Division of Infectious Diseases, Boston Children's Hospital, Boston, MA, USA.
Division of Gastroenterology, Hepatology and Nutrition, Boston Children's Hospital, Boston, MA, USA.
Transpl Infect Dis. 2020 Aug;22(4):e13304. doi: 10.1111/tid.13304. Epub 2020 May 19.
A 20-year-old male presented 3.5 years after intestinal transplantation with rapidly progressive sensorineural hearing loss. Initial brain imaging was consistent with inflammation and/or demyelination. Lumbar puncture was initially non-diagnostic and a broad infectious workup was unrevealing. Three months after presentation, a repeat LP detected JC virus for which tests had not earlier been conducted. He continued to deteriorate despite withdrawal of prior immunosuppression and addition of mirtazapine, maraviroc, and steroids. He died of progressive neurologic decompensation 5 months after his initial presentation. This case highlights progressive multifocal leukoencephalopathy (PML) as a rare complication after solid organ transplantation and acute sensorineural hearing loss as an unusual first presenting symptom of PML. JC virus should be considered in the differential diagnosis of acute sensorineural hearing loss in any immunocompromised patient.
一名 20 岁男性在肠移植后 3.5 年出现进行性感觉神经性听力损失。最初的脑部成像符合炎症和/或脱髓鞘。腰椎穿刺最初没有明确诊断,广泛的感染性检查也没有发现任何结果。在出现症状 3 个月后,再次进行腰椎穿刺检测到了 JC 病毒,而之前并未进行过此类检测。尽管先前的免疫抑制药物已被停用,并且添加了米氮平、马拉维若和类固醇,但他仍持续恶化。在初次出现症状 5 个月后,他因进行性神经功能恶化而死亡。本病例强调了进行性多灶性白质脑病(PML)是实体器官移植后的罕见并发症,以及急性感觉神经性听力损失是 PML 的不常见首发症状。在任何免疫功能低下的患者中,都应考虑将 JC 病毒纳入急性感觉神经性听力损失的鉴别诊断中。