Background: Dentinogenesis imperfecta type I (DGI-I) is a hereditary alteration of dentin associated with osteogenesis imperfecta (OI). Aim: To describe and study the morphological characteristics of DGI-I with scanning electron microscopy (SEM). Material and methods: Twenty-five teeth from 17 individuals diagnosed with OI and 30 control samples were studied with SEM at the level of the enamel, dentin–enamel junction (DEJ) and four levels of the dentin, studying its relationship with clinical–radiographic alterations. The variables were analysed using Fisher’s exact test, with a confidence level of 95% and asymptotic significance. Results: OI teeth showed alterations in the prismatic structure in 56%, interruption of the union in the enamel and dentin in 64% and alterations in the tubular structure in all of the cases. There is a relationship between the severity of OI and the morphological alteration of the dentin in the superficial (p = 0.019) and pulpar dentin (p 0.004) regions. Conclusions: Morphological alterations of the tooth structure are found in OI samples in the enamel, DEJ and dentin in all teeth regardless of the presence of clinical–radiographic alterations. Dentin structural anomalies and clinical dental alterations were observed more frequently in samples from subjects with a more severe phenotype of OI.