Ewing's sarcoma (ES) is an aggressive, poorly differentiated round cell tumor in bone or soft tissues. This rare neoplasm is found primarily in the long bones of limbs and flat bone of pelvis with a predilection in the second decade in life. Primary ES of the maxillofacial region is exceptional and constitute 1% ± 4% of all cases. This case report is aimed at highlighting a very rare clinical presentation of extraosseous ES of the chin region-a first of its kind.