一例无明显免疫抑制的年轻成人由穗状弯孢霉引起的鼻颅感染：病例报告及文献综述

文献信息

中文摘要

背景： 弯孢霉属是广泛分布于土壤、植物和腐烂有机物中的黑色素化暗色真菌。人类感染并不常见，通常累及鼻旁窦、皮肤、角膜或呼吸道，而中枢神经系统（CNS）受累罕见。
方法： 我们报告一例由穗状弯孢霉引起的局部侵袭性鼻颅感染，患者为一名20岁免疫功能正常的男性，患有慢性鼻窦炎，并具有符合变应性真菌性鼻窦炎（AFRS）的特征。脑磁共振成像显示急性加重于慢性鼻窦炎，并并发额窦黏液囊肿伴眶内和颅内延伸。处理包括紧急神经外科感染源控制，随后进行内镜下鼻窦手术。从独立的深部手术标本中反复分离出穗状弯孢霉。通过整合形态学、蛋白质组学（MALDI-TOF MS）和分子证据的多相方法，将该分离株鉴定为穗状弯孢霉。组织病理学检查显示混合性中性粒细胞和嗜酸性粒细胞炎症，符合AFRS。开始抗真菌治疗，使用脂质体两性霉素B联合伏立康唑；随后因严重低钾血症停用两性霉素B，而伏立康唑在治疗药物监测下继续使用。
结果： 截至撰写本文时，患者已完成三个月的伏立康唑治疗，临床逐步改善且耐受性良好，并正在等待进一步的影像学随访以指导治疗疗程的决定。对已发表病例的回顾表明，慢性鼻窦疾病的连续传播是CNS弯孢霉感染的常见致病机制，并且即使没有明显的免疫抑制，也可能发生严重疾病。
结论： 本病例强调了早期多学科管理、积极的感染源手术控制、准确的微生物学鉴定以及延长具有抗霉菌活性的抗真菌治疗并仔细监测治疗相关毒性的重要性。

英文摘要

Curvularia species are melanized dematiaceous molds widely distributed in soil, plants, and decaying organic matter. Human infections are uncommon and typically involve the paranasal sinuses, skin, cornea, or respiratory tract, whereas central nervous system (CNS) involvement is rare. We report a locally invasive sino-cranial infection caused by Curvularia spicifera in a 20-year-old immunocompetent man with chronic sinusitis and features consistent with allergic fungal rhinosinusitis (AFRS). Brain magnetic resonance imaging revealed acute-on-chronic sinusitis complicated by a frontal sinus mucocele with orbital and intracranial extension. Management involved urgent neurosurgical source control followed by endoscopic sinonasal surgery. C. spicifera was repeatedly isolated from independent deep surgical specimens. The isolate was identified as Curvularia spicifera using a polyphasic approach integrating morphological, proteomic (MALDI-TOF MS), and molecular evidence. Histopathological examination demonstrated mixed neutrophilic and eosinophilic inflammation consistent with AFRS. Antifungal treatment was initiated with liposomal amphotericin B plus voriconazole; amphotericin B was subsequently discontinued because of severe hypokalemia, whereas voriconazole was continued under therapeutic drug monitoring. At the time of writing, the patient had completed three months of voriconazole therapy, with progressive clinical improvement and good tolerability, and was awaiting further radiological follow-up to guide the decision on treatment duration. Review of published cases indicates that contiguous spread from chronic sinonasal disease represents a frequent pathogenic mechanism of CNS Curvularia infection and that severe disease may occur even in the absence of overt immunosuppression. This case highlights the importance of early multidisciplinary management, aggressive surgical source control, accurate microbiological identification, and prolonged mold-active antifungal therapy with careful monitoring for treatment-related toxicity.