ClinicoEconomics and outcomes research : CEOR · 2026
Medical Claim Costs of Facioscapulohumeral Muscular Dystrophy in the United States.
СтатьяНазвание и аннотация: Перевод в очереди
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Hill Amanda A, Kinoshita June, Deypalan Mariefel Nicole, Kolocha Marina, Hung Man, Hon Eric, Sarraille William, Winegarden Wayne, Ghanjanasak Tanyatorn, Arjomand Jamshid
Аннотация
PURPOSE: Facioscapulohumeral muscular dystrophy (FSHD) is a rare, progressive genetic disorder characterized by asymmetric muscle weakness and functional decline. Although it is one of the most common muscular dystrophies, its economic burden in the United States (US) is largely unknown. This study aimed to provide an initial, conservative estimate of annual medical claims costs and identify comorbidities disproportionately affecting individuals with FSHD in the US.
PATIENTS AND METHODS: This retrospective, 1:5 matched case-control study analyzed de-identified claims data from Medicare and commercially insured enrollees from 2018 to 2021. Medical and prescription costs were aggregated and described as means with 95% confidence intervals. T-tests with Bonferroni correction and chi-square tests were used to compare comorbidity prevalence.
RESULTS: The study included 383 individuals with FSHD and 1915 matched controls. The mean annual medical claims cost for the FSHD cohort was $19,370 per person with commercial insurance and $11,704 with Medicare, compared with $5250 among controls. Individuals with FSHD also incurred higher prescription claims costs in commercial and Medicare subgroups without comorbidities. The prevalence of cerebrovascular disease (10.18% vs 4.44%) and ear disorders (8.09% vs. 3.39%) was significantly greater in the FSHD cohort (P<0.05).
CONCLUSION: This exploratory study provides the first US-based description of the direct medical costs of FSHD, demonstrating a substantially higher healthcare burden than that of matched controls. Because the study design likely underestimates costs, the true economic impact may be even greater. These findings establish a critical foundation for future research into the lifetime medical, nonmedical, and caregiver-related costs of FSHD, and highlight the urgent need for effective therapies and supportive care strategies.
PATIENTS AND METHODS: This retrospective, 1:5 matched case-control study analyzed de-identified claims data from Medicare and commercially insured enrollees from 2018 to 2021. Medical and prescription costs were aggregated and described as means with 95% confidence intervals. T-tests with Bonferroni correction and chi-square tests were used to compare comorbidity prevalence.
RESULTS: The study included 383 individuals with FSHD and 1915 matched controls. The mean annual medical claims cost for the FSHD cohort was $19,370 per person with commercial insurance and $11,704 with Medicare, compared with $5250 among controls. Individuals with FSHD also incurred higher prescription claims costs in commercial and Medicare subgroups without comorbidities. The prevalence of cerebrovascular disease (10.18% vs 4.44%) and ear disorders (8.09% vs. 3.39%) was significantly greater in the FSHD cohort (P<0.05).
CONCLUSION: This exploratory study provides the first US-based description of the direct medical costs of FSHD, demonstrating a substantially higher healthcare burden than that of matched controls. Because the study design likely underestimates costs, the true economic impact may be even greater. These findings establish a critical foundation for future research into the lifetime medical, nonmedical, and caregiver-related costs of FSHD, and highlight the urgent need for effective therapies and supportive care strategies.