Bezrodnik L, Samara R, Krasovec S, García Erro M, Sevlever G
Servicio de Immunología, Hospital de Niños Ricardo Gutiérrez, Buenos Aires, Argentina.
Clin Infect Dis. 1998 Jul;27(1):181-4. doi: 10.1086/514617.
We describe a child with congenital hypogammaglobulinemia that was diagnosed at 13 months of age. When he was 4 years old, gait disturbances began. The main neurological manifestations were progressive spastic tetraparesis and intellectual and speech deterioration. No infectious agent was identified. A magnetic resonance imaging scan of the central nervous system revealed periventricular demyelinating areas in the frontal, temporal, and parietal lobes with cortical atrophy. Stereotactic brain biopsy confirmed the diagnosis of progressive multifocal leukoencephalopathy caused by JC virus. He was treated with intravenous and intraventricular cytarabine and interferon-alpha, and there was clinical improvement. We emphasize the need for brain biopsy as soon as a neurological complication is suspected in patients with congenital hypogammaglobulinemia for whom cerebrospinal cultures or polymerase chain reaction analyses are negative.
我们描述了一名13个月大时被诊断为先天性低丙种球蛋白血症的儿童。他4岁时开始出现步态障碍。主要神经学表现为进行性痉挛性四肢轻瘫以及智力和言语退化。未发现感染病原体。中枢神经系统的磁共振成像扫描显示额叶、颞叶和顶叶脑室周围脱髓鞘区域伴皮质萎缩。立体定向脑活检确诊为由JC病毒引起的进行性多灶性白质脑病。他接受了静脉和脑室内阿糖胞苷以及α干扰素治疗,临床症状有所改善。我们强调,对于先天性低丙种球蛋白血症且脑脊液培养或聚合酶链反应分析为阴性、怀疑有神经并发症的患者,一旦怀疑应尽快进行脑活检。