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造血干细胞移植受者中棘阿米巴肉芽肿性阿米巴脑炎的存活病例。

A Surviving Case of Acanthamoeba Granulomatous Amebic Encephalitis in a Hematopoietic Stem Cell Transplant Recipient.

机构信息

Department of Hematology, St. James's Hospital, Dublin, Ireland.

Department of Histopathology, Beaumont Hospital, Dublin, Ireland.

出版信息

Am J Case Rep. 2020 Jun 30;21:e923219. doi: 10.12659/AJCR.923219.

DOI:10.12659/AJCR.923219
PMID:32603318
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7347033/
Abstract

BACKGROUND Acanthamoeba are free-living amoebae with potential to infect immunocompromised hosts. The mortality rate of granulomatous amebic encephalitis (GAE) due to Acanthamoeba exceeds 90% and there are currently no reports of survival of this infection in recipients of hematopoietic stem cell transplant. CASE REPORT We report herein the case of a 32-year-old man presenting to our service with abrupt neurological deterioration and seizures 5 months after allogeneic stem cell transplantation for Hodgkin lymphoma. Clinical and imaging findings were non-specific at presentation. Multiple circumscribed, heterogenous, mass-like lesions were identified on MRI. Brain biopsy was performed and revealed multiple cysts and trophozoites suggesting a diagnosis of granulomatous amebic encephalitis. PCR testing confirmed Acanthamoeba. Treatment with miltefosine, metronidazole, azithromycin, fluconazole, pentamidine isethionate, and co-trimoxazole was instituted and the patient survived and shows continued improvement with intensive rehabilitation. CONCLUSIONS We report the first successful outcome in this setting. The diagnosis would have been missed on cerebrospinal fluid analysis alone, but was rapidly made by histological analysis of brain biopsy. This diagnostically challenging infection is likely under-recognized. Early brain biopsy and commencement of a prolonged miltefosine-containing anti-ameba regimen can be curative.

摘要

背景

棘阿米巴是一种自由生活的阿米巴原虫,有潜在能力感染免疫功能低下的宿主。由棘阿米巴引起的肉芽肿性阿米巴脑炎(GAE)的死亡率超过 90%,目前尚无造血干细胞移植受者存活的报道。

病例报告

我们在此报告一例 32 岁男性,在霍奇金淋巴瘤异基因干细胞移植后 5 个月出现突发性神经恶化和癫痫。临床表现和影像学表现均无特异性。MRI 显示多个界限清楚、异质性、块状病变。脑活检显示多个囊肿和滋养体,提示诊断为肉芽肿性阿米巴脑炎。PCR 检测证实为棘阿米巴。采用米替福新、甲硝唑、阿奇霉素、氟康唑、戊烷脒和复方磺胺甲噁唑进行治疗,患者存活并通过强化康复治疗持续改善。

结论

我们报告了首例成功的治疗结果。如果仅依靠脑脊液分析,可能会漏诊,但通过脑活检的组织学分析,很快就做出了诊断。这种诊断具有挑战性的感染可能被低估了。早期脑活检和开始使用包含米替福新的长期抗阿米巴治疗方案可能是有效的。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e7fc/7347033/e8ec17a70632/amjcaserep-21-e923219-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e7fc/7347033/288d2d6d5ad8/amjcaserep-21-e923219-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e7fc/7347033/e8ec17a70632/amjcaserep-21-e923219-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e7fc/7347033/288d2d6d5ad8/amjcaserep-21-e923219-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e7fc/7347033/e8ec17a70632/amjcaserep-21-e923219-g002.jpg

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