Prosopometamorphopsia is a disorder of face perception in which faces appear distorted to the perceiver. Cases with unilateral prosopometamorphopsia caused by splenial lesion have been very rarely reported.

A 52-year-old right-handed woman complained that the left half of people's faces looked distorted. She stated that objects other than the face looked normal. Brain magnetic resonance imaging revealed an infarction of the left splenium of the corpus callosum. Electroencephalography and automated perimetry were normal.

The mechanism of unilateral prosopometamorphopsia remains unclear. However, it could be a dominant hemisphere-specific disconnection sign.