Objective To report the diagnosis and treatment of a patient with bloodstream infection (BSI) caused by Trichoderma koningiopsis (T. koningiopsis). Methods Clinical diagnosis and treatment processes of an adult patient admitted to a hospital due to BSI caused by T. koningiopsis were summarized. Relevant reports were retrieved from databases for reviewing. Results A 60-year-old male patient was admitted to hospital due to "intermittent fever with lumbago and knee pain for more than half a month". According to the examination results, the patient was treated with ceftriaxone sodium, levofloxacin and ganciclovir, but didn't achieve obvious improvement. T. koningiopsis was identified by next-generation sequencing (NGS) of peripheral blood, thus T. koningiopsis BSI was considered. After oral administration of voriconazole, the interval between episodes of patients gradually prolonged, from one episode every 2-3 days to one episode every 4-5 days, peak temperature of the patient decreased, and the lumbago and knee pain alleviated. After discharge, the patient continued taking voriconazole orally. After a follow-up of more than 1 month, fever gradually subsided, lumbago and knee pain was relieved, but the patient died suddenly 2 months after discharge. A total of 40 cases were retrieved from literatures, out of which 38 cases were reported by foreign literatures and 2 reported in Chinese. According to the literature retrieval statistics, the under-lying diseases of patients with Trichoderma infection were mainly organ transplantation (n=12), hematological di-sease (n=9), and peritoneal dialysis (n=6). The main clinical manifestations of patients were mainly pulmonary infection (n=8), peritonitis (n=6), and disseminated infection (n=5). The regional distribution was mainly in European and American countries (n=26), 5 cases in Asia, 2 cases in Africa, 1 case in South America, and 6 cases in unknown regions. Trichoderma longibrachiatum was as the main detected pathogen (n=24). Conclusion Trichoderma is an opportunistic pathogen, whose infection often occurs in patients with low immunity, and is easy to be misdiagnosed. It is recommended to conduct NGS to identify the pathogen as soon as possible for timely treatment and reduce mortality.