Oro-Dental Characteristics in Patients With Adult-Onset Hypophosphatasia Compared to a Healthy Control Group-A Case-Control Study.

IF 3.1 3区 医学 Q1 DENTISTRY, ORAL SURGERY & MEDICINE Journal of oral rehabilitation Pub Date : 2024-10-11 DOI:10.1111/joor.13878
Freja Fribert Jørgensen, Xenia Hermann, Nicola Hepp, Liselotte Sonnesen
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Abstract

Background: Hypophosphatasia (HPP) is a rare inherited disease that affects multiple organ systems including bone and teeth. Limited knowledge exists on dental and oral health in patients with adult-onset HPP (aHPP).

Objective: The aim of this study was to investigate oro-dental characteristics in patients with aHPP compared to healthy controls.

Methods: This case-control study included 20 patients with aHPP compared to 31 healthy controls. Oro-dental manifestations were examined by standardised interviews, clinical examinations as well as radiological registrations on panoramic radiograph (OP) and cone-beam computed tomography (CBCT) scans.

Results: The subjective experience of tooth fractures (p = 0.010), caries in permanent teeth (p = 0.032) and early loss of permanent teeth (p = 0.002) was significantly higher in patients with aHPP compared to the controls. In the aHPP group, the presence of specific teeth (p ≤ 0.045) and attrition of 11 were significantly lower (p = 0.012) compared to the controls. Opacity of a few teeth (p ≤ 0.049), presence of denticles (p = 0.024), the distance between the enamel-cement junction (CEJ) and the marginal bone level at specific sites (p ≤ 0.021) and crown height of 11 (p = 0.017) were significantly higher in patients with aHPP than in healthy controls.

Conclusion: The results indicate that patients with aHPP have a subjective experience of having poorer dental health. Loss of permanent teeth, less attrition, tooth opacities, denticles and larger distance between CEJ and marginal bone level are possible oro-dental findings in patients with aHPP.

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与健康对照组相比,成人发病型低磷酸盐血症患者的口腔特征--一项病例对照研究。
背景:低磷酸盐血症(HPP)是一种罕见的遗传性疾病,会影响包括骨骼和牙齿在内的多个器官系统。有关成人型 HPP(aHPP)患者牙齿和口腔健康的知识有限:本研究旨在调查与健康对照组相比,aHPP 患者的口腔特征:这项病例对照研究包括 20 名 aHPP 患者和 31 名健康对照者。通过标准化访谈、临床检查以及全景X光片(OP)和锥束计算机断层扫描(CBCT)的放射学登记,对口腔表现进行了研究:结果:与对照组相比,aHPP 患者对牙齿骨折(p = 0.010)、恒牙龋坏(p = 0.032)和恒牙早期脱落(p = 0.002)的主观感受明显较高。在 aHPP 组中,与对照组相比,特定牙齿的存在率(p ≤ 0.045)和 11 颗牙齿的损耗率明显较低(p = 0.012)。与健康对照组相比,aHPP 患者少数牙齿的不透明度(p ≤ 0.049)、牙列的存在(p = 0.024)、特定部位的釉质-水泥交界处(CEJ)与边缘骨水平之间的距离(p ≤ 0.021)和 11 的牙冠高度(p = 0.017)均明显较高:结果表明,aHPP 患者的主观感觉是牙齿健康状况较差。aHPP 患者可能会出现恒牙缺失、牙齿损耗较少、牙齿不透明、牙列和 CEJ 与边缘骨水平之间距离较大等口腔发现。
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来源期刊
Journal of oral rehabilitation
Journal of oral rehabilitation 医学-牙科与口腔外科
CiteScore
5.60
自引率
10.30%
发文量
116
审稿时长
4-8 weeks
期刊介绍: Journal of Oral Rehabilitation aims to be the most prestigious journal of dental research within all aspects of oral rehabilitation and applied oral physiology. It covers all diagnostic and clinical management aspects necessary to re-establish a subjective and objective harmonious oral function. Oral rehabilitation may become necessary as a result of developmental or acquired disturbances in the orofacial region, orofacial traumas, or a variety of dental and oral diseases (primarily dental caries and periodontal diseases) and orofacial pain conditions. As such, oral rehabilitation in the twenty-first century is a matter of skilful diagnosis and minimal, appropriate intervention, the nature of which is intimately linked to a profound knowledge of oral physiology, oral biology, and dental and oral pathology. The scientific content of the journal therefore strives to reflect the best of evidence-based clinical dentistry. Modern clinical management should be based on solid scientific evidence gathered about diagnostic procedures and the properties and efficacy of the chosen intervention (e.g. material science, biological, toxicological, pharmacological or psychological aspects). The content of the journal also reflects documentation of the possible side-effects of rehabilitation, and includes prognostic perspectives of the treatment modalities chosen.
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