南方医科大学学报

南方医科大学学报 ›› 2022, Vol. 42 ›› Issue (5): 780-784.doi: 10.12122/j.issn.1673-4254.2022.05.22

• • 上一篇    

儿童小克银汉毛霉病的临床特征分析：1例报告并文献复习

吴飞凤，田继东，佘 周，刘 瑛，万伍卿，文 川   

  1. 中南大学湘雅二医院儿童医学中心，湖南 长沙 410011
  • 出版日期:2022-05-20 发布日期:2022-06-02

Clinical features of children with Cunninghamella spp. infection: a case report and literature review

WU Feifeng, TIAN Jidong, SHE Zhou, LIU Ying, WAN Wuqing, WEN Chuan   

  1. Department of Pediatrics, Second Xiangya Hospital of Central South University, Changsha 410011, China
  • Online:2022-05-20 Published:2022-06-02

RichHTML

24

PDF

664

摘要： 目的 探讨儿童小克银汉毛霉病的临床特征及诊治方法，提高对该病的认识。方法 报道某院1例宏基因组学二代测序技术（mNGS）临床诊断的小克银汉毛霉病患儿，经两性霉素B及卡泊芬净治疗有效；检索Pubmed、Embase、知网、万方数据库，纳入儿童小克银汉毛霉病病例，分析该病的临床特征及诊治。结果 该例急性淋巴细胞白血病患儿在化疗后出现发热、呼吸道症状，临床诊断侵袭性真菌病，采用mNGS对外周血DNA序列进行分析，通过与PMDB病原学数据库对比，鉴定出小克银汉霉属，序列数514，接受两性霉素B联合泊沙康唑治疗有效。纳入文献分析小克银汉毛霉病临床特征：22例患者中男性12例，中位年龄13.5（3~18）岁，最常见的基础疾病为血液系统恶性肿瘤（19/22）。大部分患者急性起病，快速进展，最常见的首发症状为呼吸症状（14/20）及发热（16/20）。肺部CT表现以单侧病变多见，影像学表现多样，可出现肺结节/肿块、浸润改变、胸腔积液等。22例中确诊18例，临床诊断4例，最常见受累的器官为肺脏及皮肤；确诊手段仍依赖于组织病理学及真菌培养，分子诊断技术如PCR、mNGS等也渐显露头角。诊断后几乎所有病例都接受了抗真菌治疗（21/22），其中13例联合手术治疗，最终，22例患者中死亡9例，总体死亡率42%，症状/体征出现至死亡的平均时间为19（4~54）天。抗真菌治疗联合手术者生存率（9/13，69%）高于单独抗真菌治疗者（3/8，37%）。结论 侵袭性真菌病是血液肿瘤患者常见并发症，但小克银汉毛霉感染少见，死亡率较高，当临床高度怀疑侵袭性真菌病时，应积极诊断，早期启动治疗，以提高其生存率。

关键词: 毛霉病；小克银汉毛霉；白血病；儿童

Abstract: We report a case of mucormycosis induced by Cunninghamella spp. infection in a ten-year-old girl with acute lymphoblastic leukemia, who developed fever and respiratory symptoms after chemotherapy and was diagnosed with invasive fungal disease. Peripheral blood DNA sequences were analyzed using metagenomic next-generation sequencing (mNGS), and by comparison with the Pathogens Metagenomics Database (PMDB), we identified Cunninghamella spp. with sequence number 514 as the pathogen. The patient was treated with amphotericin B combined with posaconazole and showed a favorable response. We searched Pubmed, Embase, CNKI, and Wanfang database for reports of cases of Cunninghamella spp. infection in children and retrieved 22 reported cases (including 12 males) with a median age of 13.5 (3-18) years. In these 22 cases, hematological malignancy was the most common underlying condition (19/22), and most of patients experienced an acute onset and rapid progression with respiratory symptoms (14/20) and fever (16/20) as the most common symptoms. CT imaging often showed unilateral lesions with varying imaging findings, including pulmonary nodules or masses, infiltrative changes, and pleural effusion. Definite diagnoses were established in 18 of the cases, and 4 had probable diagnoses; the lungs and skin were the most frequent organs compromised by the infection. A definite diagnosis of Cunninghamella spp. infection still relied on histopathological examination and fungal culture, but the molecular techniques including PCR and mNGS had shown potentials in the diagnosis. Almost all the cases received antifungal treatment after diagnosis (21/22), and 13 patients also underwent surgeries. Death occurred in 9 (42% ) of the cases at a median of 19 (4-54) days after onset of the signs or symptoms. The patients receiving antifungal therapy combined with surgery had a high survival rate (9/13, 69% ) than those with antifungal therapy alone (3/8, 37%). Invasive fungal disease is a common complication in immunoco-mpromised patients, but Cunninghamella spp. infection is rare and has a high mortality rate. In cases highly suspected of this disease, active diagnosis and early treatment are critical to improve the survival outcomes of the patients.

Key words: mucormycosis; Cunninghamella spp.; leukemia; children