Akash J Patel1, Aliya I Sarwar2, Joseph Jankovic2, Ashwin Viswanathan3. 1. Department of Neurosurgery, Baylor College of Medicine, Houston, Texas, USA. 2. Department of Neurology, Baylor College of Medicine, Houston, Texas, USA. 3. Department of Neurosurgery, Baylor College of Medicine, Houston, Texas, USA. Electronic address: ashwinv@bcm.edu.
Abstract
BACKGROUND: X-linked dystonia-parkinsonism (XDP) is a progressively debilitating movement disorder that begins with focal dystonia and eventually generalizes. It exclusively affects Filipino inhabitants of the island of Panay. We report a case of XDP successfully treated by deep brain stimulation (DBS) and review the literature. METHODS: A 36-year-old man with XDP failed medical management and underwent bilateral globus pallidus internus DBS. A search of the PubMed database was performed to identify all articles discussing DBS and XDP. "Stimulation," "DYT3," "Lubag," "torsion dystonia," and "dystonia-parkinsonism" were used as MeSH headings. RESULTS: The patient's postoperative course was notable for delayed emergence from anesthesia. When stimulation was started, he had immediate improvement in his symptoms, and at 6-month follow-up, he is able to ambulate with the assistance of a walker. Review of the literature revealed 5 previously reported cases of XDP treated with DBS. CONCLUSIONS: The published experience with globus pallidus internus DBS for XDP has been very positive to date. Although long-term follow-up data are needed, early results provide optimism for patients with this debilitating disorder. Published by Elsevier Inc.
BACKGROUND:X-linked dystonia-parkinsonism (XDP) is a progressively debilitating movement disorder that begins with focal dystonia and eventually generalizes. It exclusively affects Filipino inhabitants of the island of Panay. We report a case of XDP successfully treated by deep brain stimulation (DBS) and review the literature. METHODS: A 36-year-old man with XDP failed medical management and underwent bilateral globus pallidus internus DBS. A search of the PubMed database was performed to identify all articles discussing DBS and XDP. "Stimulation," "DYT3," "Lubag," "torsion dystonia," and "dystonia-parkinsonism" were used as MeSH headings. RESULTS: The patient's postoperative course was notable for delayed emergence from anesthesia. When stimulation was started, he had immediate improvement in his symptoms, and at 6-month follow-up, he is able to ambulate with the assistance of a walker. Review of the literature revealed 5 previously reported cases of XDP treated with DBS. CONCLUSIONS: The published experience with globus pallidus internus DBS for XDP has been very positive to date. Although long-term follow-up data are needed, early results provide optimism for patients with this debilitating disorder. Published by Elsevier Inc.
Authors: Norbert Brüggemann; Aloysius Domingo; Dirk Rasche; Christian K E Moll; Raymond L Rosales; Roland Dominic G Jamora; Henrike Hanssen; Alexander Münchau; Julia Graf; Anne Weissbach; Vera Tadic; Cid C Diesta; Jens Volkmann; Andrea Kühn; Thomas F Münte; Volker Tronnier; Christine Klein Journal: JAMA Neurol Date: 2019-02-01 Impact factor: 18.302
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