Intra-osseous Schwannomas are rare primary tumors, accounting for 1% of all primary tumors seen in the maxillofacial region. Schwannomas commonly manifest as well-encapsulated, asymptomatic, slow-growing lesions with late evidence of cortical expansion. Radiographically, intra-osseous Schwannomas present as well-defined unilocular radiolucent lesions with no pathognomonic radiological features. Here an unusual case of a 30 years old female patient with a diffuse swelling in the left malar

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... has been reported. On histopathological analysis it was diagnosed as a case of benign aggressive Schwannoma. Routine radiological examination along with computed tomography and Magnetic resonance imaging revealed extensive involvement of the bone with intra-cranial extension. The rare occurrence of intra-osseous Schwannoma with clinical presentation mimicking various other lesions makes diagnosis of this lesion a challenge.