In addition to neuromotor, cognitive, vision and hearing impairments, it is also possible to observe dental occlusion and maxillofacial changes in children with microcephaly diagnosis. But, how about long-term effects on the craniofacial growth and development of children affected by this clinical condition in skeletal maturity? In this context, two clinical cases of teenage dizygotic twins will be presented, in which one had microcephaly, focusing on their oral and maxillofacial features.