Sigmoid sinus thrombosis was once one of the most common intracranial complications of otitis media, but this has become less common since the advent of antibiotics. The frequency of sigmoid sinus hypoplasia reported in the literature is about 17%. Considering the possibility of postoperative intracranial hypertension, sigmoid sinus and jugular bulb surgeries were contraindicated in this case.We present a case with sigmoid sinus thrombosis of the sinus due to compression flowing surgical injury. This is a unique case, which presents with contralateral sigmoid sinus hypoplasia. Eventually, the patient presented to us with otitic hydrocephalus, increased intracranial pressure, papilledema, oculomotor and abducens nerve palsy, and severe right-side visual loss are prominent features.