Premolar transplantation in a patient with solitary median maxillary central incisor syndrome

•A single upper central incisor without trauma history might be a predictor of holoprosencephaly.•Tooth transplantation is considered an advantageous treatment to replace a maxillary incisor.•Orthodontic treatment of adolescents with SMMCI syndrome can be performed successfully.

This case report describes the orthodontic treatment of an 11-year-old girl with solitary median maxillary central incisor syndrome, a presumed microform of holoprosencephaly. Because both second premolars were missing in the maxilla, deciduous molar extraction and orthodontic space opening were performed, moving the solitary median maxillary central incisor electively off-center. A mandibular second premolar was transplanted to replace the missing incisor. The resulting spaces could be orthodontically closed in both arches. Prosthodontic reshaping of the transplanted tooth after debonding completed the dental treatment.