Introduction: Polymicrogyria, the appearance of irregular small and over numbered gyri on the surface of the brain, has been reported as the most frequent finding, when exists, on the temporal lobe. Case Presentation: A patient, male, age of 35, came to the regular head and brain MRI scan due to psychiatric and hormonal treatment within transsexualism. There were no data of epilepsy in a patient’s history. MRI scans were acquired with a 1.5 T Siemens Magnetom system with a standard head coil. Three-dimensional 3D RAGE, contiguous 1.0 mm slice, and 1 acquisition sequence underwent final 3D rendering and subsequent volumetry. On the definitive 3D image, we have revealed an unusual gyrisation on the left temporal lobe which had a picture of partial polymicrogyria. Conclusion: This is a unique finding, to our knowledge, of the unilateral temporal polymicrogyria in a person with transsexualism. Although polymicrogyria is mostly related to epileptifom attacks, its impact on the transsexualism appearance is opened to be examined.