Cachia Mintoff, Jasmine May; (2020) Oral Health Related Quality of Life in Children with Osteogenesis Imperfecta. Doctoral thesis (D.Dent), UCL (University College London).

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Abstract

Background: Osteogenesis Imperfecta (OI) is a genetic condition resulting from a mutation in the genes involved in the modification or biosynthesis of collagen. The study involved two parts and took place in the OI department at Great Ormond Street Hospital (GOSH). Aims: To investigate the oral health related quality of life of children aged eight to sixteen-years-old with OI. Service Evaluation: Methodology: Five questions designed to assess if children with OI are receiving adequate dental care. Results: 86 participants (43% female, 57% males, mean age 8.31) 78 (91%) of children were receiving some form of dental care. 49 (57%) of participants reported one or more dental concerns. 27/41 (66%) of the mixed dentition group reported at least one type of concern; most frequently appearance [18/41 (44%)]. Conclusions: Dental concerns in children with OI are present. It is important to have ongoing dental care to address concerns. Questionnaire: Methodology: Ethical approval and consent were obtained The Child Oral-Health Impact Profile – Short Form (COHIP-SF) was used, with demographic and qualitative data Children aged 8 – 16 years participated between January – October 2019 Statistical analysis with parametric and non-parametric tests Results 106 children participated (44 female, 62 male, mean age 11.93 years) COHIP-SF Data A higher COHIP-SF score indicates better OHRQoL (maximum score, 76). The median score was 59. Children reporting mild OI had higher median score (62) than those reporting severe OI (55) [P=0.087]. When comparing mixed (<12 years, n=46) vs permanent dentition (>=12, n=60), no statistically significant difference in OHRQoL was seen [P= 0.977]. The 3 COHIP-SF domains are Oral Health, Functional and Socio-Emotional Well-Being. There was no significant difference between severities for each domain. Perceived severity was not associated with significantly lower OHRQoL scores for females [P=0.125] or males [P=0.406]. Limited data on the presence of Dentinogenesis Imperfecta did not impact overall score [P=0.109] but was significant in the Oral Health domain [P=0.033]. 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.

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