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免疫功能正常患者播散性毕赤酵母菌感染的尸检病例:病例报告。

An autopsy case of disseminated Cunninghamella bertholletiae infection in an immunocompetent patient: a case report.

机构信息

Department of Respiratory Medicine, Fujieda Municipal General Hospital, Fujieda, Japan.

Department of Respiratory Medicine, Iwata City Hospital, Okubo, Iwata, Shizuoka, 512-3426-8677, Japan.

出版信息

BMC Pulm Med. 2023 Mar 17;23(1):88. doi: 10.1186/s12890-023-02382-y.

Abstract

BACKGROUND

Recently, deaths due to mucormycosis in immunocompromised hosts have increased; however, the clinical and pathological features of mucormycosis are not fully understood, especially in view of the associated high mortality and rare incidence in immunocompetent patients.

CASE PRESENTATION

We have described a rare autopsy case of a 67-year-old Japanese man with chronic obstructive pulmonary disease who contracted mucormycosis. He had not been on any immunosuppressants, and his immune functions were intact. Since 3 days prior to admission to our hospital, he had experienced progressive dyspnea, productive cough, and fever. Chest computed tomography revealed pleural effusion in the left lower hemithorax and consolidation in the right lung field. Although he was administered with tazobactam-piperacillin hydrate (13.5 g/day), renal dysfunction occurred on the ninth disease day. Therefore, it was switched to cefepime (2 g/day). However, his general condition and lung-field abnormality worsened gradually. Cytological analysis of the sputum sample at admission mainly revealed sporangiophores and unicellular sporangioles, while repeated sputum culture yielded Cunninghamella species. Therefore, he was diagnosed with pulmonary mucormycosis. Liposomal amphotericin B (5 mg/kg/day) was initiated on the 28 disease day. However, chest radiography and electrocardiography detected cardiomegaly and atrial fibrillation, respectively, and he died on the 37 disease day. A postmortem examination revealed clusters of fungal hyphae within the arteries of the right pulmonary cavity wall, the subpericardial artery, intramyocardial capillary blood vessels, and the esophageal subserosa vein. Direct sequencing revealed that all fungal culture samples were positive for Cunninghamella bertholletiae.

CONCLUSIONS

Cunninghamella bertholletiae could rapidly progress from colonizing the bronchi to infecting the surrounding organs via vascular invasion even in immunocompetent patients.

摘要

背景

最近,免疫功能低下宿主的毛霉菌病死亡人数有所增加;然而,毛霉菌病的临床和病理特征尚未完全了解,特别是考虑到其相关的高死亡率和免疫功能正常患者的罕见发病率。

病例介绍

我们描述了一例罕见的尸检病例,一名 67 岁的日本男性患有慢性阻塞性肺疾病,感染了毛霉菌病。他没有服用任何免疫抑制剂,免疫功能完好。从他入院前 3 天开始,他出现了进行性呼吸困难、咳痰和发热。胸部计算机断层扫描显示左胸腔下部胸腔积液和右肺野实变。尽管他接受了他唑巴坦-哌拉西林三水合物(13.5g/天)治疗,但在第 9 天出现肾功能障碍。因此,将其转换为头孢吡肟(2g/天)。然而,他的一般状况和肺部异常逐渐恶化。入院时痰液样本的细胞学分析主要显示孢子梗和单细胞孢子囊,而反复的痰培养均培养出 Cunninghamella 种。因此,他被诊断为肺毛霉菌病。入院第 28 天开始使用脂质体两性霉素 B(5mg/kg/天)。然而,胸部 X 线摄影和心电图分别检测到心脏扩大和心房颤动,他于入院第 37 天死亡。尸检显示右肺腔壁动脉、心包下动脉、心肌毛细血管血管和食管下膜静脉内有一簇簇真菌菌丝。直接测序显示,所有真菌培养样本均为 Cunninghamella bertholletiae 阳性。

结论

即使在免疫功能正常的患者中,Cunninghamella bertholletiae 也可以通过血管侵袭迅速从定植于支气管进展为感染周围器官。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8d9d/10022292/62c2778c5a70/12890_2023_2382_Fig1_HTML.jpg

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