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Correlation between Oral Health-Related Quality of Life and Objectively Measured Oral Health in People with Ehlers-Danlos Syndromes.

ABSTRACT: It is a well-known fact that people with Ehlers-Danlos syndromes (EDS) report a worse oral health-related quality of life (OHRQoL) than the general population. The aim of this study was to examine whether there is a correlation between the subjective OHRQoL and the objectively measured oral health in people with EDS. To determine the subjective OHRQoL, the German version of the 14-item Oral Health Impact Profile (OHIP-14) questionnaire was used. Furthermore, all participants in the study were clinically examined, and the measured parameters were recorded using the Physical Oral Health Index (PhOX). Altogether, records of 46 participants were collected. The median (interquartile range (IQR)) of the OHIP-14 was 17 (23) points and the median of the PhOX was 73 (16) points. However, a statistically significant correlation could not be demonstrated (r = -0.240, p = 0.108). In the observed cohort, for participants who reported having pain often or very often, the OHIP score (median = 24, IQR = 18, p < 0.01) was higher than the score of the group given by participants who never or intermittently experienced pain (median = 8, IQR = 18). In conclusion, patients with EDS showed a reduced OHRQoL, although it was not possible to observe an association between the subjective OHRQoL and the objective oral health. However, participants who often or very often suffer from pain in their tooth, mouth, and jaw areas showed a reduced OHRQoL.

SUBMITTER: Oelerich O

PROVIDER: S-EPMC7664661 | biostudies-literature | 2020 Nov

REPOSITORIES: biostudies-literature

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Correlation between Oral Health-Related Quality of Life and Objectively Measured Oral Health in People with Ehlers-Danlos Syndromes.

Oelerich Ole O Kleinheinz Johannes J Reissmann Daniel R DR Köppe Jeanette J Hanisch Marcel M

International journal of environmental research and public health 20201108 21

It is a well-known fact that people with Ehlers-Danlos syndromes (EDS) report a worse oral health-related quality of life (OHRQoL) than the general population. The aim of this study was to examine whether there is a correlation between the subjective OHRQoL and the objectively measured oral health in people with EDS. To determine the subjective OHRQoL, the German version of the 14-item Oral Health Impact Profile (OHIP-14) questionnaire was used. Furthermore, all participants in the study were cl ...[more]

PMID: 33171578

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Project description:Background and objective: To date, there have only been a few studies on oral health-related quality of life (OHRQoL) of people with Ehlers-Danlos syndromes (EDS) and oral conditions. The aim of this study was, therefore, to analyze the OHRQoL of people with EDS from their own point of view as well as obtain information about their age at the time of the diagnosis, the period of time until diagnosis, and the presence of oral conditions (if any) and their association with oral health quality. Methods: The study was designed as an anonymous questionnaire-based cross-sectional study. We conducted a descriptive analysis of the Oral Health Impact Profile-14 (OHIP-14) scores, age of the participants, age at diagnosis, and the time-period between the first signs of the disease and the diagnosis of EDS. To verify the differences in OHIP-14 scores between patients with and without oral conditions, a Mann-Whitney U test was performed. A multivariate quantile (median) regression analysis was performed to evaluate the effect of different general characteristics (gender, age, and the presence of oral conditions) on the OHIP 14 scores. Furthermore, using a Mann-Whitney U test, the influence of different oral conditions was verified by testing the differences between patients without any oral conditions and patients with a specific diagnosis. Results: A total of 79 evaluable questionnaires from 66 female (83.5%) and 13 male (16.5%) participants were analyzed. On average, after the first condition, it takes 18.36 years before EDS are correctly diagnosed. Oral conditions were described by 69.6% of the participants. The median (interquartile range) OHIP-14 score was eight (ten) points for patients without oral conditions and 19 (15) for patients with oral conditions. The multivariable quantile regression shows a statistical notable association between OHIP-14 score and oral conditions (p < 0.001). OHIP-14 scores for dysgnathia, periodontitis, TMD (Temporomandibular dysfunction), a high-arched palate, malocclusion, and the anomaly of tooth formation were statistical notably different between the participants with and the participants without oral conditions. Conclusions: Long diagnostic pathways seem to be a typical problem in patients with EDS. Oral conditions associated with the underlying disease occurred regularly and showed a negative correlation with OHRQoL.

Project description:The Ehlers-Danlos syndromes (EDS) are a group of heritable connective tissues disorders mainly characterized by skin hyperextensibility, joint hypermobility and generalized tissue fragility. Currently, 14 EDS subtypes each with particular phenotypic features are recognized and are caused by genetic defects in 20 different genes. All of these genes are involved in the biosynthesis and/or fibrillogenesis of collagens at some level. Although great progress has been made in elucidating the molecular basis of different EDS subtypes, the pathogenic mechanisms underlying the observed phenotypes remain poorly understood, and consequentially, adequate treatment and management options for these conditions remain scarce. To date, several animal models, mainly mice and zebrafish, have been described with defects in 14 of the 20 hitherto known EDS-associated genes. These models have been instrumental in discerning the functions and roles of the corresponding proteins during development, maturation and repair and in portraying their roles during collagen biosynthesis and/or fibrillogenesis, for some even before their contribution to an EDS phenotype was elucidated. Additionally, extensive phenotypical characterization of these models has shown that they largely phenocopy their human counterparts, with recapitulation of several clinical hallmarks of the corresponding EDS subtype, including dermatological, cardiovascular, musculoskeletal and ocular features, as well as biomechanical and ultrastructural similarities in tissues. In this narrative review, we provide a comprehensive overview of animal models manifesting phenotypes that mimic EDS with a focus on engineered mouse and zebrafish models, and their relevance in past and future EDS research. Additionally, we briefly discuss domestic animals with naturally occurring EDS phenotypes. Collectively, these animal models have only started to reveal glimpses into the pathophysiological aspects associated with EDS and will undoubtably continue to play critical roles in EDS research due to their tremendous potential for pinpointing (common) signaling pathways, unveiling possible therapeutic targets and providing opportunities for preclinical therapeutic interventions.

Project description:Objective To conduct a systematic review examining the effect of exercise and rehabilitation in people with Ehlers-Danlos syndrome (EDS). Data Sources The following databases were systematically searched: MEDLINE, MEDLINE In-Process/ePubs, Embase, Cochrane Central Register of Controlled Trials, PsycINFO, and Cumulative Index to Nursing and Allied Health. The final time point captured by the search is November 27, 2020. Study Selection Eligible study designs included case-control, case-series, prospective cohort, retrospective cohort, and intervention studies of structured exercise or rehabilitation interventions. Eligible populations included adults (18 years or older) with EDS (all subtypes) and hypermobility spectrum disorders. The search was restricted to articles published in English. Data Extraction Data were extracted by 2 independent reviewers. Risk of bias was assessed using the Physiotherapy Evidence Database (PEDro) scale for randomized controlled trials (RCTs) and Risk Of Bias In Nonrandomized Studies of Interventions (ROBINS-I) for non-RCTs. Reporting quality of RCTs was assessed using the Consolidated Standards for Reporting of Trials statement with the harms extension. Reporting was guided by the Preferred Reporting Items for Systematic Reviews and Meta-Analyses checklist. Data Synthesis The search yielded 10 eligible studies including 330 participants. The study designs included 5 RCTs, 1 cohort, 2 single-arm interventions, 1 retrospective, and 1 feasibility study. All studies showed some improvement in a physical and/or psychological outcome after the intervention period. One adverse event (nonserious) potentially related to the intervention was reported. Of the 5 RCTs, 2 were rated as high quality with low risk of bias using PEDro, and the majority of non-RCTs were rated as critical risk of bias by ROBINS-I. Conclusions The results suggest that exercise and rehabilitation may be beneficial for various physical and psychological outcomes. Adequately powered and rigorous RCTs of exercise and rehabilitation interventions for people with EDS are needed.

Dataset Information

Correlation between Oral Health-Related Quality of Life and Objectively Measured Oral Health in People with Ehlers-Danlos Syndromes.

Publications

Correlation between Oral Health-Related Quality of Life and Objectively Measured Oral Health in People with Ehlers-Danlos Syndromes.

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