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免疫功能低下儿童中Tintelnotia destructans侵袭性鼻窦炎:病例报告及文献综述。

Tintelnotia destructans Invasive Rhinosinusitis in an Immunocompromised Child: A Case Report and Review of the Literature.

临床研究鼻科IF 3.5Q2

文献信息

中文摘要

Tintelnotia destructans是Phaeosphaeriaceae科中最近描述的一种真菌,此前仅报道于人类浅表感染,如角膜炎、甲真菌病和非侵袭性窦腔真菌球。我们描述首例由该病原体引起的儿童侵袭性鼻窦感染,患儿患有重型再生障碍性贫血。诊断通过组织病理学显示黏膜下真菌侵袭,以及靶向18S rRNA的广谱真菌聚合酶链反应鉴定出T. destructans而确立。联合内镜下手术清创、抗真菌治疗(脂质体两性霉素B和艾沙康唑)以及紧急单倍体相合造血干细胞移植后的免疫重建,最终使患者在治疗后18个月完全康复且无复发,尽管因移植物抗宿主病仍持续接受治疗性免疫抑制。本病例扩展了T. destructans已知的致病谱,证明其在严重免疫功能低下宿主中引起侵袭性疾病的可能性。分子诊断工具的进步可能通过鉴定新的致病菌种来扩大引起侵袭性真菌感染的真菌谱。

英文摘要

Tintelnotia destructans is a recently described fungus within the family Phaeosphaeriaceae, previously reported only in superficial human infections such as keratitis, onychomycosis, and noninvasive sinus fungus ball. We describe the first case of invasive rhinosinusal infection due to this pathogen in a child with severe aplastic anemia. Diagnosis was established by histopathology showing submucosal fungal invasion and broad-range fungal polymerase chain reaction targeting 18S rRNA identifying T. destructans. Combined endoscopic surgical debridement, antifungal therapy (liposomal amphotericin B and isavuconazole), and immune reconstitution after emergency haploidentical hematopoietic stem cell transplant ultimately led to complete recovery without recurrence 18 months after treatment, despite continued therapeutic immunosuppression for graft-vs-host disease. This case expands the known pathogenic spectrum of T. destructans, demonstrating its potential to cause invasive disease in severely immunocompromised hosts. Advances in molecular diagnostic tools may expand the spectrum of fungi causing invasive fungal infections by identification of novel pathogenic species.