A 60 year-old man presented with acute on set of left eye proptosis and ptosis. It was associated with poor vision, eye pain and restriction of eye movement of the same eye after 5 hours post left upper molar tooth extraction. The visual acuity in the left eye was hand movement. There was severe ptosis and proptosis of the left eye. The conjunctiva was chemotic with quite anterior chamber. The pupil was mid dilated and sluggished to light. The ocular movement was restricted in all directions. Fundoscopy of the left eye revealed features of central retinal artery occlusion with hyperaemic disc and subretinal exudates at posterior pole. The right eye appeared normal. Urgent MRI brain and orbit revealed severe left paranasal sinusitis with anterior displacement of the left globe and presence orbital abscess. Patient was managed with Otorhinolaryngology and Neurosurgery teams. He underwent emergency transnasal drainage of abscess. Histopathological examination of unhealthy sinus mucosa showed evidence of fungal infection. However, the culture and sensitivity result was inconclusive. Patient was treated with amphotericin B, ceftriaxone, amoxicillin clavulanate and metronidazole. Patient was detected to have high blood sugar level and was managed accordingly. The proptosis improved with treatment. However, his vision, ptosis and ophthalmoplegia remained static. Assessing the immunocompromised status is important for the management of patient presented as acute orbital apex syndrome to avoid fatal outcome.