Oral Health-Related Quality of Life and Paediatric Osteogenesis Imperfecta

Objectives: Background

Dental concerns are common in children with Osteogenesis Imperfecta (OI), but poorly reported.

Objectives

This study assessed the effect on oral health-related quality of life (OHRQoL).
Methods: * Ethical approval and consent was obtained
* The Child Oral-Health Impact Profile – Short Form (COHIP-SF) was used, plus questions for demographic and qualitative data
* Children aged 8 – 16 years completed COHIP-SF, during appointments at the Highly Specialised Paediatric OI Service at Great Ormond Street Hospital (GOSH), between January – October 2019
* Statistical analysis with Z-tests
Results: Ninety-three children participated (42 female, 51 male, mean age 12.05 years)

Table 1 - Participant perception of severity
Table 2 - OI Type (Participant Response)

COHIP-SF Data
A higher COHIP-SF score indicates better OHRQoL (maximum score, 76). The mean score was 56.1. Children reporting mild OI had higher mean score (58.6) than those reporting severe OI (49.0) [P=0.022]. When comparing mixed (<12 years, n=38) vs permanent dentition (>=12, n=55), no statistically significant difference in OHRQoL was seen [P= 0.88].

The 3 COHIP domains are:
Oral Health Well-Being
Functional Well-Being
Socio-Emotional Well-Being

Perceived severity was associated with significantly lower OHRQoL scores in the Oral Health and Functional Well-Being domains [P=0.047/P=0.039], but not for Socio-Emotional Well-Being [P=0.195].

The presence of Dentinogenesis Imperfecta did not impact OHRQoL score [P=0.564]. However the data was limited, with only 14 patients known to have DI.


Qualitative Data
Common themes were the need for braces (straightness/ gaps between teeth), discolouration, pain and function.
Conclusions: This study confirmed that children with OI have dental concerns, including oral health, functional and socio-emotional well-being. This was related to severity of OI.
Compared to the general child population, children with OI had similar OHRQoL scores.