Evidence map›Paper›PMID 38575756›Full record

ArticleJournal of neurology2024

Pallidal deep brain stimulation for patients with myoclonus-dystonia without SGCE mutations.

Jun Ikezawa, Fusako Yokochi, Ryoichi Okiyama, Ayako Isoo, Takashi Agari, Tsutomu Kamiyama, Akihiro Yugeta, Maya Tojima, Takashi Kawasaki, Katsushige Watanabe and 2 more

Abstract readCase Reports
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In one paragraph

Article in Journal of neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.7field-weighted citation impact, top 30% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 2 citations in OpenAlex.

  1. Review
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

12 authors at 6 institutions in 1 country.

Jun IkezawaDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan. jun_ikezawa@tmhp.jp.ORCID http://orcid.org/0000-0003-2424-2218
Fusako YokochiDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Ryoichi OkiyamaDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Ayako IsooDepartment of Neurosurgery, Tokyo Metropolitan Neurological Hospital, Tokyo, Japan.
Takashi AgariDepartment of Neurosurgery, Tokyo Metropolitan Neurological Hospital, Tokyo, Japan.
Tsutomu KamiyamaDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Akihiro YugetaDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Maya TojimaDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Takashi KawasakiDepartment of Neurosurgery, Tokyo Metropolitan Neurological Hospital, Tokyo, Japan.
Katsushige WatanabeDepartment of Neurosurgery, Tokyo Metropolitan Neurological Hospital, Tokyo, Japan.
Satoko KumadaDepartment of Neuropediatrics, Tokyo Metropolitan Neurological Hospital, Tokyo, Japan.
Kazushi TakahashiDepartment of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai, Fuchu, Tokyo, Japan.
Tokyo Metropolitan Neurological Hospital · JPKyoto University · JPNational Center of Neurology and Psychiatry · JPSaiseikai Utsunomiya hospital · JPTokyo Metropolitan Matsuzawa Hospital · JPYokohama City University Medical Center · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundPallidal deep brain stimulation (GPi-DBS) is effective for treating myoclonus and dystonia caused by SGCE mutations (DYT-SGCE, DYT11). However, it is unknown whether GPi-DBS is effective for the treatment of myoclonus-dystonia which is not associated with the SGCE gene mutations. In this study, we investigated the efficacy of GPi-DBS in treating myoclonus-dystonia in SGCE mutation-negative cases.

methodsThree patients with myoclonus-dystonia without SGCE mutations who underwent GPi-DBS were evaluated preoperatively and 6 months postoperatively using the Unified Myoclonus Rating Scale (UMRS) and Fahn-Marsden Dystonia Rating Scale (FMDRS) for myoclonus and dystonia, respectively. In two of the three patients, myoclonus was more evident during action. Myoclonus was predominant at rest in the other patient, and he was unaware of his dystonia symptoms. The results were compared with those of the four DYT-SGCE cases.

resultsThe mean UMRS score in patients with myoclonus-dystonia without SGCE mutations improved from 61.7 to 33.7 pre- and postoperatively, respectively, and the mean FMDRS score improved from 7.2 to 4.5. However, the degree of improvement in myoclonus-dystonia in patients without SGCE mutations was inferior to that in patients with DYT-SGCE (the UMRS score improved by 45% and 69%, respectively).

conclusionsGPi-DBS is effective for treating myoclonus-dystonia in patients with and without SGCE mutations. GPi-DBS should be considered as a treatment option for myoclonus-dystonia without SGCE mutations.

Indexed as

Deep Brain StimulationDystonic DisordersGlobus PallidusMutationSarcoglycansAdolescentAdultFemaleHumansMaleMiddle AgedTreatment OutcomeYoung AdultSarcoglycansSGCE protein, humanDeep brain stimulationDYT-SGCEGlobus pallidusGPi-DBSMyoclonus-dystoniaSubcortical myoclonus

Identifiers

PMID38575756
OpenAlexW4393952184

What OpenQuestion holds

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Read underepoch 390

Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.