Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen.

Autor:	Yae Y; Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan., Yuge K; Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan., Maeda T; Department of Pediatrics, Faculty of Medicine, Saga University, Saga, Japan., Ichinose F; Department of Pediatrics, Faculty of Medicine, Saga University, Saga, Japan., Matsuo M; Department of Pediatrics, Faculty of Medicine, Saga University, Saga, Japan., Kobayashi O; Department of Pediatrics, Oita University Faculty of Medicine, Yufu, Oita, Japan., Okanari K; Department of Pediatrics, Oita University Faculty of Medicine, Yufu, Oita, Japan., Baba Y; Department of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan., Yonee C; Department of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan., Maruyama S; Department of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, Japan., Shibata M; Department of Pediatrics, Shiga Medical Center for Children, Moriyama, Japan., Fujii T; Department of Pediatrics, Shiga Medical Center for Children, Moriyama, Japan., Chinen M; Biogen Japan Ltd., Tokyo, Japan., Yamashita Y; Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, Japan.
Jazyk:	angličtina
Zdroj:	Frontiers in neurology [Front Neurol] 2022 Sep 30; Vol. 13, pp. 918255. Date of Electronic Publication: 2022 Sep 30 (Print Publication: 2022).
DOI:	10.3389/fneur.2022.918255
Abstrakt:	Objective: This study evaluated the feasibility of a matching-pair test using eye-tracking technology to assess nusinersen effectiveness in patients with advanced spinal muscular atrophy (SMA) type I. Methods: This prospective, observational study enrolled patients with 5q-SMA type I who had lost gross motor function. Three different levels of matching-pair tests were conducted using the eye-gaze system (My Tobii; TobiiDynavox Inc.) at baseline, and after 9 and 24 weeks of nusinersen treatment. The primary endpoint was the change from baseline in matching-pair test scores and response times (i.e., the time to answer matching-pair test) at 24 weeks from baseline. Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP-INTEND), Pediatric Quality of Life inventory for patients with Neuromuscular Disease (PedsQL-NM) and Numerical Rating Scale (NRS) scores were also assessed as secondary endpoints. Analysis of ocular fixation was performed as an additional analysis. This study was registered at https://www.umin.ac.jp/ctr/ (UMIN000033935). Results: Seven patients (one male, six female) aged 5-21 years (median 11 years) were enrolled; all patients were bedridden and six patients were ventilated. All seven patients were able to conduct level 1 matching-pair tests at each assessment; five patients were also able to conduct levels 2 and 3. Two patients (those with the highest CHOP-INTEND scores) were able to complete all tests correctly within 60 s. There was a non-significant trend toward improvement in CHOP-INTEND, PedsQL-NM, and NRS scores over the 6-month period. There were no significant differences in the number of actions, errors, correct answers, or response times between baseline and Week 9 or 24 at any level. However, the result of an additional analysis suggests that detection of eye movement would be useful to evaluate for advanced SMA. Conclusions: Eye-tracking systems are possibly feasible for the assessment of treatment efficacy in patients with advanced SMA type I. Competing Interests: This study received funding from Biogen Japan Ltd (Tokyo, Japan). The funder had the following involvement with the study: design, analysis and interpretation, writing and the decision to submit this article for publication. All named authors have contributed significantly to the intellectual content of the manuscript, and have critically reviewed all drafts and approved the final version for submission. Author KY has received speaker honoraria from Biogen Japan, Novartis Japan, and Chugai Pharmaceutical for activities unrelated to the present work. Author OK has received honoraria from Biogen Japan and UCB Japan for activities unrelated to the present work. Authors SM and TF have attended a Biogen Japan advisory board unrelated to the present work. Author YYu has received honoraria from Shionogi, Novartis, Takeda, Daiichi Sankyo, Novel Pharma and Janssen for activities unrelated to the present work. Author MC is an employee of Biogen Japan Ltd. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. (Copyright © 2022 Yae, Yuge, Maeda, Ichinose, Matsuo, Kobayashi, Okanari, Baba, Yonee, Maruyama, Shibata, Fujii, Chinen and Yamashita.)
Databáze:	MEDLINE
Externí odkaz:	Zobrazit plný text záznamu