title: Assessing the quality of life of health-referred children and adolescents with short stature: development and psychometric testing of the QoLISSY instrument.
abstract: BACKGROUND
  When evaluating the outcomes of treatment in paediatric endocrinology, the health-related quality of life (HrQoL) of the child is to be taken into consideration. Since few self-reported HrQoL instruments exist for children with diagnosed short stature (dSS), the objective of this study was to develop and psychometrically test a targeted HrQoL instrument for use in multinational clinical research.
  
  
  METHODS
  The target population were short stature (height<-2 SDS) children and adolescents (age 8-12 and 13-18 years) with a diagnosis of growth hormone deficiency (GHD) or idiopathic short stature (ISS), differing in growth hormone treatment status. Focus group discussions for concept and item generation, piloting of the questionnaire with cognitive debriefing, and instrument field testing with a retest were conducted simultaneously in five countries. After qualitative and preliminary quantitative analyses, psychometric testing of field test data in terms of reliability and validity including confirmatory factor analyses (CFA) was performed.
  
  
  RESULTS
  Following item generation from focus group discussions, 124 items were included in a pilot test with a cognitive debriefing exercise providing preliminary feedback on item and domain operating characteristics. A field test with 268 participants showed high internal consistency reliabilities (alpha 0.82-0.95), good correlations with generic measures (up to r=.58), significant known group differences (e.g. in height: F=32, df 244, p<0.001) and an acceptable CFA model fit suggesting construct validity of the three-domain core structure with 22 items, supplemented by three mediator domains with 28 items.
  
  
  CONCLUSIONS
  The QoLISSY questionnaire is a promising step forward in assessing the impact of dSS on HrQoL. It is based on items generated from the subjective experience of short stature children referred for endocrine investigation, is validated for use in five languages and it is easy to administer in clinical and research settings.
authors: University Hamburg-Eppendorf, Department of Medical Psychology, Martinistr, 52, W26, Hamburg 20246, Germany. bullinge@uke.de
authors: Monika
authors: M
authors: Bullinger
authors: Julia
authors: J
authors: Quitmann
authors: Mick
authors: M
authors: Power
authors: Michael
authors: M
authors: Herdman
authors: Emmanuelle
authors: E
authors: Mimoun
authors: Kendra
authors: K
authors: DeBusk
authors: Eva
authors: E
authors: Feigerlova
authors: Carolina
authors: C
authors: Lunde
authors: Maria
authors: M
authors: Dellenmark-Blom
authors: Dolores
authors: D
authors: Sanz
authors: Anja
authors: A
authors: Rohenkohl
authors: Andreas
authors: A
authors: Pleil
authors: Hartmut
authors: H
authors: Wollmann
authors: John E
authors: JE
authors: Chaplin
mesh_terms: D000293:Adolescent; D002648:Child; D004393:Dwarfism, Pituitary / Q000523:psychology*; D005260:Female; D017144:Focus Groups; D006801:Humans; D008297:Male; D010865:Pilot Projects; D011788:Quality of Life; D015203:Reproducibility of Results; D011795:Surveys and Questionnaires / Q000592:standards*
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