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Objective: Osteogenesis imperfecta (OI) is a rare, hereditary disease affecting collagen type-1 in connective tissue. Collagen type-1 is a substantial component of dentine, and it is speculated, whether affected dentine could cause altered mesiodistal tooth dimension possibly affecting restorative treatment regimen. Therefore, the aim of the present study was to measure mesiodistal tooth dimensions in individuals with OI and compare them with healthy controls. Materials and methods: Fifty-seven individuals aged 20–77 years with OI type 1–4 were included and 70 control patients aged 11–34 years were drawn from an orthodontic database. Mesiodistal tooth dimensions of all tooth types, except third molars, were measured in mm (two decimals) on digital 3 D-models of the tooth-bearing arches. Results: Multilevel mixed-effects linear regression analysis showed that mesiodistal tooth dimension on average was 0.17 mm (95% CI = (−0.33; −0.01)) reduced for the OI group compared to controls. The analysis revealed variation between tooth types; incisors and first premolars were most affected and molars minimally affected. Conclusions: The mesiodistal tooth dimension in individuals diagnosed with OI is significantly smaller compared to healthy controls, which should be taken into consideration in the restorative treatment planning of individuals with OI, although the magnitude of the deviation is relatively small. The results on mesiodistal tooth dimensions of the present controls may be used as a standard for comparisons in future studies on tooth dimensions.