Staged bilateral pallidotomy for dystonic camptocormia: case report

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Camptocormia is a rare, involuntary movement disorder, presenting as truncal flexion while standing or walking, and is mainly observed as a feature of Parkinson’s disease (PD) and primary dystonia. Deep brain stimulation (DBS) of the globus pallidus internus is effective for refractory camptocormia observed with PD or dystonia. However, the effectiveness of pallidotomy for camptocormia has not been investigated. The authors report the case of a 38-year-old man with anterior truncal bending that developed when he was 36 years old. Prior to the onset of the symptom, he had been taking antipsychotic drugs for schizophrenia. There were no features of PD; the symptom severely interfered with his walking and daily life. He was given anticholinergics, clonazepam, and botulinum toxin injections, which did not result in much success. Because of the patient’s unwillingness to undergo implantation of a hardware device, he underwent staged bilateral pallidotomy with complete resolution for a diagnosis of tardive dystonic camptocormia. The Burke-Fahn-Marsden dystonia rating scale subscore for the trunk before and after bilateral pallidotomy was 3 and 0, respectively. No perioperative adverse events were observed. Effects have persisted for 18 months. Bilateral pallidotomy can be a treatment option for medically refractory dystonic camptocormia without the need for device implantation.

ABBREVIATIONS AC-PC = anterior commissure–posterior commissure; BFMDRS = Burke-Fahn-Marsden dystonia rating scale; DBS = deep brain stimulation; ECT = electroconvulsive therapy; GPi = globus pallidus internus; PD = Parkinson’s disease; STN = subthalamic nucleus.

Article Information

Correspondence Takaomi Taira: Neurological Institute, Tokyo Women’s Medical University, Tokyo, Japan. ttaira@twmu.ac.jp.

INCLUDE WHEN CITING Published online October 19, 2018; DOI: 10.3171/2018.5.JNS1840.

Disclosures Takaomi Taira received consulting fees from St. Jude Japan and a speaking fee from Daiichi-Sankyo.

© AANS, except where prohibited by US copyright law.

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Figures

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    Time course of the neurological condition. A: Preoperatively, anterior supine bending was severe. B: After left pallidotomy, camptocormia became mild but was still significant. C: Three days after bilateral pallidotomy, the camptocormia dramatically improved. Figure is available in color online only.

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    Coronal MR image obtained 3 months after right pallidotomy, showing lesions in the left (arrowhead) and right (arrow) GPi.

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