首例确诊的短弯镰刀菌鼻窦炎在急性髓细胞白血病儿科患者中的报告。
First report of proven Fusarium brachygibbosum sinusitis in a paediatric patient with acute myeloblastic leukaemia.
文献信息
| PMID | 42864586 |
|---|---|
| 原文 | 在 PubMed 查看原文 ↗ |
| 发表日期 | 2026 |
| 作者 | Alfredo Maldonado-Barrueco |
| 作者单位 | Clinical Microbiology and Parasitology Department, Hospital Universitario La Paz, Paseo de La Castellana 261, 28046, Madrid, Spain. alfredo.maldonado@salud.madrid.org. |
| 期刊 | European journal of clinical microbiology & infectious diseases : official publication of the European Society of Clinical Microbiology |
| SCI 分区 | Q2 |
| IF | 3.3 |
| 研究类型 | 临床研究 · 临床 |
| 所属专科 | 鼻科 |
中文摘要
目的： 短弯镰刀菌（Fusarium brachygibbosum）是一种属于茄病镰刀菌复合种（FSAMSC）的环境霉菌，仅极少数报道为人类病原体。侵袭性镰刀菌病是血液系统恶性肿瘤患者公认的并发症；然而，由短弯镰刀菌引起的确诊感染此前尚未被描述。我们报告首例在接受泊沙康唑预防治疗的难治性急性髓细胞白血病（AML）儿科女性患者中发生的突破性鼻窦炎确诊病例。
方法： 我们描述了一名13岁中性粒细胞减少的难治性AML女性患者发生侵袭性真菌性鼻窦炎的临床表现、微生物学诊断、抗真菌药敏谱、治疗和结局。通过延伸因子1-α的Sanger测序确定菌种鉴定，并根据EUCAST参考方法进行抗真菌药敏试验。
结果： 计算机断层扫描显示广泛的鼻旁窦受累，鼻窦组织直接镜检显示透明分隔菌丝。培养产生一种霉菌，最初被MALDI-TOF MS误鉴定为厚壁孢子镰刀菌（F. chlamydosporium）。分子测序确认其为短弯镰刀菌。该分离株对两性霉素B、唑类、棘白菌素类和olorofim表现出升高的最低抑菌浓度（MIC），而fosmanogepix显示出较低的体外MIC。进行了广泛的手术清创，联合脂质体两性霉素B、伏立康唑，随后使用olorofim。尽管鼻窦感染得到局部控制，且无微生物学或组织病理学证据表明播散，患者仍在诊断后23天死亡。死亡发生在难治性AML进展、侵袭性真菌病、肺部异常及随后的中性粒细胞减少并发症的背景下。
结论： 本报告代表了首例由短弯镰刀菌（FSAMSC）分离株引起的侵袭性鼻窦炎儿科确诊病例，该分离株对经典抗真菌药物具有较高的MIC。该病例突出了MALDI-TOF MS对罕见镰刀菌属物种的诊断局限性、分子鉴定和抗真菌药敏试验的价值，以及环境霉菌突破性真菌感染的出现。它还强调了对耐药镰刀菌属物种有效新型抗真菌药物的迫切需求。
英文摘要
PURPOSE: Fusarium brachygibbosum is an environmental mould belonging to the Fusarium sambucinum species complex (FSAMSC) and is only exceptionally reported as a human pathogen. Invasive fusariosis is a recognized complication in patients with hematological malignancies; however, proven infection caused by F. brachygibbosum has not previously been described. We report the first case of proven breakthrough sinusitis in a pediatric female with refractory acute myeloblastic leukaemia (AML) receiving posaconazole prophylaxis.
METHODS: We describe the clinical presentation, microbiological diagnosis, antifungal susceptibility profile, treatment, and outcome of a 13-year-old neutropenic female with refractory AML who developed invasive fungal sinusitis. Species identification was established by Sanger sequencing of the elongation factor 1-alpha, and antifungal susceptibility testing was performed according to the EUCAST reference method.
RESULTS: Computed tomography revealed extensive paranasal sinus involvement, and direct microscopy of sinus tissue demonstrated hyaline septate hyphae. Culture yielded a mould initially misidentified by MALDI-TOF MS as F. chlamydosporium. Molecular sequencing confirmed F. brachygibbosum. The isolate exhibited elevated Minimal inhibitory concentrations (MICs) to amphotericin B, azoles, echinocandins, and olorofim, while fosmanogepix showed a lower in vitro MIC. Extensive surgical debridement combined with liposomal amphotericin B, voriconazole, and subsequently olorofim was performed. Despite local control of the sinonasal infection and no microbiological or histopathological evidence of dissemination, the patient died 23 days after diagnosis. The death occurred in the context of progressive refractory AML, invasive fungal disease, pulmonary abnormalities, and subsequent neutropenic complications.
CONCLUSION: This report represents the first proven pediatric case of invasive sinusitis caused by F. brachygibbosum (FSAMSC) isolate with higher MICs to classical antifungals. The case highlights the diagnostic limitations of MALDI-TOF MS for rare Fusarium species, the value of molecular identification and antifungal susceptibility testing, and the emergence of environmental moulds breakthrough fungal infections. It also underscores the urgent need for effective novel antifungal agents against resistant Fusarium species.