Prevalence of morbidities across the lifespan for adults with spinal muscular atrophy: a retrospective cohort study

Autor: Daniel G. Whitney, Erin E. Neil Knierbein, Alecia K. Daunter
Jazyk: angličtina
Rok vydání: 2023
Předmět:
Zdroj: Orphanet Journal of Rare Diseases, Vol 18, Iss 1, Pp 1-11 (2023)
Druh dokumentu: article
ISSN: 1750-1172
DOI: 10.1186/s13023-023-02872-6
Popis: Abstract Background Recently approved treatments for spinal muscular atrophy (SMA) may shift clinical care priorities to secondary complications associated with SMA-related aging. To date, there is little knowledge about the natural history of morbidities across the adult lifespan for SMA. The objective of this study was to identify the prevalence and odds ratio (OR) of various morbidities among adults with vs. without SMA prior to SMA-related treatment. Methods This was a retrospective cohort study that accessed Medicare fee-for-service and commercial claims data from 01/01/2008-12/22/2016. Data from adults ≥ 18 years old with SMA and without SMA matched (1:200 case:control) on demographics, region, and study entry year were included. The prevalence of 30 morbidities across physiologic systems (e.g., cardiovascular, metabolic, musculoskeletal, urinary) and mental health disorders was examined. Age- and sex-adjusted OR was estimated using logistic regression for each morbidity and effect modification by age and sex was tested. Results There were 2,427 adults with SMA (mean [SD] age, 59.7 [17.4] years; 49.0% female) and 484,528 matched adults without SMA. Adults with vs. without SMA had a higher prevalence and adjusted OR of all 30 morbidities, ranging from OR = 1.61 (95% CI = 1.45–1.80) for hypothyroidism to OR = 7.80 (95% CI = 7.10–8.57) for fluid/electrolyte disorders. There was effect modification by age for 24 morbidities. The OR was highest for the youngest age group (18–40 years; OR range, 2.38 to 117.7; all P
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