title: Development and initial validation of a disease specific outcome measure for early onset scoliosis.
abstract: BACKGROUND
  Treatment of children with early onset scoliosis (EOS) seeks to improve natural history and health related quality of life (QOL). QOL measurement presents a significant challenge given the young age, comorbidities, and heterogeneity of this population.
  
  
  OBJECTIVE
  To develop a disease specific measure reflecting issues of importance to EOS patients and caretakers.
  
  
  METHODS
  Review of the literature and relevant measures informed development of semi-structured qualitative interviews. Interviews of caregivers of EOS patients were interpreted through the framework technique. A master-list of 75-items was created and responses were scaled on a 5-point Likert-scale. Psychometric analysis was performed to group items into domains, to maintain score distribution, to create a responsive instrument, and to identify question redundancy.
  
  
  RESULTS
  Content validation revealed 8-items with low content validity indices. Item distribution analysis demonstrated 19-items with skewed distribution. Item reliability demonstrated redundant items within domains (r> or =0.5). Construct validity demonstrated unique domains measuring intended issues of interest. The resulting Early Onset Scoliosis Questionnaire (EOSQ) includes 33-items in 13-domains: General Health, Pain, Physical Function, Pulmonary Function, Daily Living, Fatigue, Emotion, Surgical Concerns, Satisfaction, Transfer, Financial Burden, Parental Burden, and Treatment Outcomes.
  
  
  CONCLUSIONS
  The EOSQ reflects QOL and caregiver burden in the EOS population. The EOSQ will expand options for outcome assessment in this unique population.
authors.affiliation: Pediatric Orthopaedic Surgery, Department of Orthopaedic Surgery, Columbia University, College of Physicians and Surgeons, New York, NY, USA.
authors.forename: Michael G
authors.initials: MG
authors.lastname: Vitale
authors.forename: Jacqueline
authors.initials: J
authors.lastname: Corona
authors.forename: Hiroko
authors.initials: H
authors.lastname: Matsumoto
authors.forename: Javier
authors.initials: J
authors.lastname: Avendano
authors.forename: Dynell
authors.initials: D
authors.lastname: Pinder
authors.forename: Daniel J
authors.initials: DJ
authors.lastname: Miller
authors.forename: David P
authors.initials: DP
authors.lastname: Roye
mesh_terms: D017668:Age of Onset; D002675:Child, Preschool; D006801:Humans; D007407:Interviews as Topic; D012600:Scoliosis / Q000175:diagnosis*; D011795:Surveys and Questionnaires / Q000592:standards*