [2 57 (SD 9)], were significantly higher than those of the normative population. The mean internalizing and externalizing T scores for the rest of the sample were close to the population norms. There was no difference between the group with a TKS of 6 [2 27 (SD 18)] and those with persistent soiling [2 30.15 (SD 17)]. There was some indication that those who achieved continence late were less well adjusted. The frequency distribution of the TPS, however, indicated that 39 patients (27%) had scores higher than the eighty-second percentile, the recommended cut-off point for indicating psychopathology. This
Table 2. Mean CBCL and TRF Total Problem T Scores for Continent (TKS 5 or 6) and Incontinent (TKS _<4) Groups CBCL Mean T Scores Continent
6 to 11 years Boys Girls 12 to 18 years Boys Girls Total
TRF Mean T Scores Incontinent
Continent
Incontinent
No.
Mean (SD)
No.
Mean (SD)
No.
Mean (SD)
16 17
56.75 (11.92)* 50.71 (6.29)
28 15
55.12 (10.52)* 52.67 (8.96)
17 17
48.24 (11.22) 43.53 (7.78)1"
28 11
49.50 (9.31) 52.27 (7.80)1
9 12
54.11 (10.85) 53.00 (12.65)
29 19
49.62 (12,36) 51.84 (11.30)
8 9
56.63 (8.35) 50.89 (13.35)
28 17
49.62 (12.36) 50.59 (9.51)
54
91
NOTE, Norm for mean T score = 50, SD = 10. No Kelly scores for 12 cases. No CBCL for 3 cases. No TRF for 14 cases. *Mean T score significantly higher than the norm. 1"Difference between group significant (P < .01).
51
No.
Mean (SD)
84
ANORECTAL ANOMALIES: PSYCHOSOCIAL ADJUSTMENT
497
prevalence is significantly higher (P < .01) than that of the normative population, or when compared with the ninetieth percentile norm (17% versus 10%) used in previous research of children with chronic medical disorders.
TRF Questionnaire Data The TRF mean T scores (52; 95% CI 48 to 57) of the incontinent young girls were significantly greater than those of the girls with control (43.5; 95%, CI 40 to 47, t = 2.90 [df = 26] P = .007) (Table 2). The difference between the young girls' internalizing mean T scores was also significant (P = .003). With the exception of the adolescent boys with control, the remaining groups were close to the population norms. When the TRF mean scores of those with a TKS = 6 were compared with those with persistent incontinence, the differences between the groups approached significance (F = 3.29 [df = 1,61] P = .07). There was also a tendency for the adolescents who had achieved continence late to be rated as being maladjusted. In contrast to the CBCL findings, the frequency distribution of the TPS T scores indicated that only 17% of the sampie were in the clinical range for psychopathology, that is, similar to the normative population.
Depression Self-Rating Scale The self-rating depression scale showed that overall 24% (35 of 148) of the patients rated themselves as having depressive symptoms, with children (30%) being more frequently affected than adolescents (17%). The difference between the age groups approached significance (×2 = 3.44, df = 1, P = .06). There was a tendency for the incontinent patients
in each age group to have elevated scores compared with those who were continent. Depressive symptoms, however, were no more frequent among those with persistent incontinence than among those with normal bowel control. Once again those who achieved continence late appeared to have more problems than the other groups. For example, the adolescent girls who achieved continence late had significantly higher scores on a number of the scale items including the total depression score when compared with the incontinent adolescent girls (z = -2.08, P < .04).
Self-Esteem There were no significant differences between the continent and the incontinent groups on the global self-worth measure. When the group means were examined in relation to age and sex, the incontinent adolescent boys had lower levels of self-esteem than the continent adolescent boys (P < .06). Similarly the adolescent boys with persistent soiling had significantly lower levels of global self-worth than those with good bowel control (TKS = 6) (P < .04). Multiple regression analyses, with CBCL TPS as the dependent variable indicated that family factors, parental mental health, the marital relationship, and educational levels were significantly and independently associated with outcome, indicating that good scores on the family variables best predict a lower prevalence of behaviour problems. Similarly, with the TRF Total Problem scores as the dependent variable, better parental educational levels predicted a lower prevalence of behaviour problems, with the girls predicted to have significantly lower scores than the boys (Table 3). The TKS and "frequent accidents" were not predictive of either outcome measure.
Table 3. Regression of CBCL and TRF Total Problem Scores on Sex, Age, TKS, and Two Family Variables CBCL Dependent Variable (Predictor Variables)
Standardized Regression Coefficient
Sex Age Kelly score Family score¶ Family education#
- . 14011 -.124 .118 -.322t -.176§
TRF* Unstandardized Regression Coefficient (Standard Error B) - 5.16 -0.66 1.17 -7.43 -3.77
(2.92) (0.42) (0.82) (1.8!) (1.75)
*To overcome skewed distribution, a square root transformation was carried out. t P < .001. 1:P < .01. §P < .05.
lip < .10. ¶Composite score of parental mental health and marital relationship (higher scores better). #Composite score of parental educational levels (higher scores better).
Standardized Regression Coefficient -.249t .110 -.091 -.102 -,202§
Unstandardized Regression Coefficient (Standard Error B) -1.19 .08 -.118 -.308 -.58
(.39) (.06) (,11) (.25) (.24)
498
LUDMAN AND SPITZ
DISCUSSION
Contrary to our expectations, the findings from this study have shown that the incontinent children/ adolescents were not less well adjusted than those with good bowel control. In fact, when factors such as the child's gender and parental factors such as mental health, marital relationship, and education were taken into account, no association was found between the level of continence as measured by the TKS and measures of psychological adjustment. Nevertheless, among the sample overall, the prevalence of clinically significant emotional problems was higher than expected in relation to normative populations, with a high proportion of the children showing internalizing behaviour problems as well as reporting depressive symptoms. Some differences between the continence groups were apparent. For example, the psychiatric interview with the child showed that those having frequent soiling accidents were more likely to be recognised as emotionally disturbed than those who had control (TKS of 6). Similarly, based on the teachers' assessment of behavioural maladjustment, the difference between these groups approached significance. However, neither the parental assessment nor the children's self-report depression scores indicated any significant differences between the "frequent accident" and the "good control" groups. When the sample was examined in relation to age and sex, other differences between the groups were noted. The teachers rated the incontinent young girls as showing significantly more behaviour and internalizing problems compared with those with good controL This is supported by the findings from the parents' report, which indicated a somewhat higher prevalence of behaviour and internalising problems among the young incontinent girls compared with the continent young girls, and the incontinent young girls tended to have higher self-report depression scores than the continent young girls. In addition, the parents' ratings indicated that behavioural maladjustment among both the continent and incontinent young boys was significantly greater than among the normative groups. Parental factors (mental health, the marital relationship, and educational levels) were strongly associated with outcome as measured by the CBCL, but these variables were not important explanatory variables for outcome as measured by the psychiatric interview, the teachers' ratings, or the self-report child depression scale. It is possible that parents with higher levels of psychological distress and/or lower levels of marital satisfaction are more likely to report behaviour
problems, or these children may indeed be more behaviourally maladjusted, which in itself affects marital satisfaction and parental health. None of these parental factors were related to levels of continence. Parental education levels were significantly associated with behaviour problem scores. With this exception, the findings indicated no major effect of family background factors such as social class, birth position, or number of siblings. Contrary to our predictions the incontinent adolescents were not more emotionally disturbed than those with good bowel control. Although a substantial minority received a psychiatric diagnosis (40%, when cases rated as mild were included), more than half appeared to be able to cope with the social problems associated with their disability, as well as those associated with the physical and emotional changes of puberty and emerging sexuality. The findings that children/adolescents who achieved continence late tended to be less well adjusted than the controls or incontinent group, are similar to those in studies that have shown that the relationship between the severity of chronic illness and psychological adjustment is not necessarily linear, 15 and may reflect the fact that a child with a minimal and invisible disability is expected to function as normal so as not to appear different from his/her healthy peers while the reality of his/her condition makes normal behaviour difficult in many situations, On the other hand, the more severely affected are forced to accept the reality of their situation, and moreover, others make allowances for them, and consequently they are not expected to compete with their normal healthy peers. 16 Another unexpected finding was that a higher proportion of the study sample had clinically significant scores when compared with a normative population. These findings, which are comparable with other studies of children with chronic medical disorders, suggest that parents of children who are born with anorectal anomalies overcompensate for their child's early life-threatening problems. As we found in an earlier longitudinal study of children who underwent major neonatal surgery, 17 mothers tended to be ineffective and inconsistent in their responses to their children's behaviour, and this is known to be detrimental to future emotional and behavioural adjustment. In conclusion, this study, which is the first to examine in detail the relationship between faecal incontinence and emotional adjustment in a large
ANORECTAL ANOMALIES: PSYCHOSOCIALADJUSTMENT
499
group of children with anorectal anomalies, offers some encouragement to the children and their families, but it also indicates that these children, regardless of the level of continence, are at risk for difficulties with psychosocial adjustment. Outcome and compliance with treatment may be enhanced if, from an early age, psychological evaluation of the child and
experienced advice and guidance for the families become an integral part of their continuing care. ACKNOWLEDGMENT The authors thank Mr Kiely, Mr Drake, Mr Brereton, and their senior registrars for their contribution, and all the children and their parents who took part in the study.
REFERENCES 1. Ditesheim JA, Templeton Jr JM: Short-term v long-term quality of life in children following repair of high imperforate anus. J Pediatr Surg 22:581-587, 1987 2. Ginn-Pease ME, King DR, Tarnowski KJ, et al: Psychosocial adjustment and physical growth in children with imperforate anus or abdominal wall defects. J Pediatr Surg 26:1129-1135, 1991 3. Diseth T, Emblem R, Solbraa I, et al: A psychosocial follow-up of ten adolescents with low anorectal malformation. Acta Paediatr 83:216-221, 1994 4. Kelly JH: The clinical and radiological assessment of anal continence in childhood. Aust N Z J Surg 42:62-63, 1972 5. Stephens FD, Durham Smith E: Classification, identification, and assessment of surgical treatment of anorectal anomalies. Pediatr Surg Int 1:200-205, 1986 6. Hodges K, Kline J, Fitch P, et al: Child assessment schedule: A diagnostic interview for research and clinical use. Catalog of Selected Documents in Psychology, vol 11 (issue 56), 1981 7. Rutter M, Graham P: The reliability and validity of the psychiatric assessment of the child. 1. Interview with the child. Br J Psychiatry 114:563-579, 1968 8. Birleson P: The validity of depressive disorder in childhood and the development of a self-rating scale: a research note. J Child Psychol Psychiatr 22:73-88, 1981 9. Harter S: Manual for the Self-Perception Profile for Children. University of Denver, Colorado, 1985
10. Achenbach TM: Manual for the Child Behaviour Checklist/ 4-18 and 1991 Profile. Burlington, VT, University of Vermont, Department of Psychiatry, 1991 11. Goldberg DF: Manual of the general health questionnaire. Windsor, England, National Foundation for Educational Research Publishing Company, 1978 12. Spanier GB: Measuring dyadic adjustment: New scales for assessing the quality of marriage and similar dyads. J Marriage Faro 38:15-28, 1976 13. Achenbach TM: Manual for the Teacher's Report Form and 1991 Profile. Burlington, VT, University of Vermont, Department of Psychiatry, 1991 14. Pearce J: Child health surveillance for psychiatric disorder: Practical guidelines. Arch Dis Child 69:394-398, 1993 15. MacLean WE, Perrin JM, Gortmaker S, et al: Psychological adjustment of children with asthma: Effects of illness severity and recent stressful life events. J Pediatr Psychol 17:159-171, 1992 16. McAnarney ER, Pless B, Satterwhite B, et al: Psychological problems of children with chronic juvenile arthritis. Pediatrics 53:523-527, 1974 17. Ludman L, Lansdown R, Spitz L: Effects of early hospitalization and surgery on the emotional development of 3 year olds: An exploratory study. Eur Child Adolescent Psychiatr 1:186-195, 1992
London, England • The psychosocial adjustment of 160 children with anorectal malformations was assessed at 6 to 17 years of age in relation to levels of continence (Kelly score), Five measures of emotional and behavioural adjustment were used to assess a number of domains of child/adolescent functioning and to include measures from multiple perspectives. The psychiatric assessment of the child identified a disorder in 29% of the group overall. Based on parental assessments, behavioural maladjustment was shown in 27% of the children, and on the basis of a self-report questionnaire 24% of the children were depressed. Behavioural adjustment as rated by teachers was similar to the norms. The level of continence achieved (total Kelly score) did not appear to influence psychological adjustment, with the exception of the incontinent young girls (6 to 11 years) who were shown to be less well adjusted than the continent young girls. Differences between children showing positive versus negative adjustment were dependent on the perspective of the respondent and were also related to the child's age and gender, age of achieving continence, frequency of accidents, and parental factors.
mentally delayed and those with multiple handicaps, of which the anorectal malformations were only one component. Associated anomalies were identified in 51 (32%) of patients reviewed. There were 90 boys and 70 girls (mean age at assessment 11.3 _ 3.42 years). Eighty-one were between ages 6 and 11 years (designated as children) and 79 were aged between 12 and 17 (designated as adolescents).
Copyright © 1995 by W.B, Saunders Company
Child/Adolescent
INDEX WORDS: Anorectal anomalies, psychosocial adjustment, imperforate anus, chronic disorder, neonatal surgery.
The interview with the child/adolescent included a standardized form of psychiatric interview modelled on the Child Assessment Schedule (CAS) 6 and semistructured interviews.7 Self-report questionnaires (excluded children/adolescents with borderline intellectual functioning). (1) The Depression SelfRating Scale (DSRS) s is an 18-item self-report screening measure for affective disorders in childhood and adolescence. (2) Harter Self-Perception Profiles9 are self-completed developmentally appropriate questionnaires for assessing the children's perception of themselves, and for providing a global measure of self-worth as well as separate subscales such as social acceptance and physical appearance.
NUMEROUS technical advances in D ESPITE the surgical repair of anorectal malformations, a significant proportion of children have long-term problems with faecal continence. Little is known about the psychosocial consequences of this chronic disability, although there is extensive anecdotal evidence that faecal incontinence is the cause of distress to both the child and the family. Only three studies have previously addressed these issues. Ditesheim and Templeton 1 assessed quality of life in relation to the success or failure of attaining faecal continence, and Ginn-Pease et al2 and Diseth et aP reported on the psychological adjustment of small groups of children with imperforate anus. The purpose of this study was to assess the emotional and social adjustment of children with anorectal malformations in relation to the level of continence achieved, and to assess whether the prevalence of behavioural and emotional disturbance increases during adolescence. MATERIALS AND METHODS Of 223 patients who had their primary definitive surgery for anorectal anomalies performed at Great Ormond Street Children's Hospital between 1974 and 1987, 160 (72%) agreed to participate in the study, 52 (23%) could not he traced, and only 11 (5%) refused. Excluded were children who were known to be developJournal of Pediatric Surgery, Vo130, No 3 (March), 1995: pp 495-499
Clinical Assessment The details of each patient were reviewed and the child/ adolescent fully evaluated clinically. The Kelly scoring scale4 was used to provide a level of faecal continence score, and the level of the lesion was classified according to the Wingspread classification system. 5 Table 1 illustrates the relationship between the level of the lesion and total Kelly scores.
Psychological Assessments In addition to individual interviews with each child and his or her parents, the participants completed standardized questionnaires.
Parents This interview included detailed questions about the child's and family's methods of managing and coping with continence problems at home, at school, and socially; about the child's social and personal relationships; and about psychological and emotional symptoms in the child.
From the Department of Paediatric Surgery, Institute of Child Health and Great Ormond Street Hospital for Children NHS Trust, London, England. Presented at the 41st Annual International Congress of the British Association of Paediatric Surgeons, Rotterdam, The Netherlands, June 29-July 1, 1994. This research was funded by a grant from the Leverhulme Trust. Address reprint requests to Dr L. Ludman, Department of Paediatric Surgery, Institute of Child Health, and Great Ormond Street Hospital for Children NHS Trust, 30 Guilford St, London WCIN 1EH, England. Copyright © 1995 by W.B. Saunders Company 0022-3468/95/3003-0030503.00/0 495
496
LUDMAN AND SPITZ
Table 1. Relationship Between Level of Lesion and TKS
Psychiatric Interview
Level of Lesion: Number (%) TKS
Low
Intermediate
High
5 or 6 _<4
45 (30) 17 (12)
5 (3) 12 (8)
6 (4) 63 (43)
Total
62 (42)
17 (11)
69 (47)
NOTE. Total sample with Kelly score 148 (no TKS for 8 cases; 4 cases had a permanent colostomy).
Standardized questionnaires. The parents completed the newly revised Child Behaviour Checklist (CBCL), 1° which yields a Total Behaviour Problem score (TPS) and two subscales, referred to as Internalizing (social withdrawal, depression, anxiety) and Externalizing Symptom (antisocial, aggressive, delinquency) scales. A T score of >_60 was used as the cut-off point for defining levels of clinical significance. Self-report questionnaires. Both parents completed the 30-item general health questionnaire 11 for assessing psychiatric morbidity, and the mothers completed a marital rating measure. 12
Teachers Teachers completed the Teacher's Report Form (TRF), t3 which is modelled on the CBCL with additional items appropriate for teachers.
RESULTS
Analyses of the data were conducted to examine the psychological adjustment of (1) The study group overall; (2) Groups based on the total Kelly score (TKS) (continent: score of 5 or 6, n = 56, incontinent: score < 4, n = 92). (A total Kelly score was not available for eight children, and four had permanent colostomies); (3) The age at which continence had been achieved: Group 1 (control group) continent before the age of 4 (n = 40), Group 2 (late continent) continent between 5 and 17 years (n = 18), and Group 3 (incontinent, n = 102); (4) Children/ adolescents with a total Kelly score of 6 (n = 38) and children/adolescents having frequent accidents
(n
=
33).
Forty-six of the 157 children/adolescents interviewed (29%) were judged as having some psychiatric disorder based on the diagnostic interview, ranging in severity from mild in 16 (10%) to moderate in 30 (19%) (a disorder severe enough to influence their daily lives). This prevalence of 19% is significantly higher when compared with a rate of 10% in the general child population ( < .001), 14 but is similar to that found in children with chronic disorders. The proportion deemed to have a disorder tended to be higher among the adolescents (35%) than among the children (24%). There were no significant differences between the continence groups based either on their total Kelly score or age of achieving continence. However, those with persistent incontinence (frequent accidents) were more likely to receive a psychiatric diagnosis when compared with those with good control (total Kelly score of 6) (z = 1.98, P < .05).
CBCL Questionnaire Data The mean Total Problem T scores (Table 2) and the internalizing mean T scores of the young boys, both continent [2 59 (SD 13)] and incontinent [2 57 (SD 9)], were significantly higher than those of the normative population. The mean internalizing and externalizing T scores for the rest of the sample were close to the population norms. There was no difference between the group with a TKS of 6 [2 27 (SD 18)] and those with persistent soiling [2 30.15 (SD 17)]. There was some indication that those who achieved continence late were less well adjusted. The frequency distribution of the TPS, however, indicated that 39 patients (27%) had scores higher than the eighty-second percentile, the recommended cut-off point for indicating psychopathology. This
Table 2. Mean CBCL and TRF Total Problem T Scores for Continent (TKS 5 or 6) and Incontinent (TKS _<4) Groups CBCL Mean T Scores Continent
6 to 11 years Boys Girls 12 to 18 years Boys Girls Total
TRF Mean T Scores Incontinent
Continent
Incontinent
No.
Mean (SD)
No.
Mean (SD)
No.
Mean (SD)
16 17
56.75 (11.92)* 50.71 (6.29)
28 15
55.12 (10.52)* 52.67 (8.96)
17 17
48.24 (11.22) 43.53 (7.78)1"
28 11
49.50 (9.31) 52.27 (7.80)1
9 12
54.11 (10.85) 53.00 (12.65)
29 19
49.62 (12,36) 51.84 (11.30)
8 9
56.63 (8.35) 50.89 (13.35)
28 17
49.62 (12.36) 50.59 (9.51)
54
91
NOTE, Norm for mean T score = 50, SD = 10. No Kelly scores for 12 cases. No CBCL for 3 cases. No TRF for 14 cases. *Mean T score significantly higher than the norm. 1"Difference between group significant (P < .01).
51
No.
Mean (SD)
84
ANORECTAL ANOMALIES: PSYCHOSOCIAL ADJUSTMENT
497
prevalence is significantly higher (P < .01) than that of the normative population, or when compared with the ninetieth percentile norm (17% versus 10%) used in previous research of children with chronic medical disorders.
TRF Questionnaire Data The TRF mean T scores (52; 95% CI 48 to 57) of the incontinent young girls were significantly greater than those of the girls with control (43.5; 95%, CI 40 to 47, t = 2.90 [df = 26] P = .007) (Table 2). The difference between the young girls' internalizing mean T scores was also significant (P = .003). With the exception of the adolescent boys with control, the remaining groups were close to the population norms. When the TRF mean scores of those with a TKS = 6 were compared with those with persistent incontinence, the differences between the groups approached significance (F = 3.29 [df = 1,61] P = .07). There was also a tendency for the adolescents who had achieved continence late to be rated as being maladjusted. In contrast to the CBCL findings, the frequency distribution of the TPS T scores indicated that only 17% of the sampie were in the clinical range for psychopathology, that is, similar to the normative population.
Depression Self-Rating Scale The self-rating depression scale showed that overall 24% (35 of 148) of the patients rated themselves as having depressive symptoms, with children (30%) being more frequently affected than adolescents (17%). The difference between the age groups approached significance (×2 = 3.44, df = 1, P = .06). There was a tendency for the incontinent patients
in each age group to have elevated scores compared with those who were continent. Depressive symptoms, however, were no more frequent among those with persistent incontinence than among those with normal bowel control. Once again those who achieved continence late appeared to have more problems than the other groups. For example, the adolescent girls who achieved continence late had significantly higher scores on a number of the scale items including the total depression score when compared with the incontinent adolescent girls (z = -2.08, P < .04).
Self-Esteem There were no significant differences between the continent and the incontinent groups on the global self-worth measure. When the group means were examined in relation to age and sex, the incontinent adolescent boys had lower levels of self-esteem than the continent adolescent boys (P < .06). Similarly the adolescent boys with persistent soiling had significantly lower levels of global self-worth than those with good bowel control (TKS = 6) (P < .04). Multiple regression analyses, with CBCL TPS as the dependent variable indicated that family factors, parental mental health, the marital relationship, and educational levels were significantly and independently associated with outcome, indicating that good scores on the family variables best predict a lower prevalence of behaviour problems. Similarly, with the TRF Total Problem scores as the dependent variable, better parental educational levels predicted a lower prevalence of behaviour problems, with the girls predicted to have significantly lower scores than the boys (Table 3). The TKS and "frequent accidents" were not predictive of either outcome measure.
Table 3. Regression of CBCL and TRF Total Problem Scores on Sex, Age, TKS, and Two Family Variables CBCL Dependent Variable (Predictor Variables)
Standardized Regression Coefficient
Sex Age Kelly score Family score¶ Family education#
- . 14011 -.124 .118 -.322t -.176§
TRF* Unstandardized Regression Coefficient (Standard Error B) - 5.16 -0.66 1.17 -7.43 -3.77
(2.92) (0.42) (0.82) (1.8!) (1.75)
*To overcome skewed distribution, a square root transformation was carried out. t P < .001. 1:P < .01. §P < .05.
lip < .10. ¶Composite score of parental mental health and marital relationship (higher scores better). #Composite score of parental educational levels (higher scores better).
Standardized Regression Coefficient -.249t .110 -.091 -.102 -,202§
Unstandardized Regression Coefficient (Standard Error B) -1.19 .08 -.118 -.308 -.58
(.39) (.06) (,11) (.25) (.24)
498
LUDMAN AND SPITZ
DISCUSSION
Contrary to our expectations, the findings from this study have shown that the incontinent children/ adolescents were not less well adjusted than those with good bowel control. In fact, when factors such as the child's gender and parental factors such as mental health, marital relationship, and education were taken into account, no association was found between the level of continence as measured by the TKS and measures of psychological adjustment. Nevertheless, among the sample overall, the prevalence of clinically significant emotional problems was higher than expected in relation to normative populations, with a high proportion of the children showing internalizing behaviour problems as well as reporting depressive symptoms. Some differences between the continence groups were apparent. For example, the psychiatric interview with the child showed that those having frequent soiling accidents were more likely to be recognised as emotionally disturbed than those who had control (TKS of 6). Similarly, based on the teachers' assessment of behavioural maladjustment, the difference between these groups approached significance. However, neither the parental assessment nor the children's self-report depression scores indicated any significant differences between the "frequent accident" and the "good control" groups. When the sample was examined in relation to age and sex, other differences between the groups were noted. The teachers rated the incontinent young girls as showing significantly more behaviour and internalizing problems compared with those with good controL This is supported by the findings from the parents' report, which indicated a somewhat higher prevalence of behaviour and internalising problems among the young incontinent girls compared with the continent young girls, and the incontinent young girls tended to have higher self-report depression scores than the continent young girls. In addition, the parents' ratings indicated that behavioural maladjustment among both the continent and incontinent young boys was significantly greater than among the normative groups. Parental factors (mental health, the marital relationship, and educational levels) were strongly associated with outcome as measured by the CBCL, but these variables were not important explanatory variables for outcome as measured by the psychiatric interview, the teachers' ratings, or the self-report child depression scale. It is possible that parents with higher levels of psychological distress and/or lower levels of marital satisfaction are more likely to report behaviour
problems, or these children may indeed be more behaviourally maladjusted, which in itself affects marital satisfaction and parental health. None of these parental factors were related to levels of continence. Parental education levels were significantly associated with behaviour problem scores. With this exception, the findings indicated no major effect of family background factors such as social class, birth position, or number of siblings. Contrary to our predictions the incontinent adolescents were not more emotionally disturbed than those with good bowel control. Although a substantial minority received a psychiatric diagnosis (40%, when cases rated as mild were included), more than half appeared to be able to cope with the social problems associated with their disability, as well as those associated with the physical and emotional changes of puberty and emerging sexuality. The findings that children/adolescents who achieved continence late tended to be less well adjusted than the controls or incontinent group, are similar to those in studies that have shown that the relationship between the severity of chronic illness and psychological adjustment is not necessarily linear, 15 and may reflect the fact that a child with a minimal and invisible disability is expected to function as normal so as not to appear different from his/her healthy peers while the reality of his/her condition makes normal behaviour difficult in many situations, On the other hand, the more severely affected are forced to accept the reality of their situation, and moreover, others make allowances for them, and consequently they are not expected to compete with their normal healthy peers. 16 Another unexpected finding was that a higher proportion of the study sample had clinically significant scores when compared with a normative population. These findings, which are comparable with other studies of children with chronic medical disorders, suggest that parents of children who are born with anorectal anomalies overcompensate for their child's early life-threatening problems. As we found in an earlier longitudinal study of children who underwent major neonatal surgery, 17 mothers tended to be ineffective and inconsistent in their responses to their children's behaviour, and this is known to be detrimental to future emotional and behavioural adjustment. In conclusion, this study, which is the first to examine in detail the relationship between faecal incontinence and emotional adjustment in a large
ANORECTAL ANOMALIES: PSYCHOSOCIALADJUSTMENT
499
group of children with anorectal anomalies, offers some encouragement to the children and their families, but it also indicates that these children, regardless of the level of continence, are at risk for difficulties with psychosocial adjustment. Outcome and compliance with treatment may be enhanced if, from an early age, psychological evaluation of the child and
experienced advice and guidance for the families become an integral part of their continuing care. ACKNOWLEDGMENT The authors thank Mr Kiely, Mr Drake, Mr Brereton, and their senior registrars for their contribution, and all the children and their parents who took part in the study.
REFERENCES 1. Ditesheim JA, Templeton Jr JM: Short-term v long-term quality of life in children following repair of high imperforate anus. J Pediatr Surg 22:581-587, 1987 2. Ginn-Pease ME, King DR, Tarnowski KJ, et al: Psychosocial adjustment and physical growth in children with imperforate anus or abdominal wall defects. J Pediatr Surg 26:1129-1135, 1991 3. Diseth T, Emblem R, Solbraa I, et al: A psychosocial follow-up of ten adolescents with low anorectal malformation. Acta Paediatr 83:216-221, 1994 4. Kelly JH: The clinical and radiological assessment of anal continence in childhood. Aust N Z J Surg 42:62-63, 1972 5. Stephens FD, Durham Smith E: Classification, identification, and assessment of surgical treatment of anorectal anomalies. Pediatr Surg Int 1:200-205, 1986 6. Hodges K, Kline J, Fitch P, et al: Child assessment schedule: A diagnostic interview for research and clinical use. Catalog of Selected Documents in Psychology, vol 11 (issue 56), 1981 7. Rutter M, Graham P: The reliability and validity of the psychiatric assessment of the child. 1. Interview with the child. Br J Psychiatry 114:563-579, 1968 8. Birleson P: The validity of depressive disorder in childhood and the development of a self-rating scale: a research note. J Child Psychol Psychiatr 22:73-88, 1981 9. Harter S: Manual for the Self-Perception Profile for Children. University of Denver, Colorado, 1985
10. Achenbach TM: Manual for the Child Behaviour Checklist/ 4-18 and 1991 Profile. Burlington, VT, University of Vermont, Department of Psychiatry, 1991 11. Goldberg DF: Manual of the general health questionnaire. Windsor, England, National Foundation for Educational Research Publishing Company, 1978 12. Spanier GB: Measuring dyadic adjustment: New scales for assessing the quality of marriage and similar dyads. J Marriage Faro 38:15-28, 1976 13. Achenbach TM: Manual for the Teacher's Report Form and 1991 Profile. Burlington, VT, University of Vermont, Department of Psychiatry, 1991 14. Pearce J: Child health surveillance for psychiatric disorder: Practical guidelines. Arch Dis Child 69:394-398, 1993 15. MacLean WE, Perrin JM, Gortmaker S, et al: Psychological adjustment of children with asthma: Effects of illness severity and recent stressful life events. J Pediatr Psychol 17:159-171, 1992 16. McAnarney ER, Pless B, Satterwhite B, et al: Psychological problems of children with chronic juvenile arthritis. Pediatrics 53:523-527, 1974 17. Ludman L, Lansdown R, Spitz L: Effects of early hospitalization and surgery on the emotional development of 3 year olds: An exploratory study. Eur Child Adolescent Psychiatr 1:186-195, 1992