Cutaneous fusariosis caused by Fusarium lichenicola in a child with hyper-immunoglobulin E syndrome
Cutaneous fusariosis caused by Fusarium lichenicola in a child with hyper-immunoglobulin E syndrome
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高免疫球蛋白 E 综合征儿童由地衣镰刀菌引起的皮肤镰刀菌病
DOI：
10.1111/1346-8138.15168
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发表时间：
2019-12-12
影响因子：
3.1
通讯作者：
Zhang, Junmin
中科院分区：
文献类型：
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作者：
Shi, Minglan;Lu, Sha;Zhang, Junmin
镰刀菌病是仅次于曲霉菌病的第二常见霉菌感染，而真菌性角膜炎是最常见的植入性感染。在此，我们报告一个4岁的汉族女孩，在她的右脸上出现了一个瘙痒的肿块，持续了将近2年。直接涂片标本真菌菌丝阳性。皮损活检显示表皮和真皮内有大量真菌菌丝。根据TEF-1 α基因的序列分析和系统发育分析，确定该病原菌为地衣镰刀菌（Fusarium lichenicola）。使用她的外周血进行的全外显子组测序分析揭示了STAT 3基因的杂合突变，该突变与常染色体显性高免疫球蛋白E综合征（AD-HIES）相关。经静脉注射和病灶内注射阿替霉素B、口服伏立康唑和外用卢立康唑乳膏治疗后，病变改善。据我们所知，这是第二个报告的情况下，一个特殊的局部皮肤病变所造成的镰刀菌属的儿童与AD-HIES。这两种情况表明，STAT 3缺乏可能会增加对镰刀菌病的易感性。
Fusariosis is the second most common mold infection after aspergillosis, and keratomycosis is the most encountered implantation infection. Here, we report a case of a 4-year-old Han Chinese girl presenting with an itchy mass on her right face of almost 2 years' duration. Direct smear of the lesion sample was positive for fungal hyphae. Biopsy of the lesion showed many fungal hyphae in the epidermis and dermis. The pathogen was identified as Fusarium lichenicola by molecular sequencing and phylogenetic analysis based on the TEF-1 alpha gene. Whole-exome sequencing analysis using her peripheral blood revealed a heterozygous mutation in the STAT3 gene, which is related to autosomal dominant hyper-immunoglobulin E syndrome (AD-HIES). The lesion improved following treatment with i.v. and intralesional amphotericin B, oral voriconazole and topical luliconazole cream. To our knowledge, this is the second reported case of a special localized cutaneous lesion caused by Fusarium species in a child with AD-HIES. Both cases suggest that STAT3 deficiency may increase susceptibility to fusariosis.