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Keywords: trisomy 18 / osteogenesis imperfecta / amelogenesis imperfecta / dentinogenesis imperfecta / dental care Abstract --Trisomy 18 and osteogenesis imperfecta are serious diseases with multiple systemic complications. Osteogenesis imperfecta is usually associated with dentinogenesis imperfecta. Observation: A young woman consulted for dental care, with the family suspecting multiple caries because of yellow spots on her teeth. Medical history revealed trisomy 18 associated with osteogenesis imperfecta. The patient was quite uncooperative, even with tooth brushing at home. We provided care after the completion of clinical and radiographic examinations. The colorations were due to amelogenesis imperfecta. Discussion: The association between osteogenesis imperfecta and amelogenesis imperfecta has not been described in the literature. Moreover, the presence of an extra abnormality in tooth structure was a delicate situation for the family. This case suggests the link between amelogenesis imperfecta and osteogenesis imperfecta. Conclusion: This case is the first description of the association of amelogenesis imperfecta and osteogenesis imperfecta.