ArticleOrphanet journal of rare diseases2019
Assessing a possible vulnerability to dental caries in individuals with rare genetic diseases that affect the skeletal development.
Article in Orphanet journal of rare diseases, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.
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14 citing papers in PubMed, 1 synthesis or guideline pooled it, 24 citations in OpenAlex.
- Dental anomalies in individuals with osteogenesis imperfecta: a systematic review and meta-analysis of prevalence and comparative studies.Journal of applied oral science : revista FOB · 2023Pooled it
- Dental Health in Pediatric Patients With Different Types of Mucopolysaccharidosis: Retrospective Cross-Sectional Study.JMIR pediatrics and parenting · 2026Article
- Mapping Dental Care for Children and Adolescents With Rare Diseases: A Brazilian Multicentre Study.Community dentistry and oral epidemiology · 2026Article
- Oral health-related quality of life in patients with osteogenesis imperfecta in Taiwan.Journal of dental sciences · 2026Article
- Dentomaxillofacial abnormalities associated with rare bone disease in two pediatric populations from southern Europe and East Africa.Orphanet journal of rare diseases · 2025Article
- Children and Adolescents with Mucopolysaccharidosis and Osteogenesis Imperfecta: The Dentistry on the Multiprofessional Team.Journal of personalized medicine · 2025Article
- Bone disease and oromaxillofacial disorders: a cross- sectional study in a Tanzanian pediatric population.Orphanet journal of rare diseases · 2025Article
- Impaired salivary gland function in children with osteogenesis imperfecta: a case-control study.Clinical oral investigations · 2024Article
- Oral Problems in Brazilian Individuals with Rare Genetic Diseases That Affect Skeletal Development.International journal of environmental research and public health · 2024Article
- Dental Profile of Brazilian Patients with Rare Skeletal Genetic Disorders: Clinical Features and Associated Factors.Healthcare (Basel, Switzerland) · 2024Article
- Oral conditions of children with microcephaly associated with congenital Zika syndrome: a cross-sectional study.Brazilian oral research · 2024Article
- Best practice guidelines in managing the craniofacial aspects of skeletal dysplasia.Orphanet journal of rare diseases · 2021Article
- Dental and maxillomandibular incidental findings in panoramic radiography among individuals with mucopolysaccharidosis: a cross-sectional study.Journal of applied oral science : revista FOB · 2021Article
- Rare genetic diseases affecting skeletal development and oral health disparities among children and adolescents: a pathway analysis.International dental journal · 2020Article
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Authors and funding
6 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundIndividuals diagnosed with a rare genetic disease that affects skeletal development often have physical limitations and orofacial problems that exert an impact on oral health. The aim of the present study was to analyze the possible vulnerability to dental caries in individuals with rare genetic diseases that affect skeletal development.
methodsA paired cross-sectional study was carried out with a sample of 140 individuals [70 with rare genetic diseases affecting skeletal development: mucopolysaccharidosis (MPS) (n = 29) and osteogenesis imperfecta (OI) (n = 41) and 70 without rare diseases] and their parents/caregivers. The participants in the first group were recruited from two reference hospitals specialized in rare genetic diseases in the city of Belo Horizonte, Brazil. All participants were examined for the evaluation of breathing type, malocclusion, dental anomalies, oral hygiene and dental caries. The parents/caregivers answered a structured questionnaire addressing the individual/behavioral characteristics and medical/dental history of the participants. Statistical analysis involved the chi-square test and multiple logistic regression analysis for the dependent variable (dental caries) (α = 5%). This study received approval from the Human Research Ethics Committee of the Universidade Federal de Minas Gerais.
resultsThe mean age of the individuals was 10.34 ± 6.55 years (median: 9.50 years). Individuals with inadequate oral hygiene were 4.70-fold more likely to have dental caries (95% CI: 2.13-10.40) and those with the rare genetic diseases (MPS/OI) were 2.92-fold more likely to have dental caries (95% CI: 1.38-6.17).
conclusionIndividuals with inadequate oral hygiene and those with MPS and OI had a greater chance of belonging to the group with dental caries. Based on the present findings, individuals with the rare genetic diseases may be considered vulnerable to caries.
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