title: Quantifying the burden of caregiving in Duchenne muscular dystrophy.
abstract: Duchenne muscular dystrophy (DMD) is a rare pediatric neuromuscular disease associated with progressive muscle degeneration and extensive care needs. Our objective was to estimate the caregiver burden associated with DMD. We made cross-sectional assessments of caregiver health-related quality of life (HRQL) and burden using the EuroQol EQ-5D, a Visual Analogue Scale (VAS), the SF-12 Health Survey, and the Zarit Caregiver Burden Interview (ZBI) administered online. Results were stratified by disease stage (early/late ambulatory/non-ambulatory) and caregivers' rating of patients' health and mental status. In total, caregivers to 770 patients participated. Mean EQ-5D utility ranged between 0.85 (95 % CI 0.82-0.88) and 0.77 (0.74-0.80) across ambulatory classes and 0.88 (0.85-0.90) and 0.57 (0.39-0.74) across caregivers' rating of patients' health and mental status. Mean VAS score was 0.74 (0.73-0.75), mean SF-12 Mental Health Component Summary score 44 (43-45), and mean ZBI score 29 (28-30). Anxiety and depression, recorded in up to 70 % of caregivers depending on patients' health and mental status, was significantly associated with annual household cost burden (>$5000 vs. <$1000, odds ratio 1.76, 95 % CI 1.18-2.63) and hours of leisure time devoted to informal care per week (25-50 vs. <25 h 2.01, 1.37-2.94; >50 vs. <25 h 3.35, 2.32-4.83) (p < 0.007). We show that caring for a person with DMD can be associated with a substantial burden and impaired HRQL. Our findings suggest that caregivers to patients with DMD should be screened for depression and emphasize the need for a holistic approach to family mental health in the context of chronic childhood disease.
authors.affiliation: Institute of Environmental Medicine, Karolinska Institutet, Nobels väg 13, SE-17177, Stockholm, Sweden. erik.landfeldt@ki.se.|OptumInsight, Klarabergsviadukten 90, SE-11164, Stockholm, Sweden. erik.landfeldt@ki.se.
authors.forename: Erik
authors.initials: E
authors.lastname: Landfeldt
authors.affiliation: Medical Management Centre, Department of Learning, Informatics, Management and Ethics, Karolinska Institutet, Tomtebodavägen 18A, SE-17177, Stockholm, Sweden.
authors.forename: Peter
authors.initials: P
authors.lastname: Lindgren
authors.affiliation: GlaxoSmithKline, 5 Moore Drive, PO Box 13398, Research Triangle Park, Durham, NC, 27709-3398, USA.
authors.forename: Christopher F
authors.initials: CF
authors.lastname: Bell
authors.affiliation: Newcastle University John Walton Muscular Dystrophy Research Centre and MRC Centre for Neuromuscular Diseases, Institute of Genetic Medicine, International Centre for Life, Central Parkway, Newcastle upon Tyne, NE1 3BZ, UK.
authors.forename: Michela
authors.initials: M
authors.lastname: Guglieri
authors.affiliation: Newcastle University John Walton Muscular Dystrophy Research Centre and MRC Centre for Neuromuscular Diseases, Institute of Genetic Medicine, International Centre for Life, Central Parkway, Newcastle upon Tyne, NE1 3BZ, UK.
authors.forename: Volker
authors.initials: V
authors.lastname: Straub
authors.affiliation: Newcastle University John Walton Muscular Dystrophy Research Centre and MRC Centre for Neuromuscular Diseases, Institute of Genetic Medicine, International Centre for Life, Central Parkway, Newcastle upon Tyne, NE1 3BZ, UK.
authors.forename: Hanns
authors.initials: H
authors.lastname: Lochmüller
authors.affiliation: Newcastle University John Walton Muscular Dystrophy Research Centre and MRC Centre for Neuromuscular Diseases, Institute of Genetic Medicine, International Centre for Life, Central Parkway, Newcastle upon Tyne, NE1 3BZ, UK.
authors.forename: Katharine
authors.initials: K
authors.lastname: Bushby
mesh_terms: D000328:Adult; D001007:Anxiety / Q000453:epidemiology / Q000209:etiology; D017028:Caregivers / Q000523:psychology*; D017281:Cost of Illness; D003430:Cross-Sectional Studies; D003863:Depression / Q000453:epidemiology / Q000209:etiology; D005260:Female; D006801:Humans; D008297:Male; D020388:Muscular Dystrophy, Duchenne / Q000191:economics / Q000628:therapy*; D015995:Prevalence; D011788:Quality of Life; D012959:Socioeconomic Factors
keywords: Caregiver burden; Informal care; Neuromuscular; Quality of life; Utilities