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

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.