The congenital absence of the corpus callosum is a rare cerebral malformation that is commonly reported, but only one case has been associated with SIDS. The case involved a 5-week-old boy who was found cold and unresponsive a few hours after being put to bed. He was rushed to the hospital in a coma and was put on a life support machine. A cranial computed tomography scan indicated severe cerebral edema. There was no evidence of a space-occupying lesion, and the characteristic wing shape of the ventricles caused by callosal agenesis could not be discerned. Postmortem examination provided neuropathological confirmation of the absent corpus callosum by demonstrating an uninterrupted continuation of the leptomeninges with the ependymal lining of the ventricles. The overall feature was that of anoxic-ischaemic encephalopathy and consistent with "near miss" SIDS. 11 references and 1 figure (Author abstract modified)