Evaluierung der mundgesundheitsbezogenen Lebensqualität bei seltenen Erkrankungen mit oraler Beteiligung

Hanisch, Marcel; Jung, Susanne; Kleinheinz, Johannes

doi:10.1055/a-0592-7039

Cited by 7 publications

(3 citation statements)

References 6 publications

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“…Rare diseases have a negative impact on general health-related quality of life for patients [21]. Several studies suggest that rare diseases can also have an undesirable impact on the oral health-related quality of life due to associated abnormal dental and craniofacial findings [8–11, 22–24]. Our study demonstrates for the first time that patients with LDS have worse OHRQoL when compared to both unaffected family members and the general U.S. population [4].…”

Section: Discussionmentioning

confidence: 99%

Oral health-related quality of life in Loeys-Dietz syndrome, a rare connective tissue disorder: an observational cohort study

Nguyen

Duverger

Mishra

et al. 2019

Orphanet J Rare Dis

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BackgroundLoeys-Dietz syndrome (LDS) is a rare connective tissue disorder whose oral manifestations and dental phenotypes have not been well-characterized. The aim of this study was to explore the influence of oral manifestations on oral health-related quality of life (OHRQoL) in LDS patients.Material and methodsLDS subjects were assessed by the craniofacial team at the National Institutes of Health Clinical Center Dental Clinic between June 2015 and January 2018. Oral Health Impact Profile (OHIP-14) questionnaire, oral health self-care behavior questionnaire and a comprehensive dental examination were completed for each subject. OHRQoL was assessed using the OHIP-14 questionnaire with higher scores corresponding to worse OHRQoL. Regression models were used to determine the relationship between each oral manifestation and the OHIP-14 scores using a level of significance of p ≤ 0.05.ResultsA total of 33 LDS subjects (51.5% female) aged 3–57 years (19.6 ± 15.1 years) were included in the study. The OHIP-14 scores (n = 33) were significantly higher in LDS subjects (6.30 [SD 6.37]) when compared to unaffected family member subjects (1.50 [SD 2.28], p < 0.01), and higher than the previously reported scores of the general U.S. population (2.81 [SD 0.12]). Regarding oral health self-care behavior (n = 32), the majority of LDS subjects reported receiving regular dental care (81%) and maintaining good-to-excellent daily oral hygiene (75%). Using a crude regression model, worse OHRQoL was found to be associated with dental hypersensitivity (β = 5.24; p < 0.05), temporomandibular joints (TMJ) abnormalities (β = 5.92; p < 0.01), self-reported poor-to-fair oral health status (β = 6.77; p < 0.01), and cumulation of four or more oral manifestations (β = 7.23; p < 0.001). Finally, using a parsimonious model, self-reported poor-to-fair oral health status (β = 5.87; p < 0.01) and TMJ abnormalities (β = 4.95; p < 0.01) remained significant.ConclusionsThe dental hypersensitivity, TMJ abnormalities, self-reported poor-to-fair oral health status and cumulation of four-or-more oral manifestations had significant influence on worse OHRQoL. Specific dental treatment guidelines are necessary to ensure optimal quality of life in patients diagnosed with LDS.

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Section: Discussionmentioning

confidence: 99%

Oral health-related quality of life in Loeys-Dietz syndrome, a rare connective tissue disorder: an observational cohort study

Nguyen

Duverger

Mishra

et al. 2019

Orphanet J Rare Dis

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“…Our research group showed previously that the oral health-related quality of life (OHRQoL) is affected by rare diseases presenting oral manifestations [14,15,16,17].…”

Section: Introductionmentioning

confidence: 99%

Self-Assessment of Oral Health-Related Quality of Life in People with Ectodermal Dysplasia in Germany

Hanisch

Sielker

Jung

et al. 2019

IJERPH

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Background: Ectodermal dysplasia describes a heterogeneous group of hereditary, congenital malformations involving developmental dystrophies of ectodermal structures. The aim of this study was to analyse the oral health-related quality of life (OHRQoL) in people with ectodermal dysplasia and to evaluate the influence of different variables. Methods: The study was designed as an anonymous epidemiological survey study among people with ectodermal dysplasia to evaluate oral symptoms, satisfaction with the health system and their respective OHRQoL using the validated German version of the OHIP-14 (Oral Health Impact Profile) questionnaire. Results: When asked about oral symptoms, 110 of the participants provided responses, of which 109 (99.09%) described oral symptoms. The average age of the female participants at the time of diagnosis was 17.02 years (range: 0 to 48 years), the average age of men was 5.19 years (range: 0 to 43 years). The average OHIP-14 overall score for female participants was 12.23 points (SD: 12.39), for male participants an average OHIP score of 11.79 points was recorded (SD: 11.08 points). Difficulty in finding a dentist (p = 0.001), and the dissatisfaction with the health system (p = 0.007) showed a negative impact on the OHRQoL. Conclusion: People with ectodermal dysplasia rate their OHRQoL worse than is usually prevalent in the normal German population (4.09 points); women are diagnosed with “ectodermal dysplasia” later than men. Participants who reported difficulties in finding a dentist for treatment exhibited higher OHIP values. Likewise, dissatisfaction with the health system demonstrated a negative impact on the oral health-related quality of life.

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“…Although few studies have assessed this outcome from the perspective of people with rare diseases [9], the existing literature suggests this population presents a reduced oral health-related quality of life [9,10,11,12]. In this respect, oral manifestations of rare diseases may be negatively associated with their oral health perception [12].…”

Section: Introductionmentioning

confidence: 99%

Association between Oral Health-Related Quality of Life in People with Rare Diseases and Their Satisfaction with Dental Care in the Health System of the Federal Republic of Germany

Hanisch

Wiemann

Bohner

et al. 2018

IJERPH

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Background: The aim of this study was to examine the current dental care situation in Germany from the perspective of those affected by a rare disease, especially concerning their satisfaction with the German dental health care system, and thus assess the relationship between their perspective and their oral health-related quality of life (OHRQoL). Methods: A questionnaire regarding their experiences with the dental assistance and the health care system, such as the OHIP-14, was sent to the member associations of the organization of self-help groups for rare diseases ACHSE e.V. The correlation between OHIP-14 values and patient’s perspective was statistically analyzed by the non-parametric Tau de Kendall test (p < 0.05). Results: There was a statistically significant correlation between the OHIP score and the patient’s perspective regarding dental assistance and health care system (p < 0.05). For those surveyed who were satisfied with the support of the health care system, an average OHIP score of 8.54 ± 10.45 points (range: 0–48) was determined. The group that did not feel sufficiently supported by the health care system had an average OHIP score of 16.07 ± 13.43 points (range: 0–56). Discussion: The majority of respondents with rare diseases are dissatisfied with the German health care system and its support with regard to dental care.

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Evaluierung der mundgesundheitsbezogenen Lebensqualität bei seltenen Erkrankungen mit oraler Beteiligung

Cited by 7 publications

References 6 publications

Oral health-related quality of life in Loeys-Dietz syndrome, a rare connective tissue disorder: an observational cohort study

Oral health-related quality of life in Loeys-Dietz syndrome, a rare connective tissue disorder: an observational cohort study

Self-Assessment of Oral Health-Related Quality of Life in People with Ectodermal Dysplasia in Germany

Association between Oral Health-Related Quality of Life in People with Rare Diseases and Their Satisfaction with Dental Care in the Health System of the Federal Republic of Germany

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