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- Title
ACTIVE (Ability Captured Through Interactive Video Evaluation) workspace volume video game to quantify meaningful change in spinal muscular atrophy.
- Authors
Alfano, Lindsay N; Miller, Natalie F; Iammarino, Megan A; Moore Clingenpeel, Melissa; Lowes, Suzanne L; Dugan, Margaret E; Kissel, John T; Al Zaidy, Samiah; Tsao, Chang‐Yong; Lowes, Linda P; Tsao, Chang-Yong
- Abstract
<bold>Aim: </bold>To evaluate the utility of Ability Captured Through Interactive Video Evaluation (ACTIVE) scaled scores to quantify meaningful change in individuals with spinal muscular atrophy (SMA) types 2 or 3 due to disease progression or treatment.<bold>Method: </bold>ACTIVE is a custom-designed video game that measures workspace volume (WSV). Participants included 62 individuals with SMA (mean age [SD] 10y 9mo [5y], range 2y 9mo-24y) and 362 frequency-matched controls (mean age [SD] 10y 9mo [3y 6mo], range 3y 2mo-24y 9mo). Participants completed ACTIVE, other traditional assessments, and patient-reported outcomes. Responsiveness to change was evaluated by comparing longitudinal data on untreated participants to those receiving Spinraza.<bold>Results: </bold>ACTIVE was significantly correlated to the Hammersmith Functional Motor Scales Expanded and Revised Upper Limb Module (ρ=0.85 and ρ=0.92 respectively; p<0.001). Relevance to patients and families was established by strong correlations to the Patient Reported Outcomes Measurement Information System self- and parent proxy-measures of upper extremity ability (ρ=0.63 and ρ=0.70 respectively; p<0.001). Responsiveness to change was demonstrated by significant change in scaled scores after treatment (median 15.9 points, Wilcoxon signed-rank test p<0.01). A preliminary minimum clinically important difference is presented.<bold>Interpretation: </bold>These results suggest that ACTIVE WSV scores are a meaningful assessment with which to quantify change over time in individuals with SMA types 2 and 3.<bold>What This Paper Adds: </bold>Ability Captured Through Interactive Video Evaluation (ACTIVE) quantifies upper extremity function in spinal muscular atrophy. ACTIVE's scaled workspace volume strongly correlates to self- and parent-report of function. ACTIVE quantifies meaningful change after treatment.
- Subjects
SPINAL muscular atrophy; INTERACTIVE videos; VIDEO games; WILCOXON signed-rank test; ARM; MUSCULAR atrophy; DISEASE progression; RESEARCH; RESEARCH methodology; FUNCTIONAL assessment; EVALUATION research; MEDICAL cooperation; SEVERITY of illness index; COMPARATIVE studies
- Publication
Developmental Medicine & Child Neurology, 2020, Vol 62, Issue 3, p303
- ISSN
0012-1622
- Publication type
journal article
- DOI
10.1111/dmcn.14230