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INTRODUCTION Pituitary apoplexy is defined as a sudden neurologic deficit as a result of infarction or hemorrhage within the pituitary gland. In this study, we report a rare case of apoplexy presenting with cerebral infarction due to direct compression of the internal carotid artery (ICA) and review the literature. CASE REPORT A 31-year-old male presented with sudden-onset headache, right hemiparesis, decreased left monocular visual acuity, and a nasal visual field deficit of the left eye. On computed tomography angiography (CTA) there was evidence of a hyperdense sellar/suprasellar mass with stenosis of the cavernous and supraclinoid segments of the ICAs bilaterally. However, on magnetic resonance imaging angiography (MRI/MRA) the following day there was a complete occlusion of the left cervical ICA as well as cystic changes of the sellar and suprasellar mass suggestive of pituitary hemorrhage. The patient underwent urgent endoscopic endonasal decompression of the mass and post-operative DSA demonstrated restored flow within the left cervical ICA. CONCLUSION Twenty-nine cases of cerebral infarction due to pituitary apoplexy have been previously documented with the majority of cases related to direct ICA compression. Vascular compression is associated with a high rate of mortality (24%) and should be treated urgently by surgical decompression in cases of severe or progressive neurological symptoms.