Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy

Background: Paroxysmal kinesigenic dyskinesia (PKD) is a movement disorder characterized by transient dyskinetic movements, including dystonia, chorea, or both, triggered by sudden voluntary movements. Carbamazepine and other antiepileptic drugs (AEDs) are widely used in the treatment of PKD, and th...

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Autores principales:	Masato Murakami, Shiro Horisawa, Kenko Azuma, Hiroyuki Akagawa, Taku Nonaka, Takakazu Kawamata, Takaomi Taira
Formato:	article
Lenguaje:	EN
Publicado:	Frontiers Media S.A. 2021
Materias:	paroxysmal kinesigenic dyskinesia dystonia antiepileptic drugs remission ventro-oral thalamotomy Neurology. Diseases of the nervous system RC346-429
Acceso en línea:	https://doaj.org/article/f311e8f11dc74d2e8ee25d327bace4c7
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spelling	oai:doaj.org-article:f311e8f11dc74d2e8ee25d327bace4c72021-12-03T05:27:31ZCase Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy1664-229510.3389/fneur.2021.789468https://doaj.org/article/f311e8f11dc74d2e8ee25d327bace4c72021-12-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fneur.2021.789468/fullhttps://doaj.org/toc/1664-2295Background: Paroxysmal kinesigenic dyskinesia (PKD) is a movement disorder characterized by transient dyskinetic movements, including dystonia, chorea, or both, triggered by sudden voluntary movements. Carbamazepine and other antiepileptic drugs (AEDs) are widely used in the treatment of PKD, and they provide complete remission in 80–90% of medically treated patients. However, the adverse effects of AEDs include drowsiness and dizziness, which interfere with patients' daily lives. For those with poor compatibility with AEDs, other treatment approaches are warranted.Case Report: A 19-year-old man presented to our institute with right hand and foot dyskinesia. He had a significant family history of PKD; his uncle, grandfather, and grandfather's brother had PKD. The patient first experienced paroxysmal involuntary left hand and toe flexion with left forearm pronation triggered by sudden voluntary movements at the age of 14. Carbamazepine (100 mg/day) was prescribed, which led to a significant reduction in the frequency of attacks. However, carbamazepine induced drowsiness, which significantly interfered with his daily life, especially school life. He underwent right-sided ventro-oral (Vo) thalamotomy at the age of 15, which resulted in complete resolution of PKD attacks immediately after the surgery. Four months after the thalamotomy, he developed right elbow, hand, and toe flexion. He underwent left-sided Vo thalamotomy at the age of 19. Immediately after the surgery, the PKD attacks resolved completely. However, mild dysarthria developed, which spontaneously resolved within three months. Left-sided PKD attacks never developed six years after the right Vo thalamotomy, and right-sided PKD attacks never developed two years after the left Vo thalamotomy without medication.Conclusion: The present case showed long-term suppression of bilateral PKDs after bilateral thalamotomy, which led to drug-free conditions.Masato MurakamiShiro HorisawaKenko AzumaHiroyuki AkagawaTaku NonakaTakakazu KawamataTakaomi TairaFrontiers Media S.A.articleparoxysmal kinesigenic dyskinesiadystoniaantiepileptic drugsremissionventro-oral thalamotomyNeurology. Diseases of the nervous systemRC346-429ENFrontiers in Neurology, Vol 12 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	paroxysmal kinesigenic dyskinesia dystonia antiepileptic drugs remission ventro-oral thalamotomy Neurology. Diseases of the nervous system RC346-429
spellingShingle	paroxysmal kinesigenic dyskinesia dystonia antiepileptic drugs remission ventro-oral thalamotomy Neurology. Diseases of the nervous system RC346-429 Masato Murakami Shiro Horisawa Kenko Azuma Hiroyuki Akagawa Taku Nonaka Takakazu Kawamata Takaomi Taira Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
description	Background: Paroxysmal kinesigenic dyskinesia (PKD) is a movement disorder characterized by transient dyskinetic movements, including dystonia, chorea, or both, triggered by sudden voluntary movements. Carbamazepine and other antiepileptic drugs (AEDs) are widely used in the treatment of PKD, and they provide complete remission in 80–90% of medically treated patients. However, the adverse effects of AEDs include drowsiness and dizziness, which interfere with patients' daily lives. For those with poor compatibility with AEDs, other treatment approaches are warranted.Case Report: A 19-year-old man presented to our institute with right hand and foot dyskinesia. He had a significant family history of PKD; his uncle, grandfather, and grandfather's brother had PKD. The patient first experienced paroxysmal involuntary left hand and toe flexion with left forearm pronation triggered by sudden voluntary movements at the age of 14. Carbamazepine (100 mg/day) was prescribed, which led to a significant reduction in the frequency of attacks. However, carbamazepine induced drowsiness, which significantly interfered with his daily life, especially school life. He underwent right-sided ventro-oral (Vo) thalamotomy at the age of 15, which resulted in complete resolution of PKD attacks immediately after the surgery. Four months after the thalamotomy, he developed right elbow, hand, and toe flexion. He underwent left-sided Vo thalamotomy at the age of 19. Immediately after the surgery, the PKD attacks resolved completely. However, mild dysarthria developed, which spontaneously resolved within three months. Left-sided PKD attacks never developed six years after the right Vo thalamotomy, and right-sided PKD attacks never developed two years after the left Vo thalamotomy without medication.Conclusion: The present case showed long-term suppression of bilateral PKDs after bilateral thalamotomy, which led to drug-free conditions.
format	article
author	Masato Murakami Shiro Horisawa Kenko Azuma Hiroyuki Akagawa Taku Nonaka Takakazu Kawamata Takaomi Taira
author_facet	Masato Murakami Shiro Horisawa Kenko Azuma Hiroyuki Akagawa Taku Nonaka Takakazu Kawamata Takaomi Taira
author_sort	Masato Murakami
title	Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
title_short	Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
title_full	Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
title_fullStr	Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
title_full_unstemmed	Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy
title_sort	case report: long-term suppression of paroxysmal kinesigenic dyskinesia after bilateral thalamotomy
publisher	Frontiers Media S.A.
publishDate	2021
url	https://doaj.org/article/f311e8f11dc74d2e8ee25d327bace4c7
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Case Report: Long-Term Suppression of Paroxysmal Kinesigenic Dyskinesia After Bilateral Thalamotomy

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