Objective To analyze clinical characteristics and therapeutic strategies for patients with Trichosporon asahii (T. asahii) bloodstream infection (BSI), and provide reference for clinical diagnosis and treatment for such disease. Methods Diagnosis and treatment process of a patient with T. asahii BSI were reported. China National Knowledge Infrastructure(CNKI), Wanfang, PubMed, and Web of Science databases were retrieved using keywords: "Trichosporon asahii" and "bloodstream infection". Patients’ age, gender, underlying diseases, immune status, treatment, and clinical outcome as well as antimicrobial susceptibility testing results of T. asahii were analyzed retrospectively. Results The leukemia patient developed T. asahii BSI during chemotherapy, and showed clinical improvement after treatment with fluconazole+flucytosine. A total of 44 cases (43 from literature plus this index case) were analyzed. Among these cases, 28 were males, the median age was 54.5 years old. The underlying diseases were predominantly hematological diseases. Catheter, urinary tract, and skin were identified as concurrent culture-positive sites. Azoles exhibited good antimicrobial activity in vitro, with voriconazole showing the strongest activity and associated with significantly higher survival rates. Under the guidance of antimicrobial susceptibility testing results, patients with fluconazole treatment had a higher survival rate. At minimum inhibitory concentrations (MICs) of 4-8 μg/mL of fluconazole, fluconazole combined with other agents was predominantly required. Amphotericin B demonstrated good in vitro activity, but the overall survival rate of patients was low when amphotericin B was used as monotherapy without azoles. Catheters removal, surgical intervention, and neutrophil recovery were influencing factors for enhanced survival rates. Conclusion BSI caused by T. asahii primarily occurs in patients with hematological diseases, with neutropenia being a high-risk factor. Infection sites at catheter, urinary tract, and skin may be infection sources. Clearing the source of infection and promoting the recovery of neutrophils can help to improve survival rates of patients. For voriconazole-intolerant patients, fluconazole monotherapy or in combination with flucytosine/amphotericin B can be used alternatively based on antimicrobial susceptibility testing results.