Objective: Infantile melanocytic neuroectodermal tumor is a rare, locally aggressive, osteolytic neoplasm, most commonly seen in the anterior maxilla of infants. The tumor contains cuboidal and epitheloid cells that contain melanin in alveolar and tubular patterns, and neuroblast-like round cells. Although this tumor usually shows rapid growth, it is generally considered benign.

Case Report: Two patients with unusual localizations at the arm and parietal bone are presented.

Conclusion: PRAME expression in this tumor has not been investigated so far. We present these two cases with PRAME immunoexpression in the light of the literature.