Holmes Tremor in CADASIL Responsive to Multi-Targeting Deep Brain Stimulation (DBS): An Educational Case with Video and Electrophysiology.

IF 1.9 Q2 CLINICAL NEUROLOGY

Tremor and Other Hyperkinetic Movements Pub Date : 2026-05-08 eCollection Date: 2026-01-01 DOI:10.5334/tohm.1128

Fahimeh H Akhoundi, Katherine Longardner, Dietrich Haubenberger, Robert Hess, Brenton A Wright, Sharona Ben-Haim

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Abstract

Background: Holmes tremor (HT) is a disabling condition, often resistant to medical therapy.

Case report: A 51-year-old woman with cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL) presented with severe right upper limb HT unresponsive to multiple medications. She underwent deep brain stimulation (DBS) with dual-lead, triple-targeting of the left ventral intermediate nucleus (VIM), posterior subthalamic area (PSA), and globus pallidus interna (GPi) with improvement in tremor.

Discussion: To our knowledge, this case represents the first reported instance of HT in CADASIL and highlights the potential of multi-target DBS for complex, treatment-refractory hyperkinetic movement disorders.

Highlights: We describe the first reported instance of Holmes tremor occurring as a manifestation of CADASIL, successfully treated with triple-target deep brain stimulation of the VIM, PSA, and GPi. This case demonstrates the potential of multi-target DBS to modulate complex motor networks in treatment-resistant tremor and enhance functional outcomes.

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多靶点深部脑刺激（DBS）对CADASIL福尔摩斯震颤的反应：一个视频和电生理学的教育案例。

背景：霍尔姆斯震颤（HT）是一种致残疾病，通常对药物治疗有抗性。病例报告：一名51岁女性大脑常染色体显性动脉病变伴皮质下梗死和白质脑病（CADASIL），表现为严重的右上肢HT，对多种药物无反应。她接受了深部脑刺激（DBS），双导联，左腹侧中间核（VIM），后丘脑底区（PSA）和内白球（GPi）的三重靶向治疗，震颤有所改善。讨论：据我们所知，该病例代表了CADASIL中首次报道的HT病例，并突出了多靶点DBS治疗复杂、难治性多运动障碍的潜力。重点：我们描述了首次报道的福尔摩斯震颤作为CADASIL的表现，通过VIM， PSA和GPi的三靶点深部脑刺激成功治疗。本病例证明了多靶点DBS在治疗抵抗性震颤中调节复杂运动网络和增强功能结果的潜力。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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