Background: Cerebral venous sinus thrombosis (CVST) is a life-threatening condition with a predilection for a younger age group. Patients with CVST can have features of raised intracranial pressure that can be suspected clinically based on ophthalmological findings such as bilateral abduction restriction, papilledema.
Case description: A 27-year-old gentleman presented with 15 days history of headache and seizures with complete abduction restriction of both eyes along with retraction of globe and narrowing of palpebral fissure on abduction. His MRI brain showed superior sagittal sinus thrombosis without any parenchymal lesion.
Conclusion: The final diagnosis in our case was bilateral inverse Duane's retraction syndrome with CVST, a co-occurrence that has not been reported in the past and can be missed if the eye movements are attributed to raised intracranial pressure alone.
Keywords: Cerebral venous sinus thrombosis; inverse Duane's retraction syndrome.