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广泛性牙发育异常:一例年龄最大的报告病例

Generalized Odontodysplasia: A Case Report of the Oldest Reported Patient.

作者信息

Vivero-Alcívar Freddy Andrés, Escoto-Vásquez Lilibeth Stephania, Hernández-Ortega Oscar Rohel, Ramírez-Lugo Raymundo, Molotla-Fragoso Adriana

机构信息

Department of Oral and Maxillofacial Surgery, Postgraduate and Research Division, Dental School, National Autonomous University of Mexico, Mexico City, Mexico.

Medical, Dental and Health Sciences Program, Postgraduate and Research Division, Dental School, National Autonomous University of Mexico, Mexico City, Mexico.

出版信息

Case Rep Dent. 2025 Jul 14;2025:5602411. doi: 10.1155/crid/5602411. eCollection 2025.

DOI:10.1155/crid/5602411
PMID:40692763
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12279413/
Abstract

Odontodysplasia is a dental anomaly that affects the maturation and formation of teeth, resulting in hypoplasia and hypocalcification of both enamel and dentin. It can affect one or several quadrants of the dentition, although generalized involvement is extremely rare. The exact cause is unclear, but trauma, infection, and nutritional or metabolic deficiencies have been suggested as possible contributing factors. Diagnosis requires a combination of clinical and radiological findings. Clinically, it presents as small teeth with yellow or brown discoloration, and it can affect both the primary and permanent dentition. Radiographically, there is reduced radiodensity, giving rise to a blurred or "ghost tooth" appearance. Histologically, these teeth show aprismatic enamel, interglobular dentin, and the presence of enamel-like calcifications known as enameloid conglomerates. Treatment depends on the extent of tooth involvement and the patient's age. This paper reports the case of a 31-year-old woman who presented with this rare anomaly in its generalized form, making her the oldest patient with this diagnosis reported in the literature.

摘要

牙发育异常是一种影响牙齿成熟和形成的牙齿异常,导致牙釉质和牙本质发育不全及钙化不全。它可影响牙列的一个或几个象限,尽管全身性受累极为罕见。确切病因尚不清楚,但创伤、感染以及营养或代谢缺陷被认为是可能的促成因素。诊断需要结合临床和影像学检查结果。临床上,表现为牙齿小且呈黄色或棕色变色,可影响乳牙列和恒牙列。在影像学上,放射密度降低,呈现出模糊或“鬼牙”外观。组织学上,这些牙齿表现为无棱柱形牙釉质、球间牙本质,以及存在称为类釉质团块的类牙釉质钙化。治疗取决于牙齿受累程度和患者年龄。本文报告了一名31岁女性的病例,她出现了这种罕见异常的全身性表现,使其成为文献报道中患有该诊断的最年长患者。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/96ee739f13d2/CRID2025-5602411.006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/947724bab8ef/CRID2025-5602411.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/ce5b08955120/CRID2025-5602411.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/bb5004b293b8/CRID2025-5602411.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/32965bb84f41/CRID2025-5602411.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/04c0d41171f1/CRID2025-5602411.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/96ee739f13d2/CRID2025-5602411.006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/947724bab8ef/CRID2025-5602411.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/ce5b08955120/CRID2025-5602411.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/bb5004b293b8/CRID2025-5602411.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/32965bb84f41/CRID2025-5602411.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/04c0d41171f1/CRID2025-5602411.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/404e/12279413/96ee739f13d2/CRID2025-5602411.006.jpg

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本文引用的文献

1
PHACES syndrome and multi-regional odontodysplasia: a case report.PHACES 综合征与多部位牙源性发育不良:病例报告。
J Clin Pediatr Dent. 2024 May;48(3):166-170. doi: 10.22514/jocpd.2024.070. Epub 2024 May 3.
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Regional Odontodysplasia: A Systematic Review of Case Reports.区域性牙源性发育不良:病例报告的系统回顾。
Int J Environ Res Public Health. 2022 Feb 1;19(3):1683. doi: 10.3390/ijerph19031683.
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Infantile hemangioma. Part 1: Epidemiology, pathogenesis, clinical presentation and assessment.婴儿血管瘤。第 1 部分:流行病学、发病机制、临床表现和评估。
J Am Acad Dermatol. 2021 Dec;85(6):1379-1392. doi: 10.1016/j.jaad.2021.08.019. Epub 2021 Aug 19.
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Treatment Strategies for Patients with Regional Odontodysplasia: A Presentation of Seven New Cases and a Review of the Literature.局部牙发育异常患者的治疗策略:七例新病例报告及文献综述
Oral Health Prev Dent. 2020 Sep 4;18(4):669-681. doi: 10.3290/j.ohpd.a45070.
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Regional odontodysplasia: An analysis of 161 cases from 1953 to 2017.局部牙发育异常:1953年至2017年161例病例分析
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Oral rehabilitation of a case with regional odontodysplasia using a regenerative approach-A case report and a review of literature.采用再生方法对一例局部牙发育异常病例进行口腔修复——病例报告及文献综述
Spec Care Dentist. 2019 May;39(3):330-339. doi: 10.1111/scd.12378. Epub 2019 Apr 16.
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Regional Odontodysplasia Affecting the Maxilla.累及上颌骨的区域牙发育异常
Head Neck Pathol. 2020 Mar;14(1):224-229. doi: 10.1007/s12105-019-01031-3. Epub 2019 Mar 21.
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Familial oligodontia and regional odontodysplasia associated with a PAX9 initiation codon mutation.家族性缺牙症和区域性牙本质发育不全伴 PAX9 起始密码子突变。
Clin Oral Investig. 2019 Nov;23(11):4107-4111. doi: 10.1007/s00784-019-02849-5. Epub 2019 Feb 26.
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Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern.一名8岁男孩的下颌区域牙发育异常,表现出牙紊乱、引导索及改变的骨分形模式。
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