Background and purpose Cerebral venous sinus thrombosis (CVST) is a rare condition but an important complication of spontaneous intracranial hypotension (SIH). We reviewed our experience of patients with SIH and assessed for the presence of CVST. Methods We reviewed the medical records and imaging studies from our tertiary referral institution, assessing patients with clinically suspected SIH as well as imaging findings of intracranial hypotension. All relevant brain MRIs were reviewed for the presence of brain sag, pachymeningeal enhancement, and CVST. Results Among 563 patients with a clinical diagnosis of SIH, 431 (76%) demonstrated brain sag. In the overall patient cohort, a total of 5 patients had CVST (0.9%) and all 5 of these patients demonstrated findings of brain sag. Of the patients with CVST, 3 had significant complications, including dural arteriovenous fistulas (2 patients) and lobar hemorrhage with seizure (1 patient). Conclusion SIH is a risk factor for the development of CVST. In our review of 563 patients with clinical and/or imaging findings of SIH, 0.9% of patients were diagnosed with CVST and 3 of these patients (60%) had additional severe complications.