成人灰色小克银汉霉侵袭性毛霉菌病1例并文献复习

Adult invasive mucormycosis caused by Cunninghamella bertholletiae:one case report and literature review

目的 探讨灰色小克银汉霉感染导致成人毛霉菌病患者的临床特点及诊治方案。方法 总结某院血液内科收治的1例灰色小克银汉霉感染致成人侵袭性毛霉菌病患者的临床诊疗过程,并检索数据库相关文献进行复习。结果 患者男性,54岁,因“反复乏力1年余,加重伴发热1周”入院,肺组织病理检查可见宽大、不规则、无分隔的菌丝,形态学鉴定为毛霉菌,肺泡灌洗液及外周血宏基因组二代测序(mNGS)检测示灰色小克银汉霉,诊断为灰色小克银汉霉感染致侵袭性毛霉菌病,给予脂质体两性霉素B联合泊沙康唑、卡泊芬净治疗后感染获得控制。检索出符合条件的文献37篇,加上本病例,共纳入44例患者,其中男性26例,女性18例,中位年龄52.5(18~79)岁;基础疾病主要为血液系统疾病(65.9%,29例),进行造血干细胞或实体器官移植者14例;最常见的侵犯部位为肺、脑及皮肤,分别为36、9、9例;组织病理学、真菌培养、直接镜检及分子学检测阳性者分别为28、37、29、17例。41例患者接受了抗真菌治疗,其中8例联合手术治疗;30例死亡,病死率为68.2%,抗真菌治疗联合手术者生存率(62.5%,5/8)高于单独抗真菌治疗者(24.2%,7/33)。结论 灰色小克银汉霉感染所致成人毛霉菌病在血液系统疾病患者中最常见,造血干细胞或实体脏器移植为高危因素,常见感染部位为肺部,并可侵犯全身多脏器,病死率高,病原学诊断十分重要,抗真菌联合手术治疗可提高生存率。

Objective To explore the clinical characteristics as well as diagnosis and treatment scheme of an adult patient with mucormycosis caused by Cunninghamella bertholletiae(C. bertholletiae) infection. Methods Clinical diagnosis and treatment process of an adult patient who was admitted to the department of hematology of a hospital due to invasive mucormycosis caused by C. bertholletiae infection were summarized. Relevant literatures were retrieved from databases for reviewing. Results A 54-year-old male patient was admitted to hospital due to “repea-ted fatigue for more than one year aggravated, and fever for one week”. Pathological examination of lung tissue showed broad, irregular and undivided hyphae, thus morphologically identified as Mucor. Metagenomic next-genera-tion sequencing(mNGS) of alveolar lavage fluid and peripheral blood detected C. bertholletiae, thus patient was diagnosed as invasive mucormycosis caused by C. bertholletiae infection. Infection was controlled after treatment with liposome amphotericin B combined with posaconazole and caspofungin. 37 literatures were retrieved and 44 patients were involved(including the case in this study). There were 26 males and 18 females, with a median age of 52.5(18-79) years. Underlying diseases were mainly hematological diseases(65.9%, n=29), and 14 cases underwent hematopoietic stem cell or solid organ transplantation. The most common sites of invasion were lung, brain and skin, with 36, 9 and 9 cases respectively. Number of cases with positive results of histopathology, fungal culture, direct microscopy and molecular detection were 28, 37, 29 and 17, respectively. 41 patients received antifungal therapy, 8 of whom underwent surgery. 30 cases died, with a mortality of 68.2%. The survival rate of patients receiving antifungal therapy combined with surgery(62.5%, 5/8) was higher than those with antifungal therapy alone(24.2%, 7/33). Conclusion Adult mucormycosis caused by C. bertholletiae infection is most common in patients with hematological diseases. Hematopoietic stem cells or solid organ transplantation are high-risk factors. The common infection site is lung and can invade multiple organs of the body, with high mortality. Pathogenic diagnosis is very important. Antifungal combined surgery can improve the survival rate.