Abstract: Objective To improve the understanding of systemic lupus erythemmatosus (SLE) associated with cryptococcal meningitis (CM) through a case report and literature review. Methods Clinical data of one case of SLE patient with CM was reported, followed by review of relevant literatures, to summarize the characteristics of this disease. Results This patient was a 33-year-old female, receiving long-term of hormone and immunosuppressive therapy for SLE. During the disease course, the patient had a sudden onset of epilepsy, which was confirmed to be CM due to the cryptococcus neofonmans in the cerebrospinal fluid smear and culture. Conclusion SLE accompanied with CM is a rare disease. When epilepsy occurs in the SLE patients, besides active SLE, the possibility of central nervous system infection should be considered. Timely and effective lumbar puncture, cerebrospinal fluid smear and culture are important to confirm this disease.