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与黑质纹状体功能障碍相关的偏侧舞蹈症:一例豆状核和内囊同侧梗死患者的病例报告。

Hemichorea Associated With Nigrostriatal Dysfunction: Case Report of a Patient With an Ipsilateral Infarct in the Lenticular Nucleus and Internal Capsule.

作者信息

Kobayashi Makoto

机构信息

Department of Neurology, Asahi General Hospital, 1326 I, Asahi, Chiba 289-2511, Japan.

出版信息

Case Rep Neurol Med. 2025 Jun 17;2025:6054686. doi: 10.1155/crnm/6054686. eCollection 2025.

Abstract

Hemichorea is a rare manifestation of ischemic stroke whose lesion is typically located in the contralateral basal ganglia. Its pathomechanism has not been elucidated completely; however, it may be related to nigrostriatal dysfunction. In patients with hemichorea, dopamine transporter-single photon emission computed tomography (DAT-SPECT) reportedly displayed decreased tracer accumulation in the contralateral striatum. Moreover, in exceptional cases, responsible lesions were located in the ipsilateral cerebral hemisphere. This case report describes an 84-year-old man who presented with three weeks of intermittent, involuntary, and twisting movements in his right limbs. On physical examination, the patient had right-sided hemichorea without other neurological deficits. The choreic movements were more frequent in the lower limb than in the upper and provoked when he tried to take a certain posture or engaged in mental arithmetic. Magnetic resonance imaging performed on suspicion of stroke detected a right hemispheric subacute infarct in the posterior part of the lenticular nucleus and posterior limb of the internal capsule. Furthermore, DAT-SPECT revealed decreased tracer accumulation in the right striatum. He was administered oral antiplatelet medication after being diagnosed with lacunar infarction. The choreic movements gradually reduced over the next 8 months and eventually disappeared. The lesion in the lenticular nucleus and internal capsule was considered to have induced ipsilesional hemichorea, considering the temporal proximity between the hemichorea and ischemic stroke. Although DAT-SPECT findings in patients with ipsilesional hemichorea have not been reported, this case suggests that nigrostriatal dopamine dysfunction can contribute to the pathogenesis of ipsilesional hemichorea.

摘要

偏身舞蹈症是缺血性卒中的一种罕见表现,其病灶通常位于对侧基底神经节。其发病机制尚未完全阐明;然而,它可能与黑质纹状体功能障碍有关。据报道,在偏身舞蹈症患者中,多巴胺转运体单光子发射计算机断层扫描(DAT-SPECT)显示对侧纹状体中示踪剂摄取减少。此外,在特殊情况下,责任病灶位于同侧脑半球。本病例报告描述了一名84岁男性,他右肢出现了三周的间歇性、不自主扭转运动。体格检查时,患者有右侧偏身舞蹈症,无其他神经功能缺损。舞蹈样动作在下肢比在上肢更频繁,当他试图采取某种姿势或进行心算时会诱发。因怀疑中风而进行的磁共振成像检测到右侧半球豆状核后部和内囊后肢有亚急性梗死。此外,DAT-SPECT显示右侧纹状体中示踪剂摄取减少。在被诊断为腔隙性梗死之后,他接受了口服抗血小板药物治疗。在接下来的8个月里,舞蹈样动作逐渐减少,最终消失。考虑到偏身舞蹈症与缺血性卒中在时间上的接近性,豆状核和内囊的病灶被认为诱发了同侧偏身舞蹈症。尽管尚未有同侧偏身舞蹈症患者的DAT-SPECT检查结果的报道,但本病例表明黑质纹状体多巴胺功能障碍可能促成同侧偏身舞蹈症的发病机制。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/54aa/12187432/d50cd55ef2fe/CRINM2025-6054686.001.jpg

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