The impact of osteogenesis imperfecta severity on oral health-related quality of life in Spain: a cross-sectional study

Orphanet J Rare Dis. 2024 Mar 8;19(1):108. doi: 10.1186/s13023-024-03096-y.

Abstract

Background: Osteogenesis imperfecta (OI) affects dental and craniofacial development; therefore, it can influence oral health-related quality of life (OHRQoL). The objective of this study was to explore the influence of the severity of OI on OHRQoL in adults older than eighteen years residing in Spain.

Method: Adults with OI were recruited from the Spanish Association of Crystal Bone (AHUCE) foundation. OHRQoL was evaluated using the Spanish version of the Oral Health Impact Profile questionnaire (OHIP-14sp), oral hygiene habits, and a dental care survey. Clinical and radiological dental examinations were performed to evaluate the patients' oral conditions.

Results: A total of 65 adults (n = 46 females) aged between nineteen and sixty-two years who were diagnosed with OI and classified as type I, III, and IV (n = 20, 14, and 31, respectively) participated in this research. The total OHIP-14sp scores were significantly greater (worse) for type III (23 [SD = 10]) and type IV (21.4 [SD = 12]) than for type I (13.8 [SD = 6]) (P < 0.05). The negative impact of OHRQoL was due to the association of type III OI with all domains except for the handicap domain, while type IV OI was associated with the physical disability, social disability, and handicap domains (P < 0.05 for all).

Conclusion: The severity of OI negatively impacted OHRQoL in adults. This association was statistically significant.

Keywords: Oral health impact profile; Oral health-related quality of life; Osteogenesis Imperfecta.

MeSH terms

  • Adult
  • Cross-Sectional Studies
  • Female
  • Humans
  • Middle Aged
  • Mouth Diseases*
  • Osteogenesis Imperfecta* / complications
  • Quality of Life
  • Spain
  • Surveys and Questionnaires
  • Young Adult