Oral Health-Related Quality of Life in Dutch Adults With Osteogenesis Imperfecta

Abstract

Objective: To explore the oral health-related quality of life and its possible risk factors among adults with Osteogenesis Imperfecta using the Oral Health Impact Profile (OHIP)-49 questionnaire. Secondary objectives were to investigate the impact of self-reported Osteogenesis Imperfecta, Dentinogenesis Imperfecta, and age on various dental parameters.

Materials and methods: A cross-sectional questionnaire was distributed online to 417 Dutch adults with Osteogenesis Imperfecta at three national referral centers. Multivariate linear regression was performed to identify indicators for OHIP-49 Scores. The effect of Osteogenesis Imperfecta, Dentinogenesis Imperfecta, and age on various dental parameters was investigated using logistic regressions (p < 0.05).

Results: One hundred and fifty-five questionnaires (37.2%) were suitable for analysis. Osteogenesis Imperfecta type III was significantly associated with higher OHIP-49 scores as compared to type I. Symptoms of temporomandibular disorders, missing teeth or dentures also increased the OHIP-49 scores significantly. Osteogenesis Imperfecta type IV and increasing age were associated with missing teeth. There was a 31.94 times (95% CI: 8.56-119.13) higher odds of tooth fracture with self-reported Dentinogenesis Imperfecta.

Conclusions: Adults with Osteogenesis Imperfecta type III report a lower oral health-related quality of life compared to Osteogenesis Imperfecta type I adults.

Keywords: Dentinogenesis Imperfecta; Oral Health Impact Profile; Oral health‐related quality of life; Osteogenesis Imperfecta; rare bone condition; rare disease; self‐reported oral health.