• Xingyue Yuan, Ruibo Li, and Qian Liu.
• Department of Pathology, Deyang Peoples' Hospital, Deyang, Sichuan Province, China.
• Medicine (Baltimore). 2025 Jan 10; 104 (2): e41210e41210.

RationaleEpendymomas are commonly prevalent intramedullary neoplasms in adults, with hardly any cases of exophytic extramedullary ependymoma being reported. Meningiomas, on the contrary, are one of the most common intradural extramedullary (IDEM) tumors. However, the occurrence of both IDEM tumors simultaneously is extremely rare.Patient ConcernsA 63-year-old female who presented with pain and numbness in both lower limbs, and symptoms rapidly progressed over the past 5 months.DiagnosisBased on the patient's clinical symptoms and imaging features, we conducted pathological examination and genetic testing, ultimately confirming that the patient had IDEM double primary ependymoma and meningioma.InterventionsSurgery was performed to remove double spinal tumors, decompress spinal nerve roots, and perform laminectomy, and she was treated with electrocardiogram monitoring, antibiotics, hemostasis, and antiedema therapy.OutcomesHistopathology confirmed World Health Organization grade II ependymoma at L2 and World Health Organization grade I meningioma at T12-L1. MYCN amplification and other genetic alterations were absent. Postoperative recovery was favorable, with no recurrence at 6-month follow-up.LessonsThis is the first reported case of IDEM double primary ependymoma and meningioma, highlighting the rarity of such cases and the importance of thorough diagnostic workup and surgical excision for IDEM tumors. Genetic analysis adds to the understanding of these rare tumors and guides management strategies.Copyright © 2025 the Author(s). Published by Wolters Kluwer Health, Inc.

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