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Background: Glioma is the most common tumor occurring in the brainstem. A primary intraparenchymal meningioma located in the brainstem without dura attachment is rare. Meanwhile, atypical meningiomas that occur in the medulla oblongata parenchyma, and without dura coverage, are extremely rare. In this study, we report the first case of atypical meningioma in the medulla oblongata parenchyma and review the existing literature.Case presentation: A 38-year-old female was admitted at our hospital with a 2-week history of progressive neck and occipital pain. Magnetic resonance imaging revealed a presence of a 1.5x0.9cm mass lesion, located in the left side of the medulla oblongata, which was hypointense on T1-weighted and hyperintense on T2-weighted images, and with inhomogeneous enhancement following gadolinium-diethylenet-riamine pentaacetic acid (Gd-DTPA) administration. The initial preoperative diagnosis was glioma or angioblastoma. The patient underwent a total surgical resection of the left medulla oblongata tumor and the histopathological examination indicated that the lesion was an atypical meningioma. The patient returned to normal life after surgery.Conclusions: Although glioma is the most common tumor occurring in the brainstem parenchyma, the possibility of meningioma cannot be ruled out in this area.