Taken together, the CAMSAP1-related neuroradiology may best be described as resembling that of an atypical tubulinopathy but with a distinct and unusual pattern of abnormalities (classic lissencephaly with a P > A gradient and complete agenesis of the corpus callosum), which appear pathognomonic of the disorder. This evidence concerns the gene CAMSAP1 and lissencephaly spectrum disorders.