title: Development of Health-Related Quality of Life Instruments for Young Children With Disorders of Sex Development (DSD) and Their Parents.
abstract: Research in disorders of sex development (DSD) is hindered by a lack of standardized measures sensitive to the experiences of affected children and families. We developed and evaluated parent proxy (children 2-6 years) and parent self-report (children ≤6 years) health-related quality of life (HRQoL) instruments for DSD.
  
  
  
  Items were derived from focus groups and open-ended interviews. Clarity and comprehensiveness were assessed with cognitive interviews. Psychometric properties were examined in a field survey of 94 families.
  
  
  
  Measures demonstrated adequate to good psychometrics, including internal consistency, test-retest reliability, convergent validity, and ability to detect known-group differences. Parents reported greatest stress on Early Experiences , Surgery , and Future Concerns scales.
  
  
  
  These instruments identify patients' and families' needs, monitor health and quality of life status, and can evaluate clinical interventions. Findings highlight the need for improved psychosocial support during the diagnostic period, better parent-provider communication, and shared decision-making. HRQoL measures are needed for older youth.
authors.affiliation: Department of Psychology, University of Miami, Ponce De Leon Blvd., Coral Gables, FL, USA.
authors.forename: Adrianne N
authors.initials: AN
authors.lastname: Alpern
authors.affiliation: Department of Pediatrics; Division of Pediatric Psychology and Child Health Evaluation & Research (CHEAR) Unit, University of Michigan, Ann Arbor, Michigan, USA.
authors.forename: Melissa
authors.initials: M
authors.lastname: Gardner
authors.affiliation: Division of Urology, Albany Medical College, Albany, NY, USA.
authors.forename: Barry
authors.initials: B
authors.lastname: Kogan
authors.affiliation: Department of Pediatrics; Division of Pediatric Psychology and Child Health Evaluation & Research (CHEAR) Unit, University of Michigan, Ann Arbor, Michigan, USA.
authors.forename: David E
authors.initials: DE
authors.lastname: Sandberg
authors.affiliation: Department of Psychology, University of Miami, Ponce De Leon Blvd., Coral Gables, FL, USA.
authors.forename: Alexandra L
authors.initials: AL
authors.lastname: Quittner
mesh_terms: D000328:Adult; D002648:Child; D002675:Child, Preschool; D012734:Disorders of Sex Development / Q000523:psychology*; D005260:Female; D017144:Focus Groups; D006305:Health Status Indicators; D006801:Humans; D007407:Interviews as Topic; D008297:Male; D010290:Parents / Q000523:psychology*; D000071066:Patient Reported Outcome Measures; D011594:Psychometrics; D011788:Quality of Life / Q000523:psychology*; D015203:Reproducibility of Results; D057566:Self Report
keywords: disorders of sex development; health-related quality of life; patient-reported outcome; psychometric evaluation