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

Lingzhi Zhong, Yinggai Song, Jian Hu, Xueyan Yao, Tingting Li, Houmin Li
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引用次数: 1

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.

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急性淋巴细胞白血病患儿播散性两性霉素耐药镰孢菌病的成功治疗:1例报告和文献复习
背景:播散性镰孢菌病是免疫功能低下患者中一种罕见且致命的感染。目的:我们报告一例小儿急性淋巴细胞白血病患者的播散性两性霉素耐药镰孢菌病,并回顾中国报告的播散性镰孢菌病的特点。材料与方法:从中国文献中检索近20年的播散性镰孢病病例报告。结果:该病例为一名15岁的女性,她出现发烧和多个疼痛的紫色斑块,伴有黑色坏死中心和血水泡。血液和皮肤病变中检出镰刀菌,两性霉素B (AMB)最低抑菌浓度高(>32 μg/mL),伏立康唑(VRC)最低抑菌浓度低(0.25 μg/mL)。通过rRNA基因分析,最终鉴定出藤黑镰刀菌属复合体。经4个多月的治疗,VRC联合特比萘芬(TRF)成功治愈了该病。弥散性镰孢病最常见的临床表现为发热、皮损和血培养阳性,共9例(64.3%)。其他部位感染包括肺、眼睛、鼻窦或骨髓8例(57.1%)。7例(50%)患者经AMB和VRC单药或联合治疗后痊愈。结论:结合本病例和文献回顾,早期发现镰孢菌感染并使用合适的抗真菌药物是成功治疗的关键。主要治疗应包括VRC或两性霉素B脂质体(L-AMB),补救性治疗包括泊沙康唑(PSC)。抗真菌药物的组合可能是必要的和更有效的。
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