Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 10 2 2025 10 15 69 74 EN Mohammad Ali Nahayati Neurology Department, Ghaem Hospital, Mashhad University of Medical Sciences, Mashhad, Iran. Maryam Payere Department of Neurology, Mashhad University of Medical Sciences, Mashhad, Iran. Amir Khanmirzaei School of Medicine, Shahroud University of Medical Sciences, Shahroud, Iran. Bahar Karimikhoshnoudian School of Medicine, Islamic Azad University of Tehran Medical Sciences, Tehran, Iran. Soheil Shokri-shakib Cardiology Department, Shahid Beheshti University of Medical Sciences, Tehran, Iran. 2025 07 09 2025 09 08 Spontaneous intracranial hypotension (SIH) is an uncommon condition caused by cerebrospinal fluid (CSF) leakage, which can lead to complications such as cerebral venous thrombosis (CVT), occurring in 1–2% of SIH patients. This case report describes the presentation, diagnosis, and management of SIH complicated by CVT. A 36-year-old woman with no comorbidities presented with a three-week history of severe, initially orthostatic headaches. Neurological examination revealed mild bilateral disc oedema, and imaging showed generalized brain oedema. MRI with MRV confirmed cerebral venous sinus thrombosis along with typical SIH features, including pachymeningeal enhancement and venous engorgement. She was treated with heparin infusion and isotonic saline, responding well without seizures or hemorrhagic complications. This case highlights the risk of CVT in SIH and underscores the importance of early diagnosis through neuroimaging and lumbar puncture. The patient’s favorable response to anticoagulation demonstrates effective management, though further studies are needed to establish definitive treatment guidelines for SIH-associated CVT. https://crcp.tums.ac.ir/index.php/crcp/article/view/1068 https://crcp.tums.ac.ir/index.php/crcp/article/download/1068/717