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Cerebral venous sinus thrombosis (CVST) is an uncommon and underreported neurological condition. This condition has varied aetiologies, clinical manifestations, and significant sequelae if left untreated. We report the case of a 10-year-old male with fever, altered sensorium, cranial nerve neuropathies, and left hemiplegia. His imaging findings revealed features consistent with CVST, and he was subsequently noticed to be passing live worms per rectum while on admission. He was managed nonoperatively, improved significantly, and was discharged with residual motor deficits. In addition, we present a literature review of published case reports of CVST associated with helminthiasis, highlighting its possible pathophysiology, clinical spectrum, and management peculiarities., Competing Interests: There are no conflicts of interest., (© 2024 Journal of The West African College of Surgeons.)