Oral Health-Related Quality of Life in People with Rare Hereditary Connective Tissue Disorders: Marfan Syndrome

Int J Environ Res Public Health. 2018 Oct 27;15(11):2382. doi: 10.3390/ijerph15112382.

Abstract

Background: The aim of this study was to analyze data on oral health-related quality of life (OHRQoL) in people with Marfan syndrome and to obtain information on the diagnosis period, orthodontic treatment, and oral symptoms.

Methods: A questionnaire was developed consisting of open questions and the standardized German version of the OHIP-14 (Oral Health Impact Profile) questionnaire for the evaluation of OHRQoL. The age of diagnosis, time period from the first signs of the disease to diagnosis, and OHIP-values were compared between male and female participants. Additionally, the OHIP-values between participants who were orthodontically treated and those who were not treated were assessed. The statistical analysis was performed using the Mann⁻Whitney test with a significance level at p = 0.05.

Results: A total of 51 questionnaires were evaluated, which included 34 female and 17 male participants. Overall, 84% of respondents reported oral symptoms. Male respondents tended to diagnose the disease earlier (p = 0.00), with a smaller period between the first symptom and the diagnosis (p = 0.04). The OHIP-14 score was gender-neutral at 13.65 ± 13.53 points.

Conclusion: In Marfan syndrome, many years (12.01 ± 11.61) elapse between the onset of first symptoms and correct diagnosis of the disease. People with Marfan syndrome have a worse OHRQoL than do the general population.

Keywords: Marfan; OHIP-14; OHRQoL; oral health-related quality of life; patient reported outcome; rare diseases.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Dental Care / statistics & numerical data*
  • Female
  • Humans
  • Male
  • Marfan Syndrome / diagnosis
  • Marfan Syndrome / psychology*
  • Middle Aged
  • Oral Health / statistics & numerical data*
  • Quality of Life / psychology*
  • Young Adult