Symptom-Oriented, Connectome-Informed Deep Brain Stimulation for Asymmetric Dystonic Tremor: Unilateral Ventral Intermediate Nucleus (VIM) DBS Targeting a Tremor-Dominant Network

<b>Background:</b> Deep brain stimulation (DBS) has traditionally followed diagnosis-driven, nucleus-centered targeting paradigms. Increasing evidence supports a circuit-based framework in which clinical outcomes depend on modulation of symptom-relevant networks rather than diagnostic labels alone. This approach is particularly relevant in mixed movement disorder phenotypes such as dystonic tremor, where the most disabling symptom may not align with the conventional surgical target. <b>Methods:</b> We report a clinically illustrative single case treated using a symptom-oriented, connectome-informed DBS strategy. Clinical phenotype, tremor severity, functional impairment, prior medical and botulinum toxin treatments, and longitudinal outcomes were systematically reviewed. DBS target selection prioritized the dominant, treatment-refractory symptom rather than the underlying dystonia diagnosis. Surgical planning incorporated high-resolution MRI with patient-specific thalamic segmentation using Brainlab Brain Elements<sup>®</sup>, followed by postoperative lead localization and volume of tissue activated visualization with the SureTune™ platform. <b>Results:</b> A 54-year-old left-handed woman with long-standing cervical dystonia developed a severe, markedly asymmetric dystonic tremor predominantly affecting the left upper limb, resulting in profound functional disability. Instead of conventional bilateral globus pallidus internus DBS, unilateral right ventral intermediate nucleus (VIM) DBS was selected to engage tremor-related cerebellothalamic circuits. Rapid and marked improvement was observed, with tremor severity reduced to mild levels within 15 days after stimulation onset. At 6-month follow-up, overall tremor severity improved from 49 to 13 points on the Fahn-Tolosa-Marin Tremor Rating Scale, corresponding to a 73.5% reduction. This improvement was associated with restoration of legible handwriting, independent feeding and drinking, and recovery of bimanual fine motor function. Clinical benefit remained stable throughout follow-up, without stimulation-related adverse effects. <b>Conclusions:</b> This case illustrates the feasibility of a symptom-oriented, connectome-informed DBS strategy in selected patients with dystonic tremor. When symptom expression and network involvement are markedly asymmetric, selective unilateral modulation of the tremor-dominant circuit may achieve meaningful and durable functional improvement. Further studies are needed to assess the generalizability of this approach.

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Journal
Journal of Clinical Medicine
Published
2026-02-23
DOI
https://doi.org/10.3390/jcm15041666
Primary Topic
Neurological disorders and treatments
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article
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article

Symptom-Oriented, Connectome-Informed Deep Brain Stimulation for Asymmetric Dystonic Tremor: Unilateral Ventral Intermediate Nucleus (VIM) DBS Targeting a Tremor-Dominant Network

Maria Teresa del Castillo, José Antonio Souto Paz, F. Grandas, P. Vidal Fernández et al.
Journal of Clinical Medicine
Neurological disorders and treatments
article

Symptom-Oriented, Connectome-Informed Deep Brain Stimulation for Asymmetric Dystonic Tremor: Unilateral Ventral Intermediate Nucleus (VIM) DBS Targeting a Tremor-Dominant Network

Maria Teresa del Castillo, José Antonio Souto Paz, F. Grandas, P. Vidal Fernández, Olga Mateo-Sierra, B. De la Casa-Fages, Javier Ricardo Pérez-Sánchez, Pascual Elvira
article en

Abstract

<b>Background:</b> Deep brain stimulation (DBS) has traditionally followed diagnosis-driven, nucleus-centered targeting paradigms. Increasing evidence supports a circuit-based framework in which clinical outcomes depend on modulation of symptom-relevant networks rather than diagnostic labels alone. This approach is particularly relevant in mixed movement disorder phenotypes such as dystonic tremor, where the most disabling symptom may not align with the conventional surgical target. <b>Methods:</b> We report a clinically illustrative single case treated using a symptom-oriented, connectome-informed DBS strategy. Clinical phenotype, tremor severity, functional impairment, prior medical and botulinum toxin treatments, and longitudinal outcomes were systematically reviewed. DBS target selection prioritized the dominant, treatment-refractory symptom rather than the underlying dystonia diagnosis. Surgical planning incorporated high-resolution MRI with patient-specific thalamic segmentation using Brainlab Brain Elements<sup>®</sup>, followed by postoperative lead localization and volume of tissue activated visualization with the SureTune™ platform. <b>Results:</b> A 54-year-old left-handed woman with long-standing cervical dystonia developed a severe, markedly asymmetric dystonic tremor predominantly affecting the left upper limb, resulting in profound functional disability. Instead of conventional bilateral globus pallidus internus DBS, unilateral right ventral intermediate nucleus (VIM) DBS was selected to engage tremor-related cerebellothalamic circuits. Rapid and marked improvement was observed, with tremor severity reduced to mild levels within 15 days after stimulation onset. At 6-month follow-up, overall tremor severity improved from 49 to 13 points on the Fahn-Tolosa-Marin Tremor Rating Scale, corresponding to a 73.5% reduction. This improvement was associated with restoration of legible handwriting, independent feeding and drinking, and recovery of bimanual fine motor function. Clinical benefit remained stable throughout follow-up, without stimulation-related adverse effects. <b>Conclusions:</b> This case illustrates the feasibility of a symptom-oriented, connectome-informed DBS strategy in selected patients with dystonic tremor. When symptom expression and network involvement are markedly asymmetric, selective unilateral modulation of the tremor-dominant circuit may achieve meaningful and durable functional improvement. Further studies are needed to assess the generalizability of this approach.

Journal of Clinical MedicineVol. 15(4)
Universidad Complutense de Madrid (ES), Hospital General Universitario Gregorio Marañón (ES), Hospital General Universitario de Alicante Doctor Balmis (ES)
Good health and well-being
Openalex Percentile: Top 51%
Neurological disorders and treatments
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