The Black Box of Technological Outcome Measures: An Example in Duchenne Muscular Dystrophy

Autor: 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
Jazyk: angličtina
Rok vydání: 2022
Předmět:
Zdroj: Journal of Neuromuscular Diseases, 9(4), 555-569. IOS PRESS
Journal of Neuromuscular Diseases, 9, 555-569
Journal of Neuromuscular Diseases, 9, 4, pp. 555-569
ISSN: 2214-3599
Popis: Background: Outcome measures for non-ambulant Duchenne muscular dystrophy (DMD) patients are limited, with only the Performance of the Upper Limb (PUL) approved as endpoint for clinical trials. Objective: We assessed four outcome measures based on devices developed for the gaming industry, aiming to overcome disadvantages of observer-dependency and motivation. Methods: Twenty-two non-ambulant DMD patients (range 8.6–24.1 years) and 14 healthy controls (HC; range 9.5–25.4 years) were studied at baseline and 16 patients at 12 months using Leap Motion to quantify wrist/hand active range of motion (aROM) and a Kinect sensor for reached volume with Ability Captured Through Interactive Video Evaluation (ACTIVE), Functional Workspace (FWS) summed distance to seven upper extremity body points, and trunk compensation (KinectTC). PUL 2.0 was performed in patients only. A stepwise approach assessed quality control, construct validity, reliability, concurrent validity, longitudinal change and patient perception. Results: Leap Motion aROM distinguished patients and HCs for supination, radial deviation and wrist flexion (range p = 0.006 to
Databáze: OpenAIRE