Financial Toxicity and Its Determinants in Individuals Living With Inherited and Acquired Neuromuscular Disorders: The BIND Study

患有遗传性和获得性神经肌肉疾病的个体所面临的财务毒性及其决定因素:BIND 研究

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Abstract

BACKGROUND AND OBJECTIVES: Individuals living with neuromuscular disease (NMD) often face substantial financial strain due to the chronic and progressive nature of these conditions. Despite growing awareness, the extent and determinants of financial toxicity (FT) in this population remain poorly understood. We assessed FT among individuals with NMD or their caregivers in Canada using a validated patient-reported outcome measure. METHODS: We conducted a national, cross-sectional survey of individuals with NMD or their caregivers registered with Muscular Dystrophy Canada (MDC). The survey, available in English and French, was completed online or by telephone with assistance from MDC personnel. FT was measured using the validated Comprehensive Score for Financial Toxicity (COST)-Functional Assessment of Chronic Illness Therapy questionnaire. Multivariable generalized linear models were used to identify factors associated with FT. RESULTS: A total of 1,426 participants (mean [SD] age, 52.0 [19.5] years; 52% female) completed the survey. Seventy-five percent reported household incomes below the national median, primarily because of reduced employment. The average COST score was 17.2 [10.4], significantly lower than scores reported in other chronic conditions, including cancer. Moreover, 70% of respondents had COST scores below 26, indicating moderate or severe FT. After adjusting for other factors, COST scores were significantly worse among caregivers than in adults with NMDs. Lower scores were also observed among individuals identified as racialized minority (rate ratio [RR] 0.74; 95% CI 0.64-0.87); those with lower household income (RR 0.59; 95% CI 0.51-0.69), those with lower education (RR 0.91; 95% CI 0.82-1.00), and those unable to work (RR 0.74; 95% CI 0.64-0.86); students (RR 0.54; 95% CI 0.34-0.86); or early retirees (RR 0.86, 95% CI 0.74-0.99). FT was also greater among those diagnosed with autoimmune myopathies (RR 0.82; 95% CI 0.73-0.92), spinal muscular atrophy (RR 0.79; 95% CI 0.66-0.96), and limb-girdle muscular dystrophy (RR 0.87; 95% CI 0.77-0.99). DISCUSSION: FT is highly prevalent among people living with NMDs, particularly caregivers and those with socioeconomic and clinical vulnerabilities. Policies to improve financial support and health care coverage for NMD-related needs could help alleviate this burden. Further research is required to understand the mechanisms contributing to FT within NMD subgroups.

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