Cytotoxic lesions of the corpus callosum are lesions secondary to different medical conditions. Radiologically, lesions are identified on magnetic resonance imaging as a hyperintense signal on diffusion-weighted imaging and decreased apparent diffusion coefficient values of the splenium of corpus callosum. Signal changes are reversible in almost totality of the cases. Previous cases of cytotoxic lesions of the corpus callosums have been associated with several metabolic disturbances, but ketotic hyperglycemia has never been reported. We here discussed the case of 28-year-old patient with complex visual hallucinations presenting with cytotoxic lesions of the corpus callosums and type I diabetes. Treatment of hyperglycemia was followed by full clinical recovery and complete regression of the radiological abnormalities at 3-month follow-up. Elevated levels of circulating pro-inflammatory mediators associated with ketotic hyperglycemia in type I diabetes support an implication of cytokines in the pathophysiology of the cytotoxic lesions of the corpus callosums.