open access

Vol 45, No 5 (2011)
OPIS PRZYPADKU
Submitted: 2011-01-16
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Successful bilateral pallidal stimulation in a patient with isolated lower limb dystonia coexistent with Langerhans cell histiocytosis and coeliac disease

Michał Sobstyl, Mirosław Ząbek, Sebastian Dzierzęcki, Zbigniew Mossakowski, Krzysztof Szczałuba
DOI: 10.1016/S0028-3843(14)60322-4
·
Neurol Neurochir Pol 2011;45(5):514-519.

open access

Vol 45, No 5 (2011)
OPIS PRZYPADKU
Submitted: 2011-01-16

Abstract

The authors report a case of bilateral globus pallidus internus (GPi) stimulation for treatment of medically intractable isolated lower limb dystonia. The 14-year-old girl developed dystonic movements in her left lower limb. At the age of 17, the patient was handicapped by dystonic movements in her lower limbs, and became wheelchair-bound. Pharmacological therapy and botulinum toxin injection resulted in transient and modest benefit. Moreover, the patient was diagnosed with histologically proven coeliac disease and Langerhans cell histiocytosis. Genetic testing revealed the presence of DYT-1 mutation. The 17-year-old girl underwent bilateral implantation of deep brain stimulation leads. Bilateral GPi stimulation resulted in remarkable improvement of phasic dystonic movements, and dystonic posture of lower limbs. Over 2 years postoperative follow-up, the patient is able to walk independently. Bilateral GPi stimulation appears to be an effective treatment modality for isolated lower limb dystonia.

Abstract

The authors report a case of bilateral globus pallidus internus (GPi) stimulation for treatment of medically intractable isolated lower limb dystonia. The 14-year-old girl developed dystonic movements in her left lower limb. At the age of 17, the patient was handicapped by dystonic movements in her lower limbs, and became wheelchair-bound. Pharmacological therapy and botulinum toxin injection resulted in transient and modest benefit. Moreover, the patient was diagnosed with histologically proven coeliac disease and Langerhans cell histiocytosis. Genetic testing revealed the presence of DYT-1 mutation. The 17-year-old girl underwent bilateral implantation of deep brain stimulation leads. Bilateral GPi stimulation resulted in remarkable improvement of phasic dystonic movements, and dystonic posture of lower limbs. Over 2 years postoperative follow-up, the patient is able to walk independently. Bilateral GPi stimulation appears to be an effective treatment modality for isolated lower limb dystonia.

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Keywords

movement disorders, lower limb dystonia, pallidal stimulation, functional neurosurgery

About this article
Title

Successful bilateral pallidal stimulation in a patient with isolated lower limb dystonia coexistent with Langerhans cell histiocytosis and coeliac disease

Journal

Neurologia i Neurochirurgia Polska

Issue

Vol 45, No 5 (2011)

Pages

514-519

DOI

10.1016/S0028-3843(14)60322-4

Bibliographic record

Neurol Neurochir Pol 2011;45(5):514-519.

Keywords

movement disorders
lower limb dystonia
pallidal stimulation
functional neurosurgery

Authors

Michał Sobstyl
Mirosław Ząbek
Sebastian Dzierzęcki
Zbigniew Mossakowski
Krzysztof Szczałuba

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