TY - JOUR
T1 - Feasibility and utility of in-home body weight support harness system use in young children treated for spinal muscular atrophy: A singlearm prospective cohort study
AU - Iammarino, Megan A.
AU - Alfano, Lindsay N.
AU - Reash, Natalie F.
AU - Sabo, Brenna
AU - Conroy, Sara
AU - Noritz, Garey
AU - Wendland, Madalynn T
AU - Lowes, Linda P.
PY - 2024/3/1
Y1 - 2024/3/1
N2 - Purpose This single-arm prospective cohort study aimed to evaluate the feasibility and utility of inhome body weight support harness system (BWSS) use in children treated for spinal muscular atrophy (SMA). Methods Individuals with 2 or 3 copies of SMN2 who received pharmacotherapeutic treatment, had head control, and weight <50lbs were enrolled. Families were provided a BWSS and documented use. Motor outcome assessments were completed at baseline, month 3 and month 6. Families provided feedback in an end of study survey. Results All 32 participants (2.9 (SD 1.9) yrs), improved or remained stable on all outcomes. Average reported frequency of use was 4.1(2.3) hrs/week. Controlling for other covariates, frequency of use explained over 70% of the variability in change scores. Family feedback was overwhelmingly positive. Conclusion Use of in-home BWSS is a safe, feasible and useful option to increase exercise dosage after treatment in SMA and may help optimize motor abilities.
AB - Purpose This single-arm prospective cohort study aimed to evaluate the feasibility and utility of inhome body weight support harness system (BWSS) use in children treated for spinal muscular atrophy (SMA). Methods Individuals with 2 or 3 copies of SMN2 who received pharmacotherapeutic treatment, had head control, and weight <50lbs were enrolled. Families were provided a BWSS and documented use. Motor outcome assessments were completed at baseline, month 3 and month 6. Families provided feedback in an end of study survey. Results All 32 participants (2.9 (SD 1.9) yrs), improved or remained stable on all outcomes. Average reported frequency of use was 4.1(2.3) hrs/week. Controlling for other covariates, frequency of use explained over 70% of the variability in change scores. Family feedback was overwhelmingly positive. Conclusion Use of in-home BWSS is a safe, feasible and useful option to increase exercise dosage after treatment in SMA and may help optimize motor abilities.
UR - https://www.scopus.com/inward/record.uri?partnerID=HzOxMe3b&scp=85188245060&origin=inward
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U2 - 10.1371/journal.pone.0300244
DO - 10.1371/journal.pone.0300244
M3 - Article
C2 - 38502672
SN - 1932-6203
VL - 19
JO - PLoS ONE
JF - PLoS ONE
IS - 3 March
M1 - e0300244
ER -