Yu Pang,1 Diefei Hu,1 Yiwu Dang,2 Siming Huang,3 Lanhui Qin,4 Meng Li5 1Department of Infectious Disease, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of China; 2Department of Pathology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of China; 3Department of Respiratory Medicine, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of China; 4Department of Radiology, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of China; 5Department of Clinical Laboratory, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of ChinaCorrespondence: Meng Li, Department of Clinical Laboratory, First Affiliated Hospital of Guangxi Medical University, Nanning, Guangxi, 530021, People’s Republic of China, Tel/Fax +8613367809642, Email gxmulimeng@foxmail.comBackground: Apiotrichum mycotoxinivorans is a rare mycotoxinivorans, and its pathogenicity is unknown. Bronchial artery shunt is a pathophysiological state following congenital or acquired chronic infection. We report a rare case of bronchial artery shunt by A. mycotoxinivorans infection in a recurrent hemoptysis patient.Case Presentation: A 45-year-old female presented with recurrent cough and hemoptysis for 4 years. Before admission, she had been treated in several hospitals for pulmonary tuberculosis and bronchiectasis and received standardized anti-tuberculosis treatment for 1 year, but it was ineffective. After admission, CTPA and bronchial arterial angiography showed left bronchial artery–left pulmonary artery shunt and right bronchial artery–right pulmonary artery shunt. Fiber-optic bronchoscopy was performed, which revealed a large amount of purulent secretions, bronchoalveolar lavage fluid fungi (1-3)-β-d glucan: 728.06, and GM test: 3.239. Fungal hyphae and spores were observed by gram staining of BALF smear. Acid-fast bacilli were not found in BALF smear and brush smear. Two consecutive BALF fungal cultures grew A. mycotoxinivorans, the identity of which was confirmed by internal-transcribed-spacer (ITS) sequencing. Intravenous amphotericin B liposome (30mg; 0.5mg/kg, QD) was given for 2 weeks, embolization was performed, and itraconazole (voriconazole allergy) was taken orally for 9 months after operation. Hemoptysis and pulmonary lesions gradually improved after treatment.Conclusion: We report the first case of bronchial artery–pulmonary artery shunt in a patient diagnosed with A. mycotoxinivorans infection. Phagocytosis of fungi by leukocytes was observed, and the pathogenicity of the fungus was confirmed in order to heighten the awareness of these infections.Keywords: Apiotrichum mycotoxinivorans, bronchial artery–pulmonary artery shunt, hemoptysis, infection