Objectives. Leptomeningeal metastasis (LM) of primary uterine cervical cancer is rare and treatment options are limited. In this case report and literature review, we aimed to present a patient with cervical cancer with LM and discuss previously reported cases in the literature.Case presentation. Our case was a 58-year-old patient who was initially diagnosed with metastatic primary uterine cervical cancer and treated with chemotherapy and chemoradiotherapy. During follow-up, she developed neurological symptoms, and LM was detected in the craniospinal regions. Cerebrospinal fluid cytology examination has confirmed metastatic disease. The patient was treated with concurrent intrathecal methotrexate and whole-brain radiotherapy (WBRT). A good clinical and cytological response was obtained. However, while intrathecal methotrexate was being continued after WBRT, she succumbed to hematological toxicity before the radiological response could be evaluated.Conclusions. LM is an extremely rare and catastrophic distant spread pattern in patients with cervical cancer.In the literature, a total of 26 patients were reported up to date. Median survival after detection of LM was nine weeks, including our case. Multimodal treatment combinations such as systemic and intrathecal chemotherapy and radiotherapy (RT) were used. However, most of these reports did not have detailed information about toxicity.Despite the combined use of aggressive treatment modalities, patients have limited survival and very high risks of hematologic toxicity. Concurrent use of intrathecal chemotherapy and radiotherapy should be avoided due to increased risk of morbidity.