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Dentin dysplasia is a defect of dentin development that was classified by Shields as type I and type II. The teeth in dentin dysplasia type I are characterized by normal enamel and abnormal dentin in the crown and root. For that reason, this disorder is characterized by dental caries, early exfoliation of the teeth and, consequently, maxillomandibular bony atrophy. In this report, we present a case of a young girl affected by dentin dysplasia type I, treated witha combination of onlay bone grafting and a sinus lift technique to accomplish implant placement. This case showed that onlay autogenous grafting and a sinus lift technique are well-tested methods of bony augmentation and can be useful in patients with congenital dentin defects, such as dentin dysplasia.