BACKGROUND: Cerebral venous thrombosis (CVT) has various risk factors, including contraception, pregnancy, neoplasia, and thrombophilia. Nitrous oxide (N₂O), historically used as an anesthetic and more recently as a recreational drug, has been associated with neurological complications such as myeloneuropathy and thromboembolic events. Here, we report two cases of CVT associated with N₂O use and provide a review of the literature on this association. METHODS: We describe two local cases of CVT associated with N RESULTS: Among the 12 patients, seven had co-existing CVT risk factors. Most patients reported chronic N CONCLUSIONS: These cases highlight an emerging association between CVT and N₂O use. Prompt recognition of this link is critical to recommend cessation of N₂O use, alongside anticoagulation therapy and consideration of vitamin supplementation to prevent complications and recurrence.