Purpose This report describes a case of a skin sporotrichosis infection and the steps taken to identify an effective antifungal treatment. Patients and methods A 50-year-old woman from Jilin province, China, presented complaining of a small mass that had been on her right upper eyelid for two years. A skin biopsy was taken and submitted for bacterial and mycological assessment. Bacterial culture from the lesion was negative, but a fungal culture was positive. In vitro susceptibility test was performed to assess its susceptibility to antifungal drugs. Results The skin biopsy showed infectious granuloma. Fungal culture was identified as Sporothrix globosa based on both the morphological features and confirmation by the molecular method; it was resistant to many kinds of antifungal drugs, including amphotericin B, voriconazole, fluconazole, and caspofungin. However, it was relatively sensitive to itraconazole. The patient was prescribed 0.2 g itraconazole to be taken twice per day. One month later, she had almost completely recovered from her symptoms. The treatment lasted for 3 months and her liver function and renal function were normal at the endpoint. Conclusion Itraconazole was an effective treatment in this case of a multidrug-resistant sporotrichosis caused by S. globosa.