title: Children With Anorectal Malformations, Hirschsprung Disease, and Their Siblings: Proxy Reports and Self-Reports.
abstract: OBJECTIVE
  The aim of the present study was to compare parent proxy reports with that of self-reports of children with anorectal malformations (ARMs) or Hirschsprung disease (HD) and healthy siblings and thereafter was examine whether these comparisons differed between patients and their siblings.
  
  
  METHODS
  Parents (n = 98) of either children with ARM (n = 44) or HD (n = 54) and a healthy sibling (n = 98) recruited from the 6 Dutch pediatric surgical centers and from the ARM and HD patient societies were included in this cross-sectional multilevel study. Agreement between child self-reports and parent proxy reports was compared through mean differences and through (intraclass) correlations. We conducted multilevel analyses to take dependencies between assessments within families into account.
  
  
  RESULTS
  All of the children (children with ARM or HD and their siblings) reported more pain and symptoms than their parents reported. We also found that only children with ARM or HD reported less positive emotions than their parents. Furthermore, higher correlations were found between parent proxy reports and patient-self reports than between parent proxy reports and sibling self-reports on cognitive functioning and social interaction.
  
  
  CONCLUSIONS
  Parents tend to overestimate the physical functioning of both their ill and healthy children, and overestimate the emotional functioning of only their children with ARM or HD. Furthermore, children with ARM or HD and parents agree more on health-related quality of life domains than healthy children and parents.
authors.affiliation: †Department of Clinical and Medical Psychology, Center of Research on Psychology in Somatic Diseases, Tilburg University, Tilburg ‡Department of Education, Faculty of Social and Behavioural Sciences §Division of Medical Psychology, University of Amsterdam ||Department of Paediatric Surgery, University Hospital, VU University Medical Centre/Emma Children's Hospital AMC, Amsterdam ¶Department of Paediatric Surgery, University Hospital, Radboud Hospital, Nijmegen #Department of Paediatric Surgery, University Hospital, Maastricht ††Department of Paediatric Surgery, University Hospital, Utrecht, The Netherlands ‡‡Division of Paediatric Surgery, Queen Elisabeth Central Hospital, College of Medicine, University of Malawi, Blantyre, Malawi §§Department of Paediatric Surgery, University Hospital, Groningen, The Netherlands.
authors.forename: Esther E
authors.initials: EE
authors.lastname: Hartman
authors.forename: Frans J
authors.initials: FJ
authors.lastname: Oort
authors.forename: Daniel C
authors.initials: DC
authors.lastname: Aronson
authors.forename: Alida F W
authors.initials: AF
authors.lastname: van der Steeg
authors.forename: Hugo A
authors.initials: HA
authors.lastname: Heij
authors.forename: Ernest
authors.initials: E
authors.lastname: van Heurn
authors.forename: Gerard C
authors.initials: GC
authors.lastname: Madern
authors.forename: David C
authors.initials: DC
authors.lastname: van der Zee
authors.forename: Ivo
authors.initials: I
authors.lastname: de Blaauw
authors.forename: A
authors.initials: A
authors.lastname: van Dijk
authors.forename: Mirjam A G
authors.initials: MA
authors.lastname: Sprangers
mesh_terms: D000293:Adolescent; D001003:Anal Canal / Q000002:abnormalities*; D000071056:Anorectal Malformations; D001006:Anus, Imperforate / Q000150:complications / Q000523:psychology*; D002648:Child; D003071:Cognition; D003430:Cross-Sectional Studies; D004644:Emotions; D005260:Female; D006627:Hirschsprung Disease / Q000150:complications / Q000523:psychology; D006801:Humans; D007398:Interpersonal Relations; D008297:Male; D009426:Netherlands; D010146:Pain; D010287:Parent-Child Relations; D010290:Parents; D019727:Proxy; D011594:Psychometrics; D011788:Quality of Life; D012007:Rectum / Q000002:abnormalities*; D012016:Reference Values; D057566:Self Report; D035781:Siblings; D011795:Surveys and Questionnaires