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The corpus callosum plays a major role in transmitting information between the two brain hemispheres. Absence of the corpus callosum (known as agenesis of the corpus callosum– [ACC]) is a congenital anomaly that can lead to difficulties in intellectual, cognitive, and emotional development. The neuropsychological profile of ACC is difficult to establish given the heterogeneity observed in subjects with this type of abnormality, especially when ACC is associated with other anomalies. This review article summarizes the main data from research studies on the neuropsychological profile of individuals with ACC, particularly those presenting a so-called primary or isolated agenesis. A neuropsychological profile with specific cognitive and emotional difficulties seems to emerge, thus enabling us to propose the possible existence of a neuropsychological syndrome of ACC.