Neurological deficits commonly associated with anterior choroidal artery infarction (AChAI) include hemiplegia, hemisensory loss, and homonymous hemianopsia, while neuropsychological and perceptual deficits are uncommon. Prominent cognitive function impairment has rarely been reported. Here, we report a case of AChAI with prominent cognitive function impairment without motor deterioration. In contrast to the typical clinical features of AChAI, near complete and rapid motor recovery was observed, while cognitive impairment persisted despite rehabilitation therapy.