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巴西患有影响骨骼发育的罕见遗传疾病患者的口腔问题。

Oral Problems in Brazilian Individuals with Rare Genetic Diseases That Affect Skeletal Development.

机构信息

Undergraduate Course in Dentistry, Pontifical Catholic University of Minas Gerais, Belo Horizonte 30535-500, Brazil.

Department of Social and Preventive Dentistry, School of Dentistry, Federal University of Minas Gerais, Belo Horizonte 31270-800, Brazil.

出版信息

Int J Environ Res Public Health. 2024 Sep 18;21(9):1227. doi: 10.3390/ijerph21091227.

DOI:10.3390/ijerph21091227
PMID:39338110
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11431253/
Abstract

The present study aimed to compare the prevalence of oral problems between individuals with rare genetic diseases that affect skeletal development and individuals without rare diseases. A cross-sectional study was conducted with 210 individuals between two and fifty-four years of age: 105 with rare genetic diseases (27 with mucopolysaccharidosis [MPS] and 78 with osteogenesis imperfecta [OI]) and 105 without rare diseases. The rare genetic disease group was recruited from hospital units that provide care for patients with MPS and OI in five states of Brazil, and the other group was recruited from the same location. The participants were examined with regards to malocclusion, dental anomalies, dental caries, and gingivitis. A questionnaire was administered addressing individual, sociodemographic, and behavioral characteristics as well as dental history. A descriptive analysis was performed, followed by unadjusted and adjusted binary logistic regression analyses. The mean age was 14.1 ± 12.2 years. Individuals with a rare disease were 12.9-fold more likely to have some type of oral problem (95% CI: 3.7-44.7) compared to the group without rare diseases. The prevalence of oral problems was higher among Brazilians with MPS and OI compared to normotypical individuals.

摘要

本研究旨在比较患有影响骨骼发育的罕见遗传疾病的个体和没有罕见疾病的个体之间口腔问题的患病率。采用横断面研究方法,对 210 名年龄在 2 至 54 岁之间的个体进行了研究:105 名患有罕见遗传疾病(27 名患有黏多糖贮积症[MPS],78 名患有成骨不全症[OI])和 105 名没有罕见疾病的个体。罕见遗传疾病组是从巴西五个州为 MPS 和 OI 患者提供护理的医院单位招募的,而另一组则从同一地点招募。研究对象接受了错颌畸形、牙齿异常、龋齿和牙龈炎检查。采用问卷对个体、社会人口统计学和行为特征以及牙科史进行了评估。进行了描述性分析,随后进行了未调整和调整后的二元逻辑回归分析。平均年龄为 14.1 ± 12.2 岁。与无罕见疾病组相比,患有罕见疾病的个体发生某种口腔问题的可能性高 12.9 倍(95%CI:3.7-44.7)。与正常体型个体相比,患有 MPS 和 OI 的巴西人患口腔问题的患病率更高。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cad9/11431253/f3f75e3256f6/ijerph-21-01227-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cad9/11431253/f3f75e3256f6/ijerph-21-01227-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cad9/11431253/f3f75e3256f6/ijerph-21-01227-g001.jpg

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本文引用的文献

1
Consideration of oral health in rare disease expertise centres: a retrospective study on 39 rare diseases using text mining extraction method.考虑罕见病专业中心的口腔健康:使用文本挖掘提取方法对 39 种罕见病的回顾性研究。
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Rare Disorders: Diagnosis and Therapeutic Planning for Patients Seeking Orthodontic Treatment.罕见疾病:寻求正畸治疗患者的诊断与治疗规划
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Mother's sense of coherence and dental characteristics in children and adolescents with osteogenesis imperfecta: A paired study.母亲的综合感知能力与成骨不全症患儿和青少年的牙齿特征:配对研究。
Spec Care Dentist. 2021 Mar;41(2):170-177. doi: 10.1111/scd.12560. Epub 2021 Jan 13.
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Int J Environ Res Public Health. 2020 Dec 28;18(1):139. doi: 10.3390/ijerph18010139.
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Oral health of Brazilian individuals with mucopolyssaccaridosis.巴西黏多糖贮积症患者的口腔健康状况。
Eur Arch Paediatr Dent. 2021 Feb;22(1):13-19. doi: 10.1007/s40368-020-00508-y. Epub 2020 Jan 31.
8
From the search for diagnosis to treatment uncertainties: challenges of care for rare genetic diseases in Brazil.从诊断探索到治疗不确定性:巴西罕见遗传病护理面临的挑战
Cien Saude Colet. 2019 Sep 26;24(10):3637-3650. doi: 10.1590/1413-812320182410.01612019. eCollection 2019.
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Assessing a possible vulnerability to dental caries in individuals with rare genetic diseases that affect the skeletal development.评估罕见的骨骼发育相关遗传疾病个体的龋齿易感性。
Orphanet J Rare Dis. 2019 Jun 18;14(1):145. doi: 10.1186/s13023-019-1114-5.
10
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.患有罕见病的人群的口腔健康相关生活质量与其对德国联邦共和国卫生系统中牙科护理满意度之间的关联。
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