Ciguatera poisoning (CP) is a marine foodborne illness caused by the ingestion of fish and marine invertebrates contaminated with neurotoxins known as ciguatoxins, produced by benthic microalgae from Gambierdiscus and Fukuyoa genera. Although the classical triad of digestive, cardiovascular, and peripheral neurological manifestations that characterizes this poisoning is now well recognized and extensively described in the literature, central neurological involvement remains poorly documented and understood. The present paper describes, to our knowledge, the fifth reported case worldwide of cytotoxic lesions of the corpus callosum syndrome (CLOCCS) occurring in the context of CP. A review of the previously reported cases was also undertaken to improve understanding of the pathophysiology of this syndrome, its clinical manifestations, imaging features, and prognosis, given the rarity of such presentations in CP.