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. The gene discussed is CAMSAP1; the disease is tubulinopathy.