title: Development of a tool to describe overall health, social independence and activity limitation of adolescents and young adults with disability.
abstract: There is a need for research that focuses on the correlation between self-perceived quality of life (QoL) and the health outcomes of adolescents with disability transitioning to adulthood. To better understand the transition experience of adolescents and young adults with disability, we developed a questionnaire to assess the impact of disability on QoL. We recruited 174 participants who were 15-24 years old and diagnosed with Fragile X syndrome (FXS), spina bifida (SB) or muscular dystrophy (MD) and conducted an exploratory factor analysis to identify factors that characterize QoL. Five factors emerged: emotional health, physical health, independence, activity limitation, and community participation. To validate the tool, we linked medical claims and other administrative data records and examined the association of the factor scores with health care utilization and found the questionnaire can be utilized among diverse groups of young people with disability.
authors.affiliation: University of South Carolina, Arnold School of Public Health, Department of Epidemiology and Biostatistics, 915 Greene Street, Columbia, SC 29208, United States. Electronic address: derochcb@email.sc.edu.
authors.forename: Chelsea B
authors.initials: CB
authors.lastname: Deroche
authors.affiliation: University of South Carolina, Arnold School of Public Health, Department of Epidemiology and Biostatistics, 915 Greene Street, Columbia, SC 29208, United States. Electronic address: hollanmm@mailbox.sc.edu.
authors.forename: Margaret M
authors.initials: MM
authors.lastname: Holland
authors.affiliation: University of South Carolina, Arnold School of Public Health, Department of Epidemiology and Biostatistics, 915 Greene Street, Columbia, SC 29208, United States. Electronic address: SCMCDERMO@mailbox.sc.edu.
authors.forename: Suzanne
authors.initials: S
authors.lastname: McDermott
authors.affiliation: Revenue and Fiscal Affairs Office, Health and Demographics, Blanding Street, Columbia, SC 29201, United States. Electronic address: Julie.Royer@rfa.sc.gov.
authors.forename: Julie A
authors.initials: JA
authors.lastname: Royer
authors.affiliation: University of South Carolina, Arnold School of Public Health, Department of Epidemiology and Biostatistics, 915 Greene Street, Columbia, SC 29208, United States. Electronic address: JHARDIN@mailbox.sc.edu.
authors.forename: James W
authors.initials: JW
authors.lastname: Hardin
authors.affiliation: University of South Carolina, School of Medicine, Department of Family and Preventive Medicine, 3209 Colonial Drive, Columbia, SC 29203, United States. Electronic address: Joshua.mann@uscmed.sc.edu.
authors.forename: Joshua R
authors.initials: JR
authors.lastname: Mann
authors.affiliation: University of South Carolina, Arnold School of Public Health, Department of Epidemiology and Biostatistics, 915 Greene Street, Columbia, SC 29208, United States. Electronic address: salzberd@mailbox.sc.edu.
authors.forename: Deborah
authors.initials: D
authors.lastname: Salzberg
authors.affiliation: University of South Carolina, Moore School of Business, Department of Economics, 1705 College Street, Columbia, SC 29208, United States. Electronic address: odozturk@moore.sc.edu.
authors.forename: Orgul
authors.initials: O
authors.lastname: Ozturk
authors.affiliation: Centers for Disease Control and Prevention, National Center on Birth Defects and Developmental Disabilities, 1600 Clifton Road, Atlanta, GA 30333, United States. Electronic address: eop9@cdc.gov.
authors.forename: Lijing
authors.initials: L
authors.lastname: Ouyang
mesh_terms: D000203:Activities of Daily Living; D000293:Adolescent; D006233:Persons with Disabilities / Q000523:psychology*; D005163:Factor Analysis, Statistical; D005260:Female; D005600:Fragile X Syndrome / Q000503:physiopathology / Q000523:psychology*; D006304:Health Status; D006801:Humans; D008297:Male; D009136:Muscular Dystrophies / Q000503:physiopathology / Q000523:psychology*; D011788:Quality of Life / Q000523:psychology*; D015203:Reproducibility of Results; D012919:Social Behavior; D016135:Spinal Dysraphism / Q000503:physiopathology / Q000523:psychology*; D011795:Surveys and Questionnaires; D055815:Young Adult
keywords: Factor analysis; Fragile X syndrome; Muscular dystrophy; Quality of life; Rare conditions; Spina bifida; Survey instrument