Linda P Lowes1, Lindsay N Alfano1, Roger Crawfis2, Katherine Berry1, Han Yin3, Igor Dvorchik3, Kevin M Flanigan1,4,5, Jerry R Mendell1,4,5. 1. The Research Institute at Nationwide Children's Hospital, Center for Gene Therapy, 700 Children's Drive, AB0040, Columbus, Ohio, 43205, USA. 2. Department of Computer Science and Engineering, The Ohio State University, Columbus, Ohio, USA. 3. The Research Institute at Nationwide Children's Hospital, Biostatistics Core, Columbus, Ohio, USA. 4. Department of Pediatrics, The Ohio State University College of Medicine, Columbus, Ohio, USA. 5. Department of Neurology, The Ohio State University College of Medicine, Columbus, Ohio, USA.
Abstract
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.
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.
Authors: David J Birnkrant; Katharine Bushby; Carla M Bann; Susan D Apkon; Angela Blackwell; David Brumbaugh; Laura E Case; Paula R Clemens; Stasia Hadjiyannakis; Shree Pandya; Natalie Street; Jean Tomezsko; Kathryn R Wagner; Leanne M Ward; David R Weber Journal: Lancet Neurol Date: 2018-02-03 Impact factor: 44.182
Authors: Karin J Naarding; Mariska M H P Janssen; Ruben D Boon; Paulina J M Bank; Robert P Matthew; Gregorij Kurillo; Jay J Han; Jan J G M Verschuuren; Imelda J M de Groot; Menno van der Holst; Hermien E Kan; Erik H Niks Journal: J Neuromuscul Dis Date: 2022
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