title: Explaining change over time in quality of life of adult patients with anorectal malformations or Hirschsprung's disease.
abstract: OBJECTIVE
  The aim of this study was to examine changes in the quality of life of adult patients with anorectal malformations or Hirschsprung's disease over a three-year interval and to identify demographic, clinical, and psychosocial variables that explain possible quality-of-life changes. Understanding the factors that affect changes in quality of life over time is particularly important to provide adequate care.
  
  
  METHODS
  Questionnaires were administered to 261 patients (77 percent), with a three-year interval. Background characteristics, including demographic and clinical variables, and psychosocial variables (i.e., self-esteem, mastery, social support, disease cognition) were measured on one occasion. Generic and disease-specific quality of life were measured twice.
  
  
  RESULTS
  On average patients indicated no change in quality-of-life level after three years. However, variance in the change scores revealed individual variation, indicating the presence of patients who improved and patients who deteriorated. Patients who were female, older, have other congenital diseases, or a stoma reported poorer quality of life over time. The psychosocial variable "disease cognition" most strongly affected the change in quality of life of patients with anorectal malformations or Hirschsprung's disease.
  
  
  CONCLUSIONS
  Our results could alert clinicians to patients who are at risk for quality-of-life deterioration and might therefore be in need for extra care. Our findings illustrate the importance of psychosocial functioning for enhancing the quality of life over time of these patients.
authors.affiliation: Pediatric Surgical Center of Amsterdam, Emma Children's Hospital AMC/VU Medical Center, The Netherlands. e.e.hartman@amc.uva.nl
authors.forename: Esther E
authors.initials: EE
authors.lastname: Hartman
authors.forename: Frans J
authors.initials: FJ
authors.lastname: Oort
authors.forename: Mechteld R
authors.initials: MR
authors.lastname: Visser
authors.forename: Mirjam A
authors.initials: MA
authors.lastname: Sprangers
authors.forename: Marianne J
authors.initials: MJ
authors.lastname: Hanneman
authors.forename: Zacharias J
authors.initials: ZJ
authors.lastname: de Langen
authors.forename: L W Ernst
authors.initials: LW
authors.lastname: va Heurn
authors.forename: Paul N M A
authors.initials: PN
authors.lastname: Rieu
authors.forename: Gerard C
authors.initials: GC
authors.lastname: Madern
authors.forename: David C
authors.initials: DC
authors.lastname: van der Zee
authors.forename: Nic
authors.initials: N
authors.lastname: Looyard
authors.forename: Marina
authors.initials: M
authors.lastname: van Silfhout-Bezemer
authors.forename: Daniel C
authors.initials: DC
authors.lastname: Aronson
mesh_terms: D000293:Adolescent; D000328:Adult; D001003:Anal Canal / Q000002:abnormalities*; D005260:Female; D006627:Hirschsprung Disease / Q000523:psychology*; D006801:Humans; D008297:Male; D008875:Middle Aged; D009426:Netherlands; D011788:Quality of Life; D012007:Rectum / Q000002:abnormalities*; D012189:Retrospective Studies; D011795:Surveys and Questionnaires; D013997:Time Factors