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X 连锁肾上腺脑白质营养不良患儿脐血移植后 EBV 和隐球菌性脑炎的序贯感染。

Sequential infection of Epstein-Barr virus and cryptococcal encephalitis after umbilical cord blood transplantation in a child with X-linked adrenoleukodystrophy.

机构信息

Department of Hematology and Oncology, Shanghai Children's Hospital, Shanghai Jiao Tong University, Shanghai, China.

出版信息

Pediatr Transplant. 2021 Aug;25(5):e13956. doi: 10.1111/petr.13956. Epub 2020 Dec 28.

Abstract

Dual infection with two pathogens can be found in few cases of encephalitis. Cases of sequential infection with EBV and cryptococcal encephalitis in post-transplant patients are rare. We describe a 5-year-old boy with X-linked adrenoleukodystrophy who presented sequential infection with EBV and cryptococcal encephalitis after umbilical cord blood transplant. The patient showed fever, vomiting and emotional agitation with EBV DNA detected in CSF on day 100. The child underwent 3 doses of intravenous rituximab with a good response. However, the child presented with right facial paralysis, headache, and fever on day 130 after 2 weeks of clinical stability. Brain MRI demonstrated chronic granuloma formed with ring enhancement. FilmArray ME PCR confirmed the existence of Cryptococcus neoformans/gattii in the CSF. The child underwent sequential treatment with amphotericin liposome B and flucytosine. Maintenance treatment with fluconazole was administered for 1 year. Facial paralysis was on longer present on day 260. Cryptococcus neoformans/gattii was not detected on day 310. The biochemistry and cell count of the CSF were completely normal on day 520. Follow-up 2.5 years after presentation, brain MRI changes showed near complete resolution of the lesions. The child survived for 3 years to the last following-up. Invasive cryptococcal encephalitis is rare and life-threatening complication of transplantation. It is important to recognize dual infections, and perform treatment quickly to improve the prognosis of encephalitis after transplantation.

摘要

两种病原体的双重感染在少数脑炎病例中可见。移植后患者中 EBV 和隐球菌性脑炎的序贯感染病例罕见。我们描述了一例 X 连锁肾上腺脑白质营养不良患儿,在脐血移植后发生 EBV 和隐球菌性脑炎的序贯感染。患儿在移植后 100 天出现发热、呕吐和情绪激动,CSF 中检测到 EBV DNA。患儿接受了 3 剂静脉注射利妥昔单抗治疗,反应良好。然而,在临床稳定 2 周后,患儿在 130 天出现右侧面瘫、头痛和发热。脑 MRI 显示慢性肉芽肿形成伴环状增强。FilmArray ME PCR 确认 CSF 中存在新型隐球菌/格特隐球菌。患儿先后接受两性霉素 B 脂质体和氟胞嘧啶治疗。给予氟康唑维持治疗 1 年。第 260 天面瘫消失。第 310 天未检测到新型隐球菌/格特隐球菌。第 520 天 CSF 生化和细胞计数完全正常。发病后 2.5 年随访,脑 MRI 改变显示病变几乎完全缓解。患儿存活 3 年,最后一次随访。侵袭性隐球菌性脑炎是移植后罕见且危及生命的并发症。认识到双重感染并迅速进行治疗以改善移植后脑炎的预后非常重要。

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