Related Experiment Video
Updated: Apr 18, 2026
Measurements of Motor Function and Other Clinical Outcome Parameters in Ambulant Children with Duchenne Muscular Dystrophy
Published on: January 12, 2019
Reliability and validity of active-seated: An outcome in dystrophinopathy
Linda P Lowes1, Lindsay N Alfano1, Roger Crawfis2
1The Research Institute at Nationwide Children's Hospital, Center for Gene Therapy, 700 Children's Drive, AB0040, Columbus, Ohio, 43205, USA.
Introduction:
Traditional upper extremity measures typically focus on distal abilities and do not quantify the unique progression of decline in dystrophinopathy. We designed ACTIVE-seated to meet this need. Our objective was to establish the tool's validity and reliability.
Methods:
ACTIVE-seated uses the Microsoft Kinect gaming interface to quantify functional reaching ability while playing a custom-designed game. A skeletal tracking algorithm was used to determine the furthest arm excursion in all planes in 61 subjects with dystrophinopathy and 16 controls.
Results:
Total reachable area was scaled based on arm length to standardize comparisons across subjects and accommodate growth. ACTIVE-seated discriminately ranked subjects from normal controls and by Brooke level (P < 0.001). Scores were highly correlated with parent reports of daily activities and mobility (P < 0.05). Test-retest reliability of ACTIVE-seated was excellent (ICC = 0.97, P < 0.0001).
Conclusions:
Initial evaluation of reliability and validity suggests that ACTIVE-seated shows promise as a clinical and research outcome for individuals with dystrophinopathy.
