Measuring carer quality of life in Duchenne muscular dystrophy: a systematic review of the reliability and validity of self-report instruments using COSMIN.

Publisher: BioMed Central Country of Publication: England NLM ID: 101153626 Publication Model: Electronic Cited Medium: Internet ISSN: 1477-7525 (Electronic) Linking ISSN: 14777525 NLM ISO Abbreviation: Health Qual Life Outcomes Subsets: MEDLINE

Original Publication: [London] : BioMed Central, c2003-

Caregivers* ; Muscular Dystrophy, Duchenne*; Humans ; Quality of Life ; Reproducibility of Results ; Self Report ; Surveys and Questionnaires

Introduction: Duchenne muscular dystrophy is a rare, progressive, life-limiting genetic neuromuscular condition that significantly impacts the quality of life of informal caregivers. Carer quality of life is measured using heterogeneous self-report scales, yet their suitability for Duchenne remains unclear. This review aimed to identify and evaluate the reliability and validity of quality of life instruments in Duchenne carers.
Materials and Methods: Systematic searches were conducted in Embase, MEDLINE, CINAHL, PsycINFO, Cochrane Library and Google Scholar. Full research articles reporting data on multiple-item self-report quality of life instruments in informal Duchenne carers were included. Extracted evidence was qualitatively synthesised and evaluated, including risk of bias, against the Consensus-based Standards for the selection of health Measurement Instruments. Duchenne carer collaborators (N = 17) helped rate the instruments' content validity.
Results: Thirty-one articles featuring thirty-two quality of life instruments were included. Content validity was rated as "inconsistent" based on very low quality evidence. For Duchenne carer collaborators, the best instrument was PedsQL Family Impact Module. Only one instrument had evidence for structural validity (rated "unsatisfactory") and measurement invariance (rated "satisfactory"). Instruments received "satisfactory" ratings for internal consistency and mixed ratings for construct validity and responsiveness. There was no evidence for reliability, measurement error, or criterion validity.
Discussion: Instruments used to measure Duchenne carer quality of life have limited and often inconsistent supportive psychometric evidence. Further work must investigate instruments' measurement properties in Duchenne carers and/or the development of new tools. In the interim, we recommend considering the PedsQL Family Impact Module based on Duchenne carer ratings.
(© 2022. The Author(s).)

Disabil Rehabil. 2012;34(12):988-96. (PMID: 22149389)
Trials. 2016 Sep 13;17(1):449. (PMID: 27618914)
J Epidemiol Community Health. 1999 Jan;53(1):46-50. (PMID: 10326053)
J Clin Psychol. 1988 May;44(3):335-54. (PMID: 2968371)
Am J Med Genet C Semin Med Genet. 2006 Nov 15;142C(4):232-40. (PMID: 17068805)
Eur J Health Econ. 2016 Apr;17 Suppl 1:19-29. (PMID: 27038625)
Fam Community Health. 2007 Oct-Dec;30(4):296-304. (PMID: 17873636)
Health Policy. 1990 Dec;16(3):199-208. (PMID: 10109801)
Patient Educ Couns. 2013 Nov;93(2):248-54. (PMID: 23993396)
Arch Gen Psychiatry. 1961 Jun;4:561-71. (PMID: 13688369)
Qual Life Res. 2006 Aug;15(6):1005-21. (PMID: 16900281)
Psychol Med. 1998 May;28(3):551-8. (PMID: 9626712)
Med Care. 1996 Mar;34(3):220-33. (PMID: 8628042)
Kaohsiung J Med Sci. 2008 Oct;24(10):514-22. (PMID: 19181582)
J Patient Rep Outcomes. 2021 Aug 10;5(1):71. (PMID: 34374872)
Qual Life Res. 2016 Apr;25(4):767-79. (PMID: 26346986)
Health Qual Life Outcomes. 2020 Aug 3;18(1):263. (PMID: 32746836)
Psychiatry Res. 1989 May;28(2):193-213. (PMID: 2748771)
J Clin Epidemiol. 2010 Jul;63(7):737-45. (PMID: 20494804)
J Nurs Res. 2010 Dec;18(4):239-48. (PMID: 21139443)
Health Info Libr J. 2010 Jun;27(2):114-22. (PMID: 20565552)
Fam Pract. 1991 Dec;8(4):396-401. (PMID: 1800209)
BMJ. 2008 Apr 26;336(7650):924-6. (PMID: 18436948)
BMJ. 2021 Mar 29;372:n71. (PMID: 33782057)
J Clin Epidemiol. 2018 Mar;95:73-93. (PMID: 29154811)
Ann Acad Med Singap. 2020 Feb;49(2):72-77. (PMID: 32246708)
J Health Soc Behav. 1983 Dec;24(4):385-96. (PMID: 6668417)
J Cardiopulm Rehabil. 2003 Nov-Dec;23(6):398-403. (PMID: 14646785)
J Fam Pract. 1978 Jun;6(6):1231-9. (PMID: 660126)
Spine (Phila Pa 1976). 2000 Dec 15;25(24):3130-9. (PMID: 11124729)
Gerontologist. 1980 Dec;20(6):649-55. (PMID: 7203086)
J Pers Assess. 1985 Feb;49(1):71-5. (PMID: 16367493)
Health Soc Work. 2014 Aug;39(3):144-52. (PMID: 25095627)
Health Policy. 1996 Jul;37(1):53-72. (PMID: 10158943)
Disabil Rehabil. 2019 Apr;41(8):966-973. (PMID: 29254382)
Neurology. 2015 Mar 10;84(10):1034-42. (PMID: 25663223)
Health Econ. 1993 Oct;2(3):217-27. (PMID: 8275167)
Qual Life Res. 2011 Dec;20(10):1727-36. (PMID: 21479777)
Gerontologist. 1979 Dec;19(6):586-93. (PMID: 527846)
Diabetologia. 2021 Jun;64(6):1213-1225. (PMID: 33528625)
Acta Psychiatr Scand. 1983 Jun;67(6):361-70. (PMID: 6880820)
Soc Sci Med. 1995 Nov;41(10):1403-9. (PMID: 8560308)
Am Heart J. 2000 Jan;139(1 Pt 1):1-9. (PMID: 10618555)
Acta Myol. 2014 Dec;33(3):136-43. (PMID: 25873782)
Med Care. 1990 Nov;28(11):1056-72. (PMID: 2250492)
Sex Med. 2014 Aug;2(3):133-40. (PMID: 25356310)
Health Soc Work. 1995 May;20(2):87-92. (PMID: 7544318)
Psychopharmacol Bull. 1973 Jan;9(1):13-28. (PMID: 4682398)
Neuromuscul Disord. 2018 Jan;28(1):100. (PMID: 29198861)
Eur J Ageing. 2016;13:271-279. (PMID: 27610055)
Sleep Breath. 2016 Mar;20(1):129-34. (PMID: 26003786)
Health Qual Life Outcomes. 2018 Dec 19;16(1):237. (PMID: 30567556)
J Clin Psychol. 1992 Jan;48(1):11-9. (PMID: 1556205)
J Occup Rehabil. 2020 Sep;30(3):420-454. (PMID: 31939009)
Med Care. 1992 Jun;30(6):473-83. (PMID: 1593914)
Orphanet J Rare Dis. 2020 Jun 5;15(1):141. (PMID: 32503598)
BMJ Open. 2017 Mar 29;7(3):e013611. (PMID: 28360239)
Arq Neuropsiquiatr. 2015 Jan;73(1):52-7. (PMID: 25608128)
Health Soc Care Community. 2010 Sep;18(5):520-8. (PMID: 20561071)
Chin Med J (Engl). 2010 Feb 20;123(4):452-7. (PMID: 20193486)
Muscle Nerve. 2015 Jul;52(1):13-21. (PMID: 25363165)
J Sex Marital Ther. 2000 Apr-Jun;26(2):191-208. (PMID: 10782451)
J Neurol. 2018 Jul;265(7):1548-1556. (PMID: 29700644)
Sleep. 1991 Dec;14(6):540-5. (PMID: 1798888)
J Child Health Care. 2019 Sep;23(3):495-506. (PMID: 31220930)
Sleep Med. 2017 Apr;32:114-121. (PMID: 28366322)
J Clin Med. 2019 Jan 27;8(2):. (PMID: 30691204)
Soc Psychiatry Psychiatr Epidemiol. 1998 Sep;33(9):405-12. (PMID: 9766166)
Qual Life Res. 2018 May;27(5):1159-1170. (PMID: 29550964)
J Child Neurol. 2021 Mar;36(3):177-185. (PMID: 33034535)
Health Qual Life Outcomes. 2004 Sep 27;2:55. (PMID: 15450120)
Biometrics. 1977 Mar;33(1):159-74. (PMID: 843571)
J Neurosci Nurs. 2012 Apr;44(2):82-90. (PMID: 22367270)
Neurology. 2014 Aug 5;83(6):529-36. (PMID: 24991029)
J Neurol. 2016 May;263(5):906-915. (PMID: 26964543)
Med Care. 1981 Aug;19(8):806-28. (PMID: 7278417)
Neurology. 2021 May 11;96(19):e2438-e2450. (PMID: 33785551)
Qual Life Res. 2018 May;27(5):1171-1179. (PMID: 29260445)
Gerontologist. 2001 Oct;41(5):652-7. (PMID: 11574710)
Eur J Epidemiol. 2020 Jul;35(7):643-653. (PMID: 32107739)
Int J Rehabil Res. 2001 Jun;24(2):83-93. (PMID: 11421396)
Eur J Hum Genet. 2016 May;24(5):633-7. (PMID: 26306645)
Injury. 2019 Nov;50(11):1816-1825. (PMID: 31353094)
Qual Life Res. 2018 May;27(5):1147-1157. (PMID: 29435801)
Dev Med Child Neurol. 2018 Oct;60(10):987-996. (PMID: 29904912)
J Gerontol. 1983 May;38(3):344-8. (PMID: 6841931)
Psychol Med. 2002 Aug;32(6):959-76. (PMID: 12214795)
Int J Nurs Stud. 2010 Aug;47(8):984-93. (PMID: 20116060)

Keywords: Carer; Duchenne muscular dystrophy; Psychometrics; Quality of life; Reliability; Systematic review; Validity

Date Created: 20220403 Date Completed: 20220405 Latest Revision: 20220408

20240829

PMC8977045

10.1186/s12955-022-01964-4

35366897