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


Journal

Journal of medical case reports
ISSN: 1752-1947
Titre abrégé: J Med Case Rep
Pays: England
ID NLM: 101293382

Informations de publication

Date de publication:
02 Sep 2021
Historique:
received: 19 07 2020
accepted: 05 08 2021
entrez: 2 9 2021
pubmed: 3 9 2021
medline: 4 9 2021
Statut: epublish

Résumé

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. 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. ITB therapy proved extremely effective in this adult with severe spasticity from tethered code syndrome.

Sections du résumé

BACKGROUND 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 METHODS
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 CONCLUSIONS
ITB therapy proved extremely effective in this adult with severe spasticity from tethered code syndrome.

Identifiants

pubmed: 34470662
doi: 10.1186/s13256-021-03049-0
pii: 10.1186/s13256-021-03049-0
pmc: PMC8411508
doi:

Substances chimiques

Muscle Relaxants, Central 0
Baclofen H789N3FKE8

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

442

Informations de copyright

© 2021. The Author(s).

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Auteurs

Yasutaka Takagi (Y)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan. takagi@p1.coralnet.or.jp.

Hiroshi Yamada (H)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Hidehumi Ebara (H)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Hiroyuki Hayashi (H)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Satoshi Kidani (S)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Kazu Toyooka (K)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Yuji Ishino (Y)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Yoshiyuki Kitano (Y)

Department of Orthopaedic Surgery, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Aki Nakanami (A)

Department of Rehabilitation Medicine, Tonami General Hospital, 1-61 Shintomi-cho, Toyama, 939-1395, Japan.

Kenji Kagechika (K)

Department of Rehabilitation Medicine, Toyama Prefectural Rehabilitation Hospital and Support Center for Children with Disabilities, 36 Shimoiino-machi, Toyama, 939-1395, Japan.

Tetsutaro Yahata (T)

Department of Rehabilitation Medicine, Kanazawa University Hospital, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8641, Japan.

Hiroyuki Tsuchiya (H)

Department of Orthopaedic Surgery, Graduate School of Medicine, Kanazawa University, 13-1 Takara-machi, Kanazawa, Ishikawa, 920-8641, Japan.

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Classifications MeSH