Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen
ObjectiveThis 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.MethodsThis prospective, observational study enrolled patients with 5q-SMA type I who had lost gross m...
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| Format: | Article |
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Frontiers Media S.A.
2022-09-01
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| Series: | Frontiers in Neurology |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fneur.2022.918255/full |
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| author | Yukako Yae Kotaro Yuge Toshiyuki Maeda Fumio Ichinose Muneaki Matsuo Osamu Kobayashi Kazuo Okanari Yusei Baba Chihiro Yonee Shinsuke Maruyama Minoru Shibata Tatsuya Fujii Madoka Chinen Yushiro Yamashita |
| author_facet | Yukako Yae Kotaro Yuge Toshiyuki Maeda Fumio Ichinose Muneaki Matsuo Osamu Kobayashi Kazuo Okanari Yusei Baba Chihiro Yonee Shinsuke Maruyama Minoru Shibata Tatsuya Fujii Madoka Chinen Yushiro Yamashita |
| author_sort | Yukako Yae |
| collection | DOAJ |
| description | ObjectiveThis 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.MethodsThis 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).ResultsSeven 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.ConclusionsEye-tracking systems are possibly feasible for the assessment of treatment efficacy in patients with advanced SMA type I. |
| first_indexed | 2024-04-12T04:15:40Z |
| format | Article |
| id | doaj.art-663f4153de6f4327be0bf3246ae41d92 |
| institution | Directory Open Access Journal |
| issn | 1664-2295 |
| language | English |
| last_indexed | 2024-04-12T04:15:40Z |
| publishDate | 2022-09-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Neurology |
| spelling | doaj.art-663f4153de6f4327be0bf3246ae41d922022-12-22T03:48:25ZengFrontiers Media S.A.Frontiers in Neurology1664-22952022-09-011310.3389/fneur.2022.918255918255Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersenYukako Yae0Kotaro Yuge1Toshiyuki Maeda2Fumio Ichinose3Muneaki Matsuo4Osamu Kobayashi5Kazuo Okanari6Yusei Baba7Chihiro Yonee8Shinsuke Maruyama9Minoru Shibata10Tatsuya Fujii11Madoka Chinen12Yushiro Yamashita13Department of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, JapanDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, JapanDepartment of Pediatrics, Faculty of Medicine, Saga University, Saga, JapanDepartment of Pediatrics, Faculty of Medicine, Saga University, Saga, JapanDepartment of Pediatrics, Faculty of Medicine, Saga University, Saga, JapanDepartment of Pediatrics, Oita University Faculty of Medicine, Yufu, Oita, JapanDepartment of Pediatrics, Oita University Faculty of Medicine, Yufu, Oita, JapanDepartment of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, JapanDepartment of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, JapanDepartment of Pediatrics, Kagoshima University Graduate School of Medical and Dental Sciences, Kagoshima, JapanDepartment of Pediatrics, Shiga Medical Center for Children, Moriyama, JapanDepartment of Pediatrics, Shiga Medical Center for Children, Moriyama, JapanBiogen Japan Ltd., Tokyo, JapanDepartment of Pediatrics and Child Health, Kurume University School of Medicine, Kurume, Fukuoka, JapanObjectiveThis 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.MethodsThis 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).ResultsSeven 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.ConclusionsEye-tracking systems are possibly feasible for the assessment of treatment efficacy in patients with advanced SMA type I.https://www.frontiersin.org/articles/10.3389/fneur.2022.918255/fulleye-trackingnusinersenspinal muscular atrophyadvancedevaluation methodobservational study |
| spellingShingle | Yukako Yae Kotaro Yuge Toshiyuki Maeda Fumio Ichinose Muneaki Matsuo Osamu Kobayashi Kazuo Okanari Yusei Baba Chihiro Yonee Shinsuke Maruyama Minoru Shibata Tatsuya Fujii Madoka Chinen Yushiro Yamashita Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen Frontiers in Neurology eye-tracking nusinersen spinal muscular atrophy advanced evaluation method observational study |
| title | Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen |
| title_full | Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen |
| title_fullStr | Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen |
| title_full_unstemmed | Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen |
| title_short | Exploratory evaluation of an eye-tracking system in patients with advanced spinal muscular atrophy type I receiving nusinersen |
| title_sort | exploratory evaluation of an eye tracking system in patients with advanced spinal muscular atrophy type i receiving nusinersen |
| topic | eye-tracking nusinersen spinal muscular atrophy advanced evaluation method observational study |
| url | https://www.frontiersin.org/articles/10.3389/fneur.2022.918255/full |
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