Davico Chiara, Canavese Carlotta, Tocchet Aba, Brusa Chiara, Vitiello Benedetto
Division of Child and Adolescent Neuropsychiatry, University of Turin, Turin, Italy.
Front Neurol. 2018 Mar 20;9:164. doi: 10.3389/fneur.2018.00164. eCollection 2018.
Acute hemichorea can occur in the context of infectious, autoimmune, metabolic, toxic, and vascular neuropathologies. Primary infection by varicella zoster virus (VZV) can result in vasculopathy with neurological manifestations, such as hemiparesis, at times accompanied by hemichorea. Isolated hemichorea, however, had not been reported. We here describe two cases of VZV-induced vasculopathy whose sole clinical manifestation was acute hemichorea. Both cases involved young boys of 3 years of age, who presented with acute hemichorea 4-6 months after initial VZV infection. All hematological, immunological, and toxicological tests were normal, except for the presence of VZV IgG. Brain structural magnetic resonance imaging (MRI) and magnetic resonance angiography revealed specific signs of vasculitis and ischemic lesions in the basal ganglia region (lentiform nucleus, thalamus, and internal capsule). Following corticosteroid and acetylsalicylic acid treatment, full symptomatic recovery was achieved within 3 weeks. Repeated MRI documented full neurostructural recovery, which was confirmed at extended follow-up for more than 1 year. These cases indicate that VZV-induced vasculopathy should be considered in the case of pediatric isolated acute hemichorea.
急性偏侧舞蹈症可发生于感染性、自身免疫性、代谢性、中毒性和血管性神经病变的情况下。水痘带状疱疹病毒（VZV）的原发性感染可导致伴有神经学表现（如偏瘫）的血管病变，有时还伴有急性偏侧舞蹈症。然而，孤立性急性偏侧舞蹈症此前尚未见报道。我们在此描述两例VZV诱导的血管病变，其唯一的临床表现为急性偏侧舞蹈症。两例均为3岁男童，在初次感染VZV后4 - 6个月出现急性偏侧舞蹈症。除VZV IgG阳性外，所有血液学、免疫学和毒理学检查均正常。脑部结构磁共振成像（MRI）和磁共振血管造影显示基底节区（豆状核、丘脑和内囊）有血管炎和缺血性病变的特异性征象。经皮质类固醇和乙酰水杨酸治疗后，3周内症状完全恢复。重复MRI显示神经结构完全恢复，在延长至1年以上的随访中得到证实。这些病例表明，小儿孤立性急性偏侧舞蹈症应考虑VZV诱导的血管病变。
