Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report

Abstract Background Patients with tethered cord syndrome often suffer severe spasticity. To the best of our knowledge, intrathecal baclofen (ITB) therapy in a patient with tethered cord syndrome has not been reported previously. We describe a case in which ITB therapy was useful for treating severe...

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Main Authors: Yasutaka Takagi, Hiroshi Yamada, Hidehumi Ebara, Hiroyuki Hayashi, Satoshi Kidani, Kazu Toyooka, Yuji Ishino, Yoshiyuki Kitano, Aki Nakanami, Kenji Kagechika, Tetsutaro Yahata, Hiroyuki Tsuchiya
Format: Article
Language:English
Published: BMC 2021-09-01
Series:Journal of Medical Case Reports
Subjects:
Online Access:https://doi.org/10.1186/s13256-021-03049-0
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author Yasutaka Takagi
Hiroshi Yamada
Hidehumi Ebara
Hiroyuki Hayashi
Satoshi Kidani
Kazu Toyooka
Yuji Ishino
Yoshiyuki Kitano
Aki Nakanami
Kenji Kagechika
Tetsutaro Yahata
Hiroyuki Tsuchiya
author_facet Yasutaka Takagi
Hiroshi Yamada
Hidehumi Ebara
Hiroyuki Hayashi
Satoshi Kidani
Kazu Toyooka
Yuji Ishino
Yoshiyuki Kitano
Aki Nakanami
Kenji Kagechika
Tetsutaro Yahata
Hiroyuki Tsuchiya
author_sort Yasutaka Takagi
collection DOAJ
description Abstract Background Patients with tethered cord syndrome often suffer severe spasticity. To the best of our knowledge, intrathecal baclofen (ITB) therapy in a patient with tethered cord syndrome has not been reported previously. We describe a case in which ITB therapy was useful for treating severe spasticity in an adult with tethered cord syndrome. Case presentation We present the case of a 50-year-old Japanese woman with tethered cord syndrome and related conditions suffering from severe spasticity and pain in the lower limbs. She was born with a lumbosacral myelomeningocele, which was closed in the neonatal period. For 4–5 years before this presentation, spasticity in the lower limbs had been exacerbated without any obvious cause. She received rehabilitation and pharmacotherapy from a local doctor, but symptoms were unimproved, and her previous doctor referred her to this department. A test with 50 μg of intrathecally delivered baclofen showed total relief of spasticity and pain, so a pump was implanted for continuous baclofen delivery. During 24 months of follow-up, spasticity has remained under excellent control with baclofen at 38.5–41.0 μg/day. Conclusions ITB therapy proved extremely effective in this adult with severe spasticity from tethered code syndrome.
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spelling doaj.art-694e8e1346394d9fa29047e57828a27c2022-12-21T22:48:27ZengBMCJournal of Medical Case Reports1752-19472021-09-011511510.1186/s13256-021-03049-0Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case reportYasutaka Takagi0Hiroshi Yamada1Hidehumi Ebara2Hiroyuki Hayashi3Satoshi Kidani4Kazu Toyooka5Yuji Ishino6Yoshiyuki Kitano7Aki Nakanami8Kenji Kagechika9Tetsutaro Yahata10Hiroyuki Tsuchiya11Department of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Orthopaedic Surgery, Tonami General HospitalDepartment of Rehabilitation Medicine, Tonami General HospitalDepartment of Rehabilitation Medicine, Toyama Prefectural Rehabilitation Hospital and Support Center for Children with DisabilitiesDepartment of Rehabilitation Medicine, Kanazawa University HospitalDepartment of Orthopaedic Surgery, Graduate School of Medicine, Kanazawa UniversityAbstract Background Patients with tethered cord syndrome often suffer severe spasticity. To the best of our knowledge, intrathecal baclofen (ITB) therapy in a patient with tethered cord syndrome has not been reported previously. We describe a case in which ITB therapy was useful for treating severe spasticity in an adult with tethered cord syndrome. Case presentation We present the case of a 50-year-old Japanese woman with tethered cord syndrome and related conditions suffering from severe spasticity and pain in the lower limbs. She was born with a lumbosacral myelomeningocele, which was closed in the neonatal period. For 4–5 years before this presentation, spasticity in the lower limbs had been exacerbated without any obvious cause. She received rehabilitation and pharmacotherapy from a local doctor, but symptoms were unimproved, and her previous doctor referred her to this department. A test with 50 μg of intrathecally delivered baclofen showed total relief of spasticity and pain, so a pump was implanted for continuous baclofen delivery. During 24 months of follow-up, spasticity has remained under excellent control with baclofen at 38.5–41.0 μg/day. Conclusions ITB therapy proved extremely effective in this adult with severe spasticity from tethered code syndrome.https://doi.org/10.1186/s13256-021-03049-0Tethered cord syndromeSpasticityBaclofenIntrathecal infusion
spellingShingle Yasutaka Takagi
Hiroshi Yamada
Hidehumi Ebara
Hiroyuki Hayashi
Satoshi Kidani
Kazu Toyooka
Yuji Ishino
Yoshiyuki Kitano
Aki Nakanami
Kenji Kagechika
Tetsutaro Yahata
Hiroyuki Tsuchiya
Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
Journal of Medical Case Reports
Tethered cord syndrome
Spasticity
Baclofen
Intrathecal infusion
title Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
title_full Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
title_fullStr Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
title_full_unstemmed Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
title_short Intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome: a case report
title_sort intrathecal baclofen therapy for severe spasticity in an adult with tethered cord syndrome a case report
topic Tethered cord syndrome
Spasticity
Baclofen
Intrathecal infusion
url https://doi.org/10.1186/s13256-021-03049-0
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