title: Parental perception of health-related quality of life in children and adolescents with short stature: literature review and introduction of the parent-reported QoLISSY instrument.
abstract: BACKGROUND
  Health-related quality of life (HrQoL) of the child diagnosed with short stature is an important outcome to be assessed both from the patient as well as from the parental perspective. The objective of this study was to review the literature on parent-reported HrQoL and to subsequently develop and psychometrically test the parent-reported version of the Quality of Life in Short Stature Youth (QoLISSY) instrument for use in clinical and epidemiologic research.
  
  
  METHODS
  A review of the literature on parental assessment of child HrQoL via PUBMED was followed by a psychometric analysis of data collected within the European QoLISSY study, in which 686 eligible parents of short statured children/adolescents (aged 4-18 years) meeting inclusion criteria participated. Patient inclusion criteria were a height below -2 SD, a diagnosis of growth hormone deficiency (GHD) or idiopathic short stature (ISS), and treatment status in terms of receiving or not receiving recombinant human growth hormone therapy. Focus groups eliciting parental HrQoL statements, pilot testing with cognitive debriefing, and a field test in 317 parents with a retest in 148 parents were conducted simultaneously in France, Germany, Spain, Sweden and the UK. The psychometric performance of the parent-reported instrument, developed in parallel to the child/ adolescent self-report version, was assessed using standard tests of reliability and validity.
  
  
  RESULTS
  Literature search failed to identify a cross-culturally developed height specific instrument available for both patient self-report and parental observer report. Analysis of the QoLISSY focus group phase conducted separately in children, adolescents and parents yielded 169 items generated from parent focus groups. A cognitive debriefing exercise followed by a pilot test of preliminary psychometric characteristics resulted in deleting poorly performing items. Field testing of the parent-reported version suggested a three-domain core HrQoL structure with 22 items, additional 44 items assessing three mediator domains and two parent specific domains. The parent report version demonstrated good criterion and construct validity as well as internal consistency and test retest reliability.
  
  
  CONCLUSIONS
  The QoLISSY parent report questionnaire closes a gap in the simultaneous assessment of parent and child perception of HrQoL in an international context. It is based on items generated from the experience of short statured children, adolescents and their parents and is validated for use in five European languages. It is feasible, relevant for this population, psychometrically sound and is easy to administer in research and clinical settings.
authors.affiliation: University Medical Center Hamburg-Eppendorf, Department of Medical Psychology, Martinistr. 52, 20246 Hamburg, Germany. j.quitmann@uke.de
authors.forename: Julia
authors.initials: J
authors.lastname: Quitmann
authors.affiliation: University Medical Center Hamburg-Eppendorf, Department of Medical Psychology, Martinistr. 52, 20246 Hamburg, Germany.
authors.forename: Anja
authors.initials: A
authors.lastname: Rohenkohl
authors.affiliation: University Medical Center Hamburg-Eppendorf, Department of Medical Psychology, Martinistr. 52, 20246 Hamburg, Germany.
authors.forename: Monika
authors.initials: M
authors.lastname: Bullinger
authors.affiliation: Sahlgrenska Academy at University of Gothenburg, Department of Paediatrics, Växthuset, Queen Silvia's Childrens Hospital, 41685 Gothenburg, Sweden.
authors.forename: John E
authors.initials: JE
authors.lastname: Chaplin
authors.affiliation: IMIM University, Insight Consulting and Research, Cami Ral 266, Mataró, Barcelona, Spain.
authors.forename: Michael
authors.initials: M
authors.lastname: Herdman
authors.affiliation: IMIM University, Insight Consulting and Research, Cami Ral 266, Mataró, Barcelona, Spain.
authors.forename: Dolores
authors.initials: D
authors.lastname: Sanz
authors.affiliation: University of Toulouse, Department of Pediatric Endocrinology, CHU, 330 avenue de Grande-Bretagne FR-31059 Toulouse, France.
authors.forename: Emmanuelle
authors.initials: E
authors.lastname: Mimoun
authors.affiliation: University of Toulouse, Department of Pediatric Endocrinology, CHU, 330 avenue de Grande-Bretagne FR-31059 Toulouse, France.
authors.forename: Eva
authors.initials: E
authors.lastname: Feigerlova
authors.affiliation: University of Edinburgh, Department of Clinical Psychology, Teviot Place, EH8 9AG Edinburgh, Scotland.
authors.forename: Kendra
authors.initials: K
authors.lastname: DeBusk
authors.affiliation: University of Edinburgh, Department of Clinical Psychology, Teviot Place, EH8 9AG Edinburgh, Scotland.
authors.forename: Michael
authors.initials: M
authors.lastname: Power
authors.affiliation: Pfizer Ltd., Specialty Care MDG, Endocrinology, Dorking Road, Walton-on-the-Hill, Tadworth Surrey, KT20 7NS Walton Oaks, UK.
authors.forename: Hartmut
authors.initials: H
authors.lastname: Wollmann
authors.affiliation: Pfizer Ltd., Specialty Care MDG, Endocrinology, Dorking Road, Walton-on-the-Hill, Tadworth Surrey, KT20 7NS Walton Oaks, UK.
authors.forename: Andreas
authors.initials: A
authors.lastname: Pleil
mesh_terms: D000293:Adolescent; D001827:Body Height / Q000502:physiology; D002648:Child; D002675:Child, Preschool; D004392:Dwarfism / Q000523:psychology; D006130:Growth Disorders / Q000523:psychology*; D006304:Health Status; D006801:Humans; D010287:Parent-Child Relations; D010290:Parents / Q000523:psychology*; D010465:Perception; D011594:Psychometrics / Q000379:methods*; D011788:Quality of Life; D011795:Surveys and Questionnaires