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

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  • Marcel Hanisch

    (Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany)

  • Sabrina Wiemann

    (Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany)

  • Susanne Jung

    (Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany)

  • Johannes Kleinheinz

    (Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany)

  • Lauren Bohner

    (Department of Cranio-Maxillofacial Surgery, Research Unit Rare Diseases with Orofacial, Manifestations (RDOM), University Hospital Münster, Albert-Schweitzer-Campus 1, Building W 30, D-48149 Münster, Germany)

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.

Suggested Citation

  • Marcel Hanisch & Sabrina Wiemann & Susanne Jung & Johannes Kleinheinz & Lauren Bohner, 2018. "Oral Health-Related Quality of Life in People with Rare Hereditary Connective Tissue Disorders: Marfan Syndrome," IJERPH, MDPI, vol. 15(11), pages 1-8, October.
    • Handle: RePEc:gam:jijerp:v:15:y:2018:i:11:p:2382-:d:178691
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    References listed on IDEAS

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    1. Roll, Kathrin, 2012. "The influence of regional health care structures on delay in diagnosis of rare diseases: The case of Marfan Syndrome," Health Policy, Elsevier, vol. 105(2), pages 119-127.
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    Cited by:

    1. Mohammad Khursheed Alam & Ahmed Ali Alfawzan & Deepti Shrivastava & Kumar Chandan Srivastava & Haytham Jamil Alswairki & Samir Mussallam & Huda Abutayyem & Naseer Ahmed, 2022. "Oral Health Status in Marfan Syndrome: A Systematic Review and Meta-Analysis of 353 Cases," IJERPH, MDPI, vol. 19(9), pages 1-13, April.
    2. Annemarie Kühne & Johannes Kleinheinz & Jochen Jackowski & Jeanette Köppe & Marcel Hanisch, 2020. "Study to Investigate the Knowledge of Rare Diseases among Dentists, Orthodontists, Periodontists, Oral Surgeons and Craniomaxillofacial Surgeons," IJERPH, MDPI, vol. 18(1), pages 1-15, December.
    3. Marcel Hanisch & Sonja Sielker & Susanne Jung & Johannes Kleinheinz & Lauren Bohner, 2019. "Self-Assessment of Oral Health-Related Quality of Life in People with Ectodermal Dysplasia in Germany," IJERPH, MDPI, vol. 16(11), pages 1-9, May.

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