To report a case of cerebral venous sinus thrombosis (CVST) secondary to pegaspargase and perform a literature review.

Pegaspargase is a chemotherapy drug used to treat acute lymphoblastic leukemia (ALL). Compared to L-asparaginase, it has a longer half-life, low immunogenicity, and fewer adverse reactions. However, CVST associated with pegaspargase has been reported.

Case report and literature review. The PubMed database was used to find studies about pegaspargase and CVST published until June 2024. The search terms were “peg-asparaginase, pegylated asparaginase, pegaspargase, and cerebral venous thrombosis.” The initial search obtained 39 studies, of which eight were selected. Of these eight reports, only six provided enough demographic data. The general data, including the six studies from the literature and our current report, will be summarized.

A 34-year-old female with T cell-ALL presented with one episode of generalized tonic-clonic seizure with return to baseline. Her initial neuroimaging revealed superior sagittal sinus and cortical vein thromboses with a left frontal lobe hemorrhagic infarction. She was recently in an induction course of CALGB (Cancer and Leukemia Group B) 10403 chemotherapy protocol and pegaspargase. Patient underwent a cerebral venous thrombectomy (CVTE), which was performed with two aspiration passes of subocclusive superior sagittal sinus (SSS) thrombosis and improvement in flow afterward. She was neurologically intact at three months follow-up.