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      Long-term effects of the gait treatment using a wearable cyborg hybrid assistive limb in a patient with spinal and bulbar muscular atrophy: a case report with 5 years of follow-up

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          Abstract

          Background

          Spinal and bulbar muscular atrophy (SBMA) is a progressive neuromuscular degenerative disease characterized by the degeneration of lower motor neurons in the spinal cord and brainstem and neurogenic atrophy of the skeletal muscle. Although the short-term effectiveness of gait treatment using a wearable cyborg hybrid assistive limb (HAL) has been demonstrated for the rehabilitation of patients with SBMA, the long-term effects of this treatment are unclear. Thus, this study aimed to investigate the long-term effects of the continued gait treatment with HAL in a patient with SBMA.

          Results

          A 68-year-old man with SBMA had lower limb muscle weakness and atrophy, gait asymmetry, and decreased walking endurance. The patient performed nine courses of HAL gait treatment (as one course three times per week for 3 weeks, totaling nine times) for ~5 years. The patient performed HAL gait treatment to improve gait symmetry and endurance. A physical therapist adjusted HAL based on the gait analysis and physical function of the patient. Outcome measurements, such as 2-min walking distance (2MWD), 10-meter walking test (maximal walking speed, step length, cadence, and gait symmetry), muscle strength, Revised Amyotrophic Lateral Sclerosis Functional Assessment Scale (ALSFRS-R), and patient-reported outcomes, were evaluated immediately before and after gait treatment with HAL for each course. 2MWD improved from 94 m to 101.8 m, and the ALSFRS-R gait items remained unchanged (score 3) for approximately 5 years. The patient could maintain walking ability in terms of gait symmetry, walking endurance, and independence walking despite disease progression during HAL treatment.

          Conclusion

          The long-term gait treatment with HAL in a patient with SBMA may contribute to the maintenance and improvement of the gait endurance and ability to perform activities of daily living. The cybernics treatment using HAL may enable patients to relearn correct gait movements. The gait analysis and physical function assessment by a physical therapist might be important to maximize the benefits of HAL treatment.

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          Most cited references25

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            The ALSFRS-R: a revised ALS functional rating scale that incorporates assessments of respiratory function. BDNF ALS Study Group (Phase III).

            A Nakanishi, N Stambler, B Thurmond … (1999)
            The ALS Functional Rating Scale (ALSFRS) is a validated rating instrument for monitoring the progression of disability in patients with amyotrophic lateral sclerosis (ALS). One weakness of the ALSFRS as originally designed was that it granted disproportionate weighting to limb and bulbar, as compared to respiratory, dysfunction. We have now validated a revised version of the ALSFRS, which incorporates additional assessments of dyspnea, orthopnea, and the need for ventilatory support. The Revised ALSFRS (ALSFRS-R) retains the properties of the original scale and shows strong internal consistency and construct validity. ALSFRS-R scores correlate significantly with quality of life as measured by the Sickness Impact Profile, indicating that the quality of function is a strong determinant of quality of life in ALS.
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              Progressive proximal spinal and bulbar muscular atrophy of late onset. A sex-linked recessive trait.

              Malcolm Kennedy, H Sung, M Alter (1968)
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                Author and article information

                Contributors
                Journal
                Journal ID (nlm-ta): Front Neurol
                Journal ID (iso-abbrev): Front Neurol
                Journal ID (publisher-id): Front. Neurol.
                Title: Frontiers in Neurology
                Publisher: Frontiers Media S.A.
                ISSN (Electronic): 1664-2295
                Publication date (Electronic): 08 June 2023
                Publication date Collection: 2023
                Volume: 14
                Electronic Location Identifier: 1143820
                Affiliations
                [1] 1Department of Rehabilitation, Hokuyukai Neurological Hospital , Sapporo, Japan
                [2] 2Department of Neurosurgery, Institute of Medicine, University of Tsukuba , Tsukuba, Japan
                [3] 3Department of Neurology, Hokuyukai Neurological Hospital , Sapporo, Japan
                Author notes

                Edited by: Ross D. Zafonte, Harvard Medical School, United States

                Reviewed by: Bo Cui, Capital Medical University, China; Masahisa Katsuno, Nagoya University Hospital, Japan

                *Correspondence: Hiroki Watanabe watanabe.hiroki.gb@ 123456u.tsukuba.ac.jp
                Article
                DOI: 10.3389/fneur.2023.1143820
                PMC ID: 10285061
                SO-VID: 14775aca-8ad6-4f07-be65-052d6967fa1a
                Copyright © Copyright © 2023 Iijima, Watanabe, Nakashiro, Iida, Nonaka, Moriwaka and Hamada.
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                History
                Date received : 13 January 2023
                Date accepted : 09 May 2023
                Page count
                Figures: 2, Tables: 2, Equations: 1, References: 26, Pages: 10, Words: 7126
                Funding
                This study was supported by the hospital's operating grant.
                Categories
                Subject: Neurology
                Subject: Case Report
                Custom metadata
                section-at-acceptance Neurorehabilitation

                ScienceOpen disciplines: Neurology
                Keywords: spinal and bulbar muscular atrophy,wearable cyborg hybrid assistive limb (hal),gait treatment,walking symmetry,walking endurance
                Data availability:
                ScienceOpen disciplines: Neurology
                Keywords: spinal and bulbar muscular atrophy, wearable cyborg hybrid assistive limb (hal), gait treatment, walking symmetry, walking endurance

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