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儿童急性淋巴细胞白血病合并播散性两性霉素 B 耐药镰刀菌病的成功治疗:病例报告及文献复习。

Successful treatment of disseminated amphotericin-resistant fusariosis in a paediatric patient with acute lymphoblastic leukaemia: a case report and literature review.

机构信息

Department of Dermatology, Peking University People’s Hospital, Beijing 100044, China

Department of Dermatology, Peking University First Hospital, Beijing 100034, China

出版信息

Eur J Dermatol. 2022 Sep 1;32(5):623-628. doi: 10.1684/ejd.2022.4319.

DOI:10.1684/ejd.2022.4319
PMID:36468720
Abstract

BACKGROUND

Disseminated fusariosis is a rare and fatal infection in immunocompromised patients.

OBJECTIVES

We report a case of disseminated amphotericin-resistant fusariosis in a paediatric patient with acute lymphoblastic leukaemia and review the features of reported disseminated fusariosis in China.

MATERIALS & METHODS: Case reports of disseminated fusariosis were searched from the Chinese literature over the last two decades.

RESULTS

The presented case is a 15-year-old female who developed fever and multiple painful purple plaques with black necrotic centres and blood blisters. Fusarium was detected in blood and skin lesions with a high minimum inhibitory concentration (MIC) of amphotericin B (AMB) (>32 μg/mL) and a low MIC of voriconazole (VRC) (0.25 μg/mL). The Fusarium fujikuroi species complex was finally identified by rRNA gene analysis. Combination therapy of VRC and terbinafine (TRF) successfully resolved the disease after more than four months of treatment. Based on the review, the most common manifestations of disseminated fusariosis were fever, skin lesions and positive blood cultures, comprising nine cases (64.3%). Other sites of infection, including the lungs, eyes, sinuses or bone marrow, occurred in eight cases (57.1%). Seven patients (50%) were cured after monotherapy or combination therapy with AMB and VRC.

CONCLUSION

In view of this case and the review of the literature, early identification of Fusarium infection and the appropriate antifungal drugs are critical for successful treatment. Primary therapy should consist of VRC or liposomal amphotericin B (L-AMB), with salvage therapy consisting of posaconazole (PSC). The combination of antifungals is probably necessary and more effective.

摘要

背景

播散性镰孢菌病是免疫功能低下患者中罕见且致命的感染。

目的

我们报告一例儿童急性淋巴细胞白血病患者播散性耐两性霉素 B 镰孢菌病,并回顾中国播散性镰孢菌病的特征。

材料与方法

在过去二十年中,从中文文献中搜索了播散性镰孢菌病的病例报告。

结果

所报告的病例为 15 岁女性,出现发热和多个疼痛性紫色斑块,伴有黑色坏死中心和血疱。在血液和皮肤病变中检测到镰孢菌,其两性霉素 B(AMB)最小抑菌浓度(MIC)较高(>32μg/mL),伏立康唑(VRC)MIC 较低(0.25μg/mL)。rRNA 基因分析最终鉴定为镰孢菌藤仓复合体。VRC 和特比萘芬(TRF)联合治疗在超过四个月的治疗后成功治愈疾病。基于文献回顾,播散性镰孢菌病最常见的表现为发热、皮肤病变和血液培养阳性,共 9 例(64.3%)。其他感染部位,包括肺部、眼睛、鼻窦或骨髓,发生在 8 例(57.1%)。7 例(50%)患者在接受 AMB 和 VRC 单药或联合治疗后治愈。

结论

鉴于该病例和文献回顾,早期识别镰孢菌感染和使用适当的抗真菌药物对于成功治疗至关重要。一线治疗应包含 VRC 或脂质体两性霉素 B(L-AMB),挽救治疗包含泊沙康唑(PSC)。联合使用抗真菌药物可能是必要且更有效的。

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