title: HDQLIFE: the development of two new computer adaptive tests for use in Huntington disease, Speech Difficulties, and Swallowing Difficulties.
abstract: Huntington disease (HD) is an autosomal dominant neurodegenerative disease which results in several progressive symptoms, including bulbar dysfunction (i.e., speech and swallowing difficulties). Although difficulties in speech and swallowing in HD have a negative impact on health-related quality of life, no patient-reported outcome measure exists to capture these difficulties that are specific to HD. Thus, we developed a new patient-reported outcome measure for use in the Huntington Disease Health-Related Quality of Life (HDQLIFE) Measurement System that focused on the impact that difficulties with speech and swallowing have on HRQOL in HD.
  
  
  
  Five hundred and seven individuals with prodromal and/or manifest HD completed 47 newly developed items examining speech and swallowing difficulties. Unidimensional item pools were identified using exploratory factor analysis and confirmatory factor analysis (EFA and CFA, respectively). Item response theory (IRT) was used to calibrate the final measures.
  
  
  
  EFA and CFA identified two separate unidimensional sets of items: Speech Difficulties (27 items) and Swallowing Difficulties (16 items). Items were calibrated separately for these two measures and resulted in item banks that can be administered as computer adaptive tests (CATs) and/or 6-item, static short forms. Reliability of both of these measures was supported through high correlations between the simulated CAT scores and the full item bank.
  
  
  
  CATs and 6-item calibrated short forms were developed for HDQLIFE Speech Difficulties and HDQLIFE Swallowing Difficulties. These measures both demonstrate excellent psychometric properties and may have clinical utility in other populations where speech and swallowing difficulties are prevalent.
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, North Campus Research Complex, 2800 Plymouth Road, Building NCRC B14, Room G216, Ann Arbor, MI, 48109-2800, USA. carlozzi@umich.edu.
authors.forename: N E
authors.initials: NE
authors.lastname: Carlozzi
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, North Campus Research Complex, 2800 Plymouth Road, Building NCRC B14, Room G216, Ann Arbor, MI, 48109-2800, USA.|Institute for Social Research, University of Michigan, Ann Arbor, MI, USA.
authors.forename: S G
authors.initials: SG
authors.lastname: Schilling
authors.affiliation: Center on Outcomes, Research and Education, Evanston Northwestern Healthcare, Northwestern University, Evanston, IL, USA.|Institute for Health Services Research and Policy Studies, Feinberg School of Medicine, Northwestern University, Chicago, IL, USA.
authors.forename: J-S
authors.initials: JS
authors.lastname: Lai
authors.affiliation: Departments of Neurology, Radiology, and Anatomy and Neurobiology, Washington University School of Medicine, St. Louis, MO, USA.|Program in Occupational Therapy and Program in Physical Therapy, Washington University School of Medicine, St. Louis, MO, USA.
authors.forename: J S
authors.initials: JS
authors.lastname: Perlmutter
authors.affiliation: Hennepin County Medical Center, Minneapolis, MN, USA.|Department of Medical and Molecular Genetics, Indiana University, Indianapolis, IN, USA.
authors.forename: M A
authors.initials: MA
authors.lastname: Nance
authors.affiliation: Department of Surgery, University of Michigan, Ann Arbor, MI, USA.
authors.forename: J F
authors.initials: JF
authors.lastname: Waljee
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, North Campus Research Complex, 2800 Plymouth Road, Building NCRC B14, Room G216, Ann Arbor, MI, 48109-2800, USA.
authors.forename: J A
authors.initials: JA
authors.lastname: Miner
authors.affiliation: Departments of Neurology, Radiology, and Anatomy and Neurobiology, Washington University School of Medicine, St. Louis, MO, USA.
authors.forename: S K
authors.initials: SK
authors.lastname: Barton
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, North Campus Research Complex, 2800 Plymouth Road, Building NCRC B14, Room G216, Ann Arbor, MI, 48109-2800, USA.
authors.forename: S M
authors.initials: SM
authors.lastname: Goodnight
authors.affiliation: Department of Neurology, University of Michigan, Ann Arbor, MI, USA.
authors.forename: P
authors.initials: P
authors.lastname: Dayalu
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D003201:Computers / Q000706:statistics & numerical data*; D003680:Deglutition Disorders / Q000628:therapy*; D005260:Female; D006801:Humans; D006816:Huntington Disease / Q000523:psychology*; D008297:Male; D008875:Middle Aged; D015203:Reproducibility of Results; D018434:Sickness Impact Profile; D013064:Speech Disorders / Q000628:therapy*; D011795:Surveys and Questionnaires; D055815:Young Adult
keywords: HDQLIFE; Health-related quality of life; Huntington disease; Neuro-QoL; PROMIS; Patient-reported outcome (PRO); Speech difficulties; Swallowing difficulties