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Abstract Background Pilocytic astrocytomas are the most common primary brain tumors in children. Most of the lesions occur in or near the midline. They are rare in extra-axial locations. Case presentation We report a 23-year-old lady who was found to have progressive right-sided sensorineural hearing loss and features of raised intracranial pressure due to a pilocytic astrocytoma of the right cerebellopontine angle. After an extensive Medline search, we could find only three cases of primary extra-axial pilocytic astrocytoma of the cerebellopontine angle (CPA) reported till now. Conclusion Even a rare occurrence, primary pilocytic astrocytoma, and in general gliomas, should be kept in mind in the differential diagnosis of CPA lesions.