Hemidystonia improved by intrathecal baclofen infusion therapy: A case report

Intrathecal baclofen therapy (ITB) is a well-established treatment for limb spasticity due to brain and spinal cord disorders, and it is also indicated for dystonia, though mainly reported in generalized or pediatric cases. This report highlights a 46-year-old male with hemidystonia from writer’s cr...

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Main Authors: Takashi Asahi, Kiyonobu Ikeda, Shiro Horisawa, Taku Nonaka, Yuko Muro, Nobutaka Yamamoto, Takaomi Taira
Format: Article
Language:English
Published: Elsevier 2025-03-01
Series:Interdisciplinary Neurosurgery
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Online Access:http://www.sciencedirect.com/science/article/pii/S2214751925000076
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Summary:Intrathecal baclofen therapy (ITB) is a well-established treatment for limb spasticity due to brain and spinal cord disorders, and it is also indicated for dystonia, though mainly reported in generalized or pediatric cases. This report highlights a 46-year-old male with hemidystonia from writer’s cramp. Diagnosed at age 30, his symptoms persisted despite oral medications, botulinum toxin injections, and deep brain stimulation targeting the left thalamus ventro-oral nucleus and globus pallidus internus. Two years later, his symptoms worsened and extended to the right side. At our clinic, a baclofen screening test significantly reduced muscle tone, enabling writing. Following the implantation of a baclofen pump, his dystonia markedly improved, restoring his writing ability. This case supports considering ITB for patients with severe, refractory dystonia and muscle hypertonus, even in cases without generalized dystonia.
ISSN:2214-7519