The state of oral health in patients with haemophilia in the Netherlands

Mulders, Greta; Verseveld, Hanneke van; Geer, Joyce van der; Wolvius, Eppo B.; Leebeek, Frank W.G.

doi:10.1111/hae.14719

Cited by 5 publications

(16 citation statements)

References 27 publications

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“…The prevalence of TMD in individual studies varied from 2% to 77%, and controls in the individual studies varied from 2% to 14%. Among the included studies, only clinical signs, symptoms and history were taken into consideration in four studies, 13,19,21,22 Diagnostic criteria for TMD were taken into consideration in the article by Yenel et al 2022 15 . The Research Diagnostic Criteria for Temporomandibular Disorders (RDC/TMD) were taken into consideration in two articles 14,20 .…”

Section: Resultsmentioning

confidence: 99%

“…This led to 11 articles included for full-text review from which four articles were excluded [25][26][27][28] Table 1: List of excluded articles and reasons for exclusion. After a stringent full-text review, seven articles were included for final qualitative analysis, [13][14][15][19][20][21][22] Out of these final seven articles, four articles were included for quantitative analysis as control group was present in these four articles (Figure 3). 15,19,20,22 Sno Title Reason for exclusion…”

Section: Study Characteristicsmentioning

confidence: 99%

“…Seven articles were included in the qualitative analysis, Most of the studies had a control group (n = 5), 14,15,19,20,22 and only two studies had no control group. 13,21 The data of the two articles without the control group were excluded from the data pooling for prevalence, so a total of 194 individuals with bleeding disorders (BD) and 376 controls were included in the review. In the BD/haemophilia group, 24.74% (48 subjects out of 194) had some clinical history, symptom or sign, an imaging-related finding suggesting a temporomandibular disorder (TMD).…”

Section: Main Findingsmentioning

confidence: 99%

“…Pertaining to previous articles published in the literature, few authors report a positive association between presence of bleeding disorders (BD) and increased prevalence of temporomandibular disorders (TMD) 13–18 . Few other studies reported no significant association between increased prevalence of TMD in BD patients 19–24 . There is no systematic review in the literature in this regard, the current systematic review intends to evaluate the association between bleeding disorders and TMD and aims to address the question ‘Is there increased tendency of TMD in patients with bleeding disorders’.…”

Section: Introductionmentioning

confidence: 96%

“…[13][14][15][16][17][18] Few other studies reported no significant association between increased prevalence of TMD in BD patients. [19][20][21][22][23][24] There is no systematic review in the literature in this regard, the current systematic review intends to evaluate the association between bleeding disorders and TMD and aims to address the question 'Is there increased tendency of TMD in patients with bleeding disorders'.…”

Section: Introductionmentioning

confidence: 99%

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Prevalence of temporomandibular disorders (TMD) in bleeding disorders: A systematic review with meta‐analysis

Minervini,

Marrapodi,

Tirupathi

et al. 2023

J of Oral Rehabilitation

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BackgroundHaemophilia A, B and von Willebrand disease are the most common bleeding disorders. There is an increased tendency of spontaneous bleeding into joints resulting in intra‐articular infection. It is believed that Temporomandibular Joint (TMJ) can be affected in a similar manner which can further lead to limited mouth opening and ankylosis. The association between bleeding disorders (BD) and development of temporomandibular disorders (TMD) is poorly understood. This systematic review intends to evaluate the association of TMD in individuals with inherited bleeding disorders as compared to healthy controls.MethodsPubMed, Ovid SP and Google Scholar were searched for articles published between the times of inception to 1 May 2023. All the articles were subjected to Population, Exposure, Comparison and Outcome model (PECO) based on which inclusion and exclusion criteria were applied. Participants (P) is children, adults or adolescents; Exposure (E) is children, adults or adolescents with a diagnosis of Haemophilia or bleeding disorder (BD); Comparator (C) is age and gender‐matched healthy controls who do not have Haemophilia or bleeding disorder; Outcome (O) is prevalence of any signs or symptoms (clinical, radiographic) that is suggestive of temporomandibular disorder (TMD). Studies showing the prevalence of TMD are included for qualitative analysis. Only the studies which provided data of the prevalence of TMD in both the groups (BD and healthy controls) were included in the quantitative analysis. TMD diagnosis can be by clinical signs and symptoms, radiographic criteria, Diagnostic Criteria for Temporomandibular Disorders (DC/TMD), The Research Diagnostic Criteria for Temporomandibular Disorders (RDC/TMD) or any other imaging criteria (MRI). We have set the exclusion criteria as articles without a control group, diagnostic sensitivity studies, case reports and systematic reviews and narrative reviews. The software Review Manager version 5.4 (Cochrane Collaboration) was used to perform the pooled analysis. We measured the risk ratio (RR) between the two groups (BD and healthy controls) for the outcome TMD.ResultsA total of seven studies are included for qualitative analysis of data. The age of the population (BD and control) in the included studies ranged between 2 and 57 years. The prevalence of TMD in individual studies varied from 2% to 77%, and controls in the individual studies varied from 2% to 14%. Among the included studies, only clinical signs, symptoms and history were taken into consideration in four studies. Four studies were included for meta‐analysis, the pooled result of the four studies suggests there is no significant difference in the prevalence of TMD in BD and control group (p value = .11, RR 2.19; 95% CI [0.84, 5.73]).ConclusionThis systematic review and meta‐analysis elicits no association between bleeding disorders and increased prevalence of TMD.

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Section: Resultsmentioning

confidence: 99%

Section: Study Characteristicsmentioning

confidence: 99%

Section: Main Findingsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 96%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Prevalence of temporomandibular disorders (TMD) in bleeding disorders: A systematic review with meta‐analysis

Minervini,

Marrapodi,

Tirupathi

et al. 2023

J of Oral Rehabilitation

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Oral health in individuals with bleeding disorders: A systematic review and meta‐analysis

Bhadauria,

Purohit,

Nilima

et al. 2024

Haemophilia

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BackgroundIndividuals with bleeding disorders have been reported to have a number of oral health issues due to varying conditions. A comprehensive evaluation of the different oral health conditions has not been carried out in the past. This systematic review and meta‐analysis was carried out to collate and critically analyse existing research, and provide a comprehensive overview of the current state of knowledge on oral health.MethodsA comprehensive search was conducted in electronic databases, including PubMed, Scopus and Embase, in October 2023. No restriction on time frame or language was applied. The risk of bias for cross‐sectional studies was assessed using the Agency for Healthcare Research and Quality (AHRQ) tool, and case control studies were assessed using the New Castle Ottawa Scale (NOS).ResultsTwenty‐two articles were included in the final analysis with a total sample size of 2422 subjects. Of the 22 articles assessed, nine quantitative assessments were included in the Meta analysis. Pooled data analysis was carried out. A total of 13 studies reported medium risk whereas the remaining nine studies showed low risk of bias. The weighted mean DMFT scores in individuals with bleeding disorders were found to be 2.43 [0.62. 4.24], mean dmft was 2.79 [1.05, 4.53] and mean OHI‐S was reported to be 1.79 [1.00, 2.57], respectively.ConclusionThe findings emphasize that these individuals have fair oral hygiene and lower dmft/DMFT scores. Oral bleeding emerged as an important oral health component to be cautiously dealt with particularly during the stages of exfoliation/shedding.

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A 360-degree perspective on adeno-associated virus (AAV)-based gene therapy for haemophilia: Insights from the physician, the nurse and the patient

Miesbach,

Mulders,

Breederveld

et al. 2024

Orphanet J Rare Dis

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Background Adeno-associated virus (AAV)-based gene therapy for haemophilia has advanced substantially in the last 13 years; recently, three products have received approvals from regulatory authorities. Although the impact on quality of life seems promising, some limitations remain, such as the presence of pre-existing anti-AAV neutralising antibodies and the occurrence of hepatotoxicity. This review follows the CSL Behring-sponsored symposium at the 27th Congress of the European Hematology Association (EHA) 2022 that examined the haemophilia gene therapy process from a 360-degree multidisciplinary perspective. Here, the faculty (haematologist, nurse and haemophilia patient) summarised their own viewpoints from the symposium, with the aim of highlighting the key considerations required to engage with gene therapy effectively, for both patients and providers, as well as the importance of multidisciplinary collaboration, including with industry. Results When considering these new therapies, patients face a complex decision-making process, which includes whether gene therapy is right for them at their current stage of life. The authors agreed that collaboration and tailored education across the multidisciplinary team (including patients and their carers/families), starting early in the process and continuing throughout the long-term follow-up period, is key for the success of gene therapy. Additionally, patient expectations, which may surround eligibility, follow-up requirements and treatment outcomes, should be continually explored. During these ongoing discussions, transparent communication of the unknown factors, such as anticipated clotting factor levels, long-term factor expression and safety, and psychological changes, is critical. To ensure efficiency and comprehensiveness, clearly‑defined protocols should outline the whole process, which should include the recording and management of long-term effects. Conclusion In order to engage effectively, both patients and providers should be familiar with these key considerations prior to their involvement with the haemophilia gene therapy process. The future after the approval of haemophilia gene therapies remains to be seen and real-world evidence is eagerly awaited.

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The state of oral health in patients with haemophilia in the Netherlands

Cited by 5 publications

References 27 publications

Prevalence of temporomandibular disorders (TMD) in bleeding disorders: A systematic review with meta‐analysis

Prevalence of temporomandibular disorders (TMD) in bleeding disorders: A systematic review with meta‐analysis

Oral health in individuals with bleeding disorders: A systematic review and meta‐analysis

A 360-degree perspective on adeno-associated virus (AAV)-based gene therapy for haemophilia: Insights from the physician, the nurse and the patient

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