Penicillium marneffei is an important opportunistic pathogen in Southeast Asia in HIV-positive individuals, but it rarely infects non-HIV ones. Four SLE patients with disseminated penicilliosis had been previously reported out of which 3 died. We describe a 46-year-old Chinese woman who had a 10 years history of SLE, associated with disseminated Penicillium marneffei infection, which presented as fever, subcutaneous masses, and fine nodular shadows disseminated over lung fields. She was initially misdiagnosed as miliary tuberculosis and panniculitis that did not respond to anti-tubercular drugs and prednisone. The correct diagnosis was finally made by histopathology and tissue culture and also culture from exudate. She responded well to antifungal therapy in the form of intravenous amphotericin B for 2 weeks followed by itraconazole plus fluconazole. The cutaneous lesions were cured leaving behind scars by secondary suture after times of epluchage, and the fine nodular shadows over lungs disappeared finally. She had no recurrence on 8 months of follow-up. We also review the literature on this topic.