Oliveira Marcos C B, Sato Douglas Kazutoshi, Soares-Neto Herval R, Lucato Leandro T, Callegaro Dagoberto, Nitrini Ricardo, Medeiros Raphael S S, Misu Tatsuro, Fujihara Kazuo, Castro Luiz H
Department of Neurology, Faculdade de Medicina da Universidade de Sao Paulo, Av. Dr. Enéas de Carvalho Aguiar, 255, 5o andar, sala 5011, 05403-900, São Paulo, SP, Brazil.
Departments of Neurology, Tohoku University School of Medicine, 2-1 Seiryo-machi, Aoba-ku, Sendai, Miyagi, 980-8575, Japan.
BMC Neurol. 2015 Sep 2;15:159. doi: 10.1186/s12883-015-0415-0.
Association of leukoencephalopathy and atypical mycobacteriosis has been rarely reported. We present a case that is relevant for its unusual presentation and because it may shed further light on the pathogenic mechanisms underlying reversible encephalopathies.
We report the case of a Hispanic 64-year-old woman with cognitive decline and extensive leukoencephalopathy. Magnetic resonance imaging revealed white-matter lesions with increased water diffusivity, without blood-brain-barrier disruption. Brain biopsy showed tissue rarefaction with vacuolation, mild inflammation, few reactive astrocytes and decreased aquaporin water-channel expression in the lesions. Six months later, she was diagnosed with atypical mycobacterial pulmonary infection. Brain lesions resolved after antimycobacterial treatment.
We hypothesize leukoencephalopathic changes and vasogenic edema were associated with decreased aquaporin expression. Further studies should clarify if reversible leukoencephalopathy has a causal relationship with decreased aquaporin expression and atypical mycobacterial infection, and mechanisms underlying leukoencephalopathy resolution after antimycobacterial treatment. This article may contribute to the understanding of pathogenic mechanisms underlying magnetic resonance imaging subcortical lesions and edema, which remain incompletely understood.
白质脑病与非典型分枝杆菌病的关联鲜有报道。我们报告一例因其不寻常的表现且可能有助于进一步阐明可逆性脑病潜在致病机制的病例。
我们报告一名64岁西班牙裔女性,有认知功能减退及广泛白质脑病。磁共振成像显示白质病变伴水扩散增加,无血脑屏障破坏。脑活检显示病变组织有稀疏空泡形成、轻度炎症、少量反应性星形胶质细胞及水通道蛋白水通道表达降低。6个月后,她被诊断为非典型分枝杆菌肺部感染。抗分枝杆菌治疗后脑病变消失。
我们推测白质脑病改变及血管源性水肿与水通道蛋白表达降低有关。进一步研究应阐明可逆性白质脑病与水通道蛋白表达降低及非典型分枝杆菌感染是否存在因果关系,以及抗分枝杆菌治疗后脑病消失的机制。本文可能有助于理解磁共振成像显示的皮质下病变及水肿的潜在致病机制,目前对此仍了解不完全。