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Effects of Non-invasive Brain Stimulation on Hereditary Ataxia: a Systematic Review and Meta-analysis

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Abstract

Numerous studies have demonstrated the potential of non-invasive brain stimulation (NIBS) techniques as a viable treatment option for cerebellar ataxia. However, there is a notable dearth of research investigating the efficacy of NIBS specifically for hereditary ataxia (HA), a distinct subgroup within the broader category of cerebellar ataxia. This study aims to conduct a comprehensive systematic review and meta-analysis in order to assess the efficacy of various NIBS methods for the treatment of HA. A thorough review of the literature was conducted, encompassing both English and Chinese articles, across eight electrical databases. The focus was on original articles investigating the therapeutic effectiveness of non-invasive brain stimulation for hereditary ataxia, with a publication date prior to March 2023. Subsequently, a meta-analysis was performed specifically on randomized controlled trials (RCTs) that fulfilled the eligibility criteria, taking into account the various modalities of non-invasive brain stimulation. A meta-analysis was conducted, comprising five RCTs, which utilized the Scale for the Assessment and Rating of Ataxia (SARA) as the outcome measure to evaluate the effects of transcranial magnetic stimulation (TMS). The findings revealed a statistically significant mean decrease of 1.77 in the total SARA score following repetitive TMS (rTMS) (p=0.006). Subgroup analysis based on frequency demonstrated a mean decrease of 1.61 in the total SARA score after high-frequency rTMS (p=0.05), while no improvement effects were observed after low-frequency rTMS (p=0.48). Another meta-analysis was performed on three studies, utilizing ICARS scores, to assess the impact of rTMS. The results indicated that there were no statistically significant differences in pooled ICARS scores between the rTMS group and the sham group (MD=0.51, 95%CI: -5.38 to 6.39; p=0.87). These findings align with the pooled results of two studies that evaluated alterations in post-intervention BBS scores (MD=0.74, 95%CI: -5.48 to 6.95; p=0.82). Despite the limited number of studies available, this systematic review and meta-analysis have revealed promising potential benefits of rTMS for hereditary ataxia. However, it is strongly recommended that further high-quality investigations be conducted in this area. Furthermore, the significance of standardized protocols for NIBS in future studies was also emphasized.

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Data Availability

All data analyzed in this study are included in published article or its supplementary information files.

Abbreviations

NIBS:

Non-invasive brain stimulation

HAs:

Hereditary ataxias

SCA:

Spinocerebellar ataxia

RCTs:

Random controlled trials

TMS:

Transcranial magnetic stimulation

tDCS:

transcranial direct current stimulation

rTMS:

repetitive pulse TMS

tACS:

transcranial alternating current stimulation

iTBS:

intermittent theta burst stimulation

SARA:

The Scale for the Assessment and Rating of Ataxia

ICARS:

International Cooperative Ataxia Rating Scale

BBS:

Berg Balance Scale

MD:

mean difference

CI:

confidence interval

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Authors and Affiliations

  1. Department of Neurology, The First Hospital of China Medical University, Shenyang City, 110001, Liaoning Province, China

    Ye Liu, Yiming Ma, Jing Zhang, Xuejing Yan & Yi Ouyang

  2. Department of Neurology, The Fifth People’s Hospital of Datong, Datong City, Shanxi Province, China

    Jing Zhang

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Contributions

YL, YM and JZ conducted the study selection process, retrieved data and performed analyses. YL contributed to writing the original manuscript draft. XY and OY contributed to data interpretation and manuscript revisions. All authors read and approved the final manuscript.

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Correspondence to Yi Ouyang.

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Liu, Y., Ma, Y., Zhang, J. et al. Effects of Non-invasive Brain Stimulation on Hereditary Ataxia: a Systematic Review and Meta-analysis. Cerebellum 23, 1614–1625 (2024). https://doi.org/10.1007/s12311-023-01638-x

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