The malignant transformation of benign intracranial epidermoid cysts is rare. Here, we report a case of an intracranial primary squamous cell carcinoma within an epidermoid cyst. Thus, we present this case along with a comprehensive review of the relevant literature. A 56-year-old woman presented with dizziness, tinnitus, and disequilibrium persisting for three months. Initial magnetic resonance imaging (MRI) indicated a neurogenic tumor on the right side of Meckel’s cave and bilateral vestibular schwannomas, suggesting neurofibromatosis type 2. However, follow-up MRI after two months revealed a significant increase in the size of the right Meckel’s cave tumor. Consequently, surgical resection was performed, which was initially considered a rapidly growing neurogenic tumor. Histopathological examination revealed a squamous cell carcinoma arising from an epidermoid cyst. Whole-body metastasis evaluation showed no primary tumors in other areas; however, leptomeningeal seeding at the sacral canal level was identified. Subsequently, the patient underwent craniospinal irradiation and chemotherapy, followed by radiation therapy. Although most epidermoid cysts follow a benign clinical course, we encountered an exceptionally rare case of malignant transformation originating from an epidermoid cyst. Because definitive management strategies are lacking, further clinical trials concerning the optimal treatment and outcomes of this disease are required.
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