Gliomatosis peritonei (GP) is a condition characterized by the dissemination of mature glial tissues throughout the peritoneal cavity. It is usually associated with immature ovarian teratoma but presents with mature cystic teratoma (MCT) in 1% of cases. GP, associated with MCT, is a benign disorder. The majority of cases remain asymptomatic and rarely recur. Here, we present a case of a 22-year-old woman with a history of abdominal enlargement and severe abdominal pain who underwent exploratory laparotomy, peritoneal fluid cytology, bilateral salpingo-oophorectomy, appendectomy, omental biopsy, and Jackson-Pratt drain insertion with histopathologic result of GP with MCT. A month later, the patient had a recurrence of abdominal enlargement, necessitating a second surgery. Immunohistochemistry for histopathologic evaluation and diagnostic imaging are crucial in confirming the diagnosis and guiding the treatment strategy. A multidisciplinary team approach in monitoring and comprehensive support is significant in optimizing outcomes for patients with aggressive GPs associated with MCT. Further research and clinical experience are essential to establish a standardized guideline to improve the management and clinical outcome of this condition.