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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author Takashi Asahi
Kiyonobu Ikeda
Shiro Horisawa
Taku Nonaka
Yuko Muro
Nobutaka Yamamoto
Takaomi Taira
author_facet Takashi Asahi
Kiyonobu Ikeda
Shiro Horisawa
Taku Nonaka
Yuko Muro
Nobutaka Yamamoto
Takaomi Taira
author_sort Takashi Asahi
collection DOAJ
description 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.
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issn 2214-7519
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publishDate 2025-03-01
publisher Elsevier
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series Interdisciplinary Neurosurgery
spelling doaj-art-4e62ff3fae364049a431fb79b08067fb2025-08-20T02:57:47ZengElsevierInterdisciplinary Neurosurgery2214-75192025-03-013910199510.1016/j.inat.2025.101995Hemidystonia improved by intrathecal baclofen infusion therapy: A case reportTakashi Asahi0Kiyonobu Ikeda1Shiro Horisawa2Taku Nonaka3Yuko Muro4Nobutaka Yamamoto5Takaomi Taira6Department of Neurosurgery, Kanazawa Neurosurgical Hospital, Nonoichi, Ishikawa, Japan; Corresponding author at: Department of Neurosurgery, Kanazawa Neurosurgical Hospital, 262-2 Go-machi, Nonoichi-shi, Ishikawa 921-8841, Japan.Department of Neurosurgery, Kanazawa Neurosurgical Hospital, Nonoichi, Ishikawa, JapanDepartment of Clinical Engineering, Kanazawa Neurosurgical Hospital, Ishikawa, JapanDepartment of Neurosurgery, Kanazawa Neurosurgical Hospital, Nonoichi, Ishikawa, JapanDepartment of Neurosurgery, Tokyo Women’s Hospital, Tokyo, JapanDepartment of Neurosurgery, Kanazawa Neurosurgical Hospital, Nonoichi, Ishikawa, JapanDepartment of Clinical Engineering, Kanazawa Neurosurgical Hospital, Ishikawa, JapanIntrathecal 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.http://www.sciencedirect.com/science/article/pii/S2214751925000076Intrathecal baclofenDystoniaDeep brain stimulation
spellingShingle Takashi Asahi
Kiyonobu Ikeda
Shiro Horisawa
Taku Nonaka
Yuko Muro
Nobutaka Yamamoto
Takaomi Taira
Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
Interdisciplinary Neurosurgery
Intrathecal baclofen
Dystonia
Deep brain stimulation
title Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
title_full Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
title_fullStr Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
title_full_unstemmed Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
title_short Hemidystonia improved by intrathecal baclofen infusion therapy: A case report
title_sort hemidystonia improved by intrathecal baclofen infusion therapy a case report
topic Intrathecal baclofen
Dystonia
Deep brain stimulation
url http://www.sciencedirect.com/science/article/pii/S2214751925000076
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