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. 2025 Mar;31(3):983-992.
doi: 10.1111/odi.15163. Epub 2024 Oct 22.

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

Emmanuelle de Kuijper-Timmermans  1   2 , Lieke Blokland  1   3 , Maurits de Kuijper  4 , Stanimira Kalaykova  5 , M Carola Zillikens  6 , Natasha M Appelman-Dijkstra  7 , Koert Gooijer  8 , Arjan Harsevoort  8 , Guus J M Janus  8
Affiliations

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

Emmanuelle de Kuijper-Timmermans et al. Oral Dis. 2025 Mar.

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.

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Conflict of interest statement

The authors declare no conflicts of interest.

Figures

FIGURE 1
FIGURE 1
Mean OHIP‐49 scores of the pooled 50‐fold imputations (with 95% CI) per domain for dentate Osteogenesis Imperfecta adults (n = 147; orange bars; mean age: 45.6 ± 14.4 years) and a reference group of 663 Dutch dentate adults without Osteogenesis Imperfecta (mean age: 50.6 ± 12.6 years) (Kieffer, Verrips, and Hoogstraten ; blue bars).
See this image and copyright information in PMC

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