Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 8 2 2023 11 18 78 81 Maryam Kaeedi Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Hosein Kaeidi School of Medicine, Shahid Beheshti University of Medical Science, Tehran, Iran. Mojtaba Shahbazi Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Sanaz Heydari Havadaragh Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. Hamed Amirifard Iranian Center of Neurological Research, Neuroscience Institute, Tehran University of Medical Sciences, Tehran, Iran. 2022 12 22 2023 03 05 Cystic hydatidosis is a rare zoonotic condition, caused by the larva of the Echinococcus tapeworm.  In this disease brain involvement is a rare condition, accounting for about 2-3% of all cases with hydatid disease. Focal deficits, symptoms of increased intracranial pressure, seizure and mental state changes are most common presentations of CNS cystic echinococcosis. We present a young age woman with resistant headache and transient visual obscurations due to increased intra-cranial pressure which caused by Cerebral venous sinus thrombosis (CVST) arising from Hydatid cyst. To the best of our knowledge, this is the first report of hydatid cyst induced CVST. Our case report highlights the importance of considering CVST as an unavoidable complication of brain’s space occupying lesions and especially infective ones as a differential diagnosis. https://crcp.tums.ac.ir/index.php/crcp/article/view/773