Aim: The overall aim of this thesis was to explore psychosocial consequences on children with high and intermediate imperforate anus and their parents.

Material/Methods: Paper I was an explorative study involving interviews. The purpose was to illuminate the children s and parents experiences of imperforate anus (IA) and to create a baseline for further research. In Papers II-V, study-specific questionnaires were used to collect data from 25 children (8-13 years old) with high and intermediate imperforate anus and from their parents. The questionnaires comprised 45 items for the children and 59 items for the parents, covering psychosocial issues, items on child bowel function, and on experiences of hospital care. The parents also filled in the Child Behavior Checklist (CBCL), and the children s teachers filled in the Teacher s Report Form (TRF). Thirty children with juvenile chronic arthritis and 32 children with no chronic condition, along with their parents, served as Comparison Groups. In Paper VI, the psychometric properties of the child self-reported psychosocial items were evaluated with Rasch analysis.

Results: Four categories of effects of the malformation were disclosed in the interviews: physical, emotional, social, and family effects. The emotional effects permeated the interviews; the parents suffering was evident. In the questionnaires the children with imperforate anus reported being generally happy, they liked school, and had good relationships with peers. According to their parents, the children had fecal incontinence and constipation. In the CBCL, the children with imperforate anus were assessed to have more emotional/behavioral problems than the children with juvenile chronic arthritis. On the CBCL competence scales no differences were found between groups. In the TRF, the teachers reported few emotional/behavioral problems for the children with imperforate anus, though they assessed them to be less adapted in school than were the children with no chronic condition. Differences were found in answers on the psychological items, between the children and their mothers, on the pair level. The mothers of children with imperforate anus rated lower on their child s expression of will and on respecting their child s will, than did the other mothers. Fathers scored school items optimistically. Several parents of children with imperforate anus reported positive experiences in relation to their child s condition involving child development, parental development, and strengthening of family unity. The psychometric properties of the psychological and the social dimension in the child self-report questionnaire were reasonably good.

Conclusions: The children with imperforate anus might have some psychosocial difficulties, though informants do not agree. According to self-report questionnaires, the children appear to be well-adjusted, despite functional bowel problems. Parents of children with imperforate anus have to go through difficult experiences associated with their child s malformation, though some parents also report positive experiences. Psychosocial issues seem crucial to children with imperforate anus and to their parents, and qualified individualized assistance should be a central part of their continuing care to ameliorate or prevent difficulties. Collaboration with expertise from child and adolescent psychiatry may be required. Child autonomy needs more attention. Psychosocial interventions are requested as well as a follow-up study on these children and their parents. Further development of the measurement tools, the study-specific questionnaires, is required.

Imperforate anus including lack of a normal anorectum is an uncommon malformation. The parents of children born with high and intermediate imperforate anus have to take a big responsibility for the follow-up treatment. It has been revealed that children with imperforate anus have psychosocial problems, though knowledge of this is limited.

Aim: The overall aim of this thesis was to explore psychosocial consequences on children with high and intermediate imperforate anus and their parents.

Methods: In Paper I, the purpose was to illuminate the children s and parents experiences of imperforate anus and to create a baseline for further research. Interviews were done with three patients with high imperforate anus who had had severe problems with the malformation, and with their parents (5). Answers to the open-ended questions were analyzed with a qualitative method, editing analysis style . In Paper II and Paper III, study-specific questionnaires were used to collect data from 25 children (8-13 years old) with high and intermediate imperforate anus, and from their parents. The questionnaires comprised 59 items for the parents and 45 items for the children, covering psychosocial domains, one physical domain and one domain on experiences of hospital care. The parents filled in the Child Behavior Checklist (CBCL), and the children s teachers filled in the Teacher s Report Form (TRF). Two groups of children along with their parents served as comparison groups. They included 30 children with juvenile chronic arthritis and 32 healthy children.

Results: Paper I: The parents were outspoken in the interviews, and the children s answers were meagre. Four categories of effects of the malformation were revealed: physical, emotional, social, and family. The emotional effects permeated the interviews; the parents suffering was overwhelming. In Paper II, the children with imperforate anus displayed an overall positive attitude; they reported that they were in a good mood, that they liked school, and that they had good relationships with peers. Furthermore, the children had fecal incontinence and constipation, according to parents. In the CBCL, the children in the index group were assessed to have significantly more emotional/behavioral problems than the children in Comparison Group I. In the TRF, the teachers reported few symptoms for the index group children. Paper III showed disagreement on psychosocial variables from children with imperforate anus and their mothers (on a pair level). Differences were found in particular on psychological variables, i.e. the child s emotional life. The competence scales in the CBCL revealed no differences between the index group and the other two groups. In the TRF, the teachers assessed the children with imperforate anus to be less adapted and adjusted in school than the children in comparison groups.

Conclusions: Parents of children with high imperforate anus have to go through difficult experiences associated with the malformation. Children with imperforate anus in this study seem to be well adjusted psychosocially, despite fecal incontinence and/or constipation. The children might have some psychosocial difficulties, though informants do not agree. It is vital to assess the children s psychosocial function from more than a single informant. Psychosocial issues are crucial to children with imperforate anus and to their parents, and qualified advice and assistance should be a central part of their continuing care. Perhaps collaboration with expertise from child and adolescent psychiatry may be required.