title: New measures to capture end of life concerns in Huntington disease: Meaning and Purpose and Concern with Death and Dying from HDQLIFE (a patient-reported outcomes measurement system).
abstract: Huntington disease (HD) is an incurable terminal disease. Thus, end of life (EOL) concerns are common in these individuals. A quantitative measure of EOL concerns in HD would enable a better understanding of how these concerns impact health-related quality of life. Therefore, we developed new measures of EOL for use in HD.
  
  
  
  An EOL item pool of 45 items was field tested in 507 individuals with prodromal or manifest HD. Exploratory and confirmatory factor analyses (EFA and CFA, respectively) were conducted to establish unidimensional item pools. Item response theory (IRT) and differential item functioning analyses were applied to the identified unidimensional item pools to select the final items.
  
  
  
  EFA and CFA supported two separate unidimensional sets of items: Concern with Death and Dying (16 items), and Meaning and Purpose (14 items). IRT and DIF supported the retention of 12 Concern with Death and Dying items and 4 Meaning and Purpose items. IRT data supported the development of both a computer adaptive test (CAT) and a 6-item, static short form for Concern with Death and Dying.
  
  
  
  The HDQLIFE Concern with Death and Dying CAT and corresponding 6-item short form, and the 4-item calibrated HDQLIFE Meaning and Purpose scale demonstrate excellent psychometric properties. These new measures have the potential to provide clinically meaningful information about end-of-life preferences and concerns to clinicians and researchers working with individuals with HD. In addition, these measures may also be relevant and useful for other terminal conditions.
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA. carlozzi@med.umich.edu.
authors.forename: N E
authors.identifier: 0000-0003-0439-9429
authors.initials: NE
authors.lastname: Carlozzi
authors.affiliation: College of Nursing, The University of Iowa, Iowa City, IA, USA.
authors.forename: N R
authors.initials: NR
authors.lastname: Downing
authors.affiliation: Department of Pathology, Rowan University, Piscataway, NJ, USA.
authors.forename: M K
authors.initials: MK
authors.lastname: McCormack
authors.affiliation: Institute for Social Research, University of Michigan, Ann Arbor, MI, USA.
authors.forename: S G
authors.initials: SG
authors.lastname: Schilling
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: Department of Medical Social Sciences, Northwestern University, Chicago, IL, USA.
authors.forename: E A
authors.initials: EA
authors.lastname: Hahn
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: Beth Israel Deaconess Medical Center, Boston, MA, USA.
authors.forename: S
authors.initials: S
authors.lastname: Frank
authors.affiliation: Department of Medical and Molecular Genetics, Indiana University, Indianapolis, IN, USA.
authors.forename: K A
authors.initials: KA
authors.lastname: Quaid
authors.affiliation: Department of Psychiatry, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA.|Department of Neurology, Carver College of Medicine, The University of Iowa, Iowa City, IA, USA.|Department of Psychology, The University of Iowa, Iowa City, IA, USA.
authors.forename: J S
authors.initials: JS
authors.lastname: Paulsen
authors.affiliation: Department of Medical Social Sciences, Northwestern University, Chicago, IL, USA.|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: D
authors.initials: D
authors.lastname: Cella
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.
authors.forename: S M
authors.initials: SM
authors.lastname: Goodnight
authors.affiliation: Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.
authors.forename: J A
authors.initials: JA
authors.lastname: Miner
authors.affiliation: Hennepin County Medical Center, Minneapolis, MN, USA.
authors.forename: M A
authors.initials: MA
authors.lastname: Nance
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D003643:Death; D005260:Female; D006801:Humans; D006816:Huntington Disease / Q000401:mortality / Q000523:psychology*; D008297:Male; D008875:Middle Aged; D000071066:Patient Reported Outcome Measures; D018434:Sickness Impact Profile; D011795:Surveys and Questionnaires; D013727:Terminal Care / Q000523:psychology*; D055815:Young Adult
keywords: End of life; HDQLIFE; Health-related quality of life; Huntington disease; Neuro-QoL; PROMIS; Patient-reported outcome (PRO)