Orofacial Features, Oral Health-Related Quality of Life, and Exposure to Bullying in Osteogenesis Imperfecta: A Cross-Sectional Study

Overview

Journal Children (Basel)

Specialty Health Services

Date 2024 Aug 29

PMID 39201835

Authors

Alice Broutin

Jean-Pierre Salles

Valerie Porquet-Bordes

Thomas Edouard

Frederic Vaysse

Emmanuelle Noirrit-Esclassan

Affiliations

Soon will be listed here.

Abstract

Background/objectives: Osteogenesis imperfecta (OI) is a rare genetic disease that is responsible for bone fragility, but also for dental malocclusions and dentinogenesis imperfecta (DI). The aim of this study was to assess whether the severity of dental malocclusion influenced the oral health-related quality of life (OHRQoL) and exposure to bullying in a paediatric OI population compared with a control group.

Methods: Dental and occlusal characteristics were noted during oral and radiographic examination. The severity of malocclusion was assessed using the PAR index. P-CPQ, COHIP(34), and BCS-A questionnaires were used to evaluate, respectively, externally and self-perceived OHRQoL and bullying.

Results: We included 39 patients with a mean age of 11.3 (±4.8 SD) in the OI group, and 45 patients with a mean age of 12.3 (±3.2 SD) in the control group. There were no significant differences between the two groups in terms of occlusal vertical and transverse dimensions. Patients with severe OI, presenting with bone fractures, bones deformities, and short stature, had significantly more anterior ( < 0.05) and posterior openbites ( < 0.05) and more DI ( < 0.05) compared to patients who had moderate or mild OI. Self-perceived OHRQoL was negatively impacted by the disease ( = 0.01), particularly in the domains of oral health ( < 0.05) and self-image ( < 0.001), but not by its severity. Exposure to bullying did not differ significantly between the two groups, although more patients with OI reported being teased (21.4% face to face and 7.1% online vs. 14.6% and 2.4% in the control group).

Conclusion: Interventions for dental malocclusion and oral health in OI patients would help to improve their quality of life and self-image.

References

Cachia Mintoff J, Riddington M, Parekh S . Oral health-related quality of life in children with osteogenesis imperfecta. Eur Arch Paediatr Dent. 2021; 23(2):261-270. PMC: 8994709. DOI: 10.1007/s40368-021-00664-9. View

Rizkallah J, Schwartz S, Rauch F, Glorieux F, Vu D, Muller K . Evaluation of the severity of malocclusions in children affected by osteogenesis imperfecta with the peer assessment rating and discrepancy indexes. Am J Orthod Dentofacial Orthop. 2013; 143(3):336-41. DOI: 10.1016/j.ajodo.2012.10.016. View

Dlesk T, Larimer K . Multimodal Pain Management of Children Diagnosed with Osteogenesis Imperfecta: An Integrative Literature Review. Pain Manag Nurs. 2022; 24(1):102-110. DOI: 10.1016/j.pmn.2022.08.014. View

Dimberg L, Arnrup K, Bondemark L . The impact of malocclusion on the quality of life among children and adolescents: a systematic review of quantitative studies. Eur J Orthod. 2014; 37(3):238-47. DOI: 10.1093/ejo/cju046. View

Malmgren B, Norgren S . Dental aberrations in children and adolescents with osteogenesis imperfecta. Acta Odontol Scand. 2002; 60(2):65-71. DOI: 10.1080/000163502753509446. View

Broder H, Wilson-Genderson M . Reliability and convergent and discriminant validity of the Child Oral Health Impact Profile (COHIP Child's version). Community Dent Oral Epidemiol. 2007; 35 Suppl 1:20-31. DOI: 10.1111/j.1600-0528.2007.0002.x. View

Faith M, Reed G, Heppner C, Hamill L, Tarkenton T, Donewar C . Bullying in medically fragile youth: a review of risks, protective factors, and recommendations for medical providers. J Dev Behav Pediatr. 2015; 36(4):285-301. DOI: 10.1097/DBP.0000000000000155. View

Alania K, Iverieli M, Abashidze N, Gogishvili K, Chigladze T . [Oral cavity features in patients suffering from osteogenesis imperfecta]. Georgian Med News. 2011; (193):34-41. View

OConnell A, Marini J . Evaluation of oral problems in an osteogenesis imperfecta population. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999; 87(2):189-96. DOI: 10.1016/s1079-2104(99)70272-6. View

10.

Ablon J . Personality and stereotype in osteogenesis imperfecta: behavioral phenotype or response to life's hard challenges?. Am J Med Genet A. 2003; 122A(3):201-14. DOI: 10.1002/ajmg.a.20257. View

11.

Friedrich R, Scheuer H, Holtje W . The effect of bisphosphonate medication on orthodontics and orthognathic surgery in patients with osteogenesis imperfecta. GMS Interdiscip Plast Reconstr Surg DGPW. 2019; 8:Doc06. PMC: 6532328. DOI: 10.3205/iprs000132. View

12.

Broutin A, Blanchet I, Canceill T, Noirrit-Esclassan E . Association between Dentofacial Features and Bullying from Childhood to Adulthood: A Systematic Review. Children (Basel). 2023; 10(6). PMC: 10297497. DOI: 10.3390/children10060934. View

13.

Gjorup H, Beck-Nielsen S, Hald J, Haubek D . Oral health-related quality of life in X-linked hypophosphataemia and osteogenesis imperfecta. J Oral Rehabil. 2020; 48(2):160-168. PMC: 7839549. DOI: 10.1111/joor.13114. View

14.

Richmond S, Shaw W, OBrien K, Buchanan I, Jones R, Stephens C . The development of the PAR Index (Peer Assessment Rating): reliability and validity. Eur J Orthod. 1992; 14(2):125-39. DOI: 10.1093/ejo/14.2.125. View

15.

Thomas H, Scott J, Coates J, Connor J . Development and validation of the Bullying and Cyberbullying Scale for Adolescents: A multi-dimensional measurement model. Br J Educ Psychol. 2018; 89(1):75-94. DOI: 10.1111/bjep.12223. View

16.

van Dijk F, Sillence D . Osteogenesis imperfecta: clinical diagnosis, nomenclature and severity assessment. Am J Med Genet A. 2014; 164A(6):1470-81. PMC: 4314691. DOI: 10.1002/ajmg.a.36545. View

17.

Marcal F, Ribeiro E, Costa F, Fonteles C, Teles G, Barros Silva P . Dental alterations on panoramic radiographs of patients with osteogenesis imperfecta in relation to clinical diagnosis, severity, and bisphosphonate regimen aspects: a STROBE-compliant case-control study. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019; 128(6):621-630. DOI: 10.1016/j.oooo.2019.07.001. View

18.

Kragt L, Dhamo B, Wolvius E, Ongkosuwito E . The impact of malocclusions on oral health-related quality of life in children-a systematic review and meta-analysis. Clin Oral Investig. 2015; 20(8):1881-1894. PMC: 5069349. DOI: 10.1007/s00784-015-1681-3. View

19.

Najirad M, Madathil S, Rauch F, Sutton V, Lee B, Retrouvey J . Malocclusion traits and oral health-related quality of life in children with osteogenesis imperfecta: A cross-sectional study. J Am Dent Assoc. 2020; 151(7):480-490.e2. PMC: 7360479. DOI: 10.1016/j.adaj.2020.03.040. View

20.

Nguyen H, Vu D, Nguyen D, Dang Q, Tran V, Le H . Dentinogenesis Imperfecta and Caries in Osteogenesis Imperfecta among Vietnamese Children. Dent J (Basel). 2021; 9(5). PMC: 8144955. DOI: 10.3390/dj9050049. View