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• W3005454283 abstract "To evaluate the effects of nusinersen on respiratory function of patients with type 1 spinal muscular atrophy.Observational, longitudinal cohort study. We collected respiratory data from 118 children with type 1 spinal muscular atrophy and differing pulmonary requirements and conducted a semistructured qualitative interview among a subsample of caregivers at baseline, 6 months, and 10 months after the first nusinersen treatment. Patients were stratified according to ventilation modalities and age at study entry.Most patients in our cohort remained stable (84/109 = 77%). More than 80% of the children treated before age 2 years survived, in contrast to the lower survival reported in natural history studies, and did so without tracheostomy or noninvasive ventilation (NIV) ≥16 hours. In those less than 2 years old, only 3 patients shifted from NIV ≤10 hours to NIV >10 hours, and the other 3 reduced the hours of NIV required. Most of the older patients remained stable; this included not only those on tracheostomy or NIV >10 hours but also 75% of those on NIV ≤10 hours.Our results suggest that nusinersen may produce some improvement in the progression of respiratory impairment, both in terms of survival and need for respiratory support ≥16 hours, especially before the age of 2 years." @default.
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• W3005454283 date "2020-04-01" @default.
• W3005454283 modified "2023-10-18" @default.
• W3005454283 title "Respiratory Needs in Patients with Type 1 Spinal Muscular Atrophy Treated with Nusinersen" @default.
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• W3005454283 doi "https://doi.org/10.1016/j.jpeds.2019.12.047" @default.
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