Mita Atsuyoshi, Hirano Shohei, Uehara Takeshi, Uehara Kai, Ohno Yasunari, Kubota Koji, Masuda Yuichi, Notake Tsuyoshi, Yoshizawa Kazuki, Shimizu Akira, Soejima Yuji
Division of Gastroenterological, Hepato-Biliary-Pancreatic, Transplantation and Pediatric Surgery, Department of Surgery, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, 390-8621, Japan.
Department of Pathology, Shinshu University School of Medicine, Matsumoto, Japan.
Surg Case Rep. 2022 Sep 2;8(1):164. doi: 10.1186/s40792-022-01516-4.
Fungal infection may develop because of immunosuppression after organ transplantation, in which invasive types, such as Aspergillus and Mucorales, fungi cause morbidity. We present a case of disseminated mucormycosis due to Cunninghamella bertholletiae after ABO-incompatible living donor liver transplantation (LDLT).
A 47-year-old man with decompensated liver cirrhosis and hepatocellular carcinoma underwent an ABO-incompatible LDLT using a graft procured from his son, who had a different blood type. Rituximab and mycophenolate mofetil were administered 3 weeks before LDLT as immunosuppressive therapy. Although liver graft function improved, mass-like infiltrates appeared in the lungs following intubation for > 1 week due to impaired consciousness. The brain magnetic resonance imaging findings were normal. Decreased ejection fraction and ST elevation were detected on echocardiography and electrocardiography, respectively. There was no dominant stenosis on coronary arteriography. The recipient underwent segmentectomy of the right lung 20 days after LDLT. C. bertholletiae was identified from a specimen using polymerase chain reaction, thus establishing a diagnosis of mucormycosis. Multiple infarctions in the brain, heart, and kidney developed within 2 weeks. Treatment with amphotericin B was ineffective. The patient developed circulatory collapse, and a temporary pacemaker and percutaneous coronary intervention were required for cardiac infarction. The recipient died of cardiac failure 27 days after the LDLT. Autopsy revealed disseminated mucormycosis involving the brain, thyroid, heart, lung, liver, gastrointestinal tract, and both kidneys. In addition, fungal endocarditis may have been responsible for septic emboli in multiple organs, resulting in multiple organ invasion. Hypothrombocytopenia was present since the pre-transplant period, and the recipient was diagnosed posthumously with myelodysplastic syndrome due to hereditary abnormalities. Multiple factors such as organ transplantation, bone marrow dysfunction, immunosuppression, and inadequate administration of antifungal reagents might have promoted mucormycosis development in our patient.
Mucormycosis by C. bertholletiae is a fatal complication; thus, early diagnosis and treatment are warranted before multiple organ invasion.
器官移植后因免疫抑制可能发生真菌感染，其中曲霉菌和毛霉目等侵袭性真菌可导致发病。我们报告1例ABO血型不相容的活体供肝肝移植（LDLT）术后由柏氏小克银汉霉引起的播散性毛霉病病例。
一名47岁失代偿期肝硬化合并肝细胞癌男性患者接受了LDLT，供肝来自其血型不同的儿子。LDLT前3周给予利妥昔单抗和霉酚酸酯作为免疫抑制治疗。尽管肝移植肝功能改善，但因意识障碍插管>1周后肺部出现团块状浸润影。脑磁共振成像检查结果正常。超声心动图和心电图分别检测到射血分数降低和ST段抬高。冠状动脉造影未发现明显狭窄。肝移植术后20天，受者接受了右肺部分切除术。通过聚合酶链反应从标本中鉴定出柏氏小克银汉霉，从而确诊为毛霉病。2周内脑、心、肾出现多处梗死。两性霉素B治疗无效。患者出现循环衰竭，心脏梗死需要临时起搏器和经皮冠状动脉介入治疗。肝移植术后27天，受者死于心力衰竭。尸检显示播散性毛霉病累及脑、甲状腺、心脏、肺、肝、胃肠道和双肾。此外，真菌性心内膜炎可能是多器官脓毒性栓子的原因，导致多器官受累。自移植前期即存在血小板减少，受者死后诊断为遗传性异常所致的骨髓增生异常综合征。器官移植、骨髓功能障碍、免疫抑制和抗真菌药物使用不足等多种因素可能促使我们的患者发生毛霉病。
柏氏小克银汉霉引起的毛霉病是一种致命并发症；因此，在多器官受累之前有必要进行早期诊断和治疗。
