Clinical and Radiographic Findings and Usefulness of Computed Tomographic Assessment in Two Children with Regional Odontodysplasia

Matsuyama, Junko; Tanaka, Ray; Iizawa, Futabako; Sano, Tomiko; Kinoshita-Kawano, Shoko; Hayashi-Sakai, Sachiko; Mitomi, Tomoe

doi:10.1155/2014/764393

Cited by 9 publications

(15 citation statements)

References 11 publications

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“…This case showed teeth with hypoplastic, hypocalcified crown, poor contrast between dental tissues demonstrated by a low radiodensity, shortened roots with wide open apices, pericoronal radiolucency representing an enlarged dental follicle, and large pulp chambers giving a “ghost teeth” appearance. 11 17 21 23 25 26 In addition, it was observed the absence of right permanent mandibular central and lateral incisors, an unusual finding according to Tervonen et al 24 These authors compiled epidemiological data of RO published in the international literature up to 2002 and found only 10.7% of missing teeth in the studied sample of 138 cases. Similarly to the present case, Ganguly and Ramesh 27 reported an unusual case of mandibular RO associated with the absence of left lower and lateral incisors which was observed on periapical radiographs.…”

Section: Discussionmentioning

confidence: 88%

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Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Cavalcante

Fonteles

Ribeiro

et al. 2018

International Journal of Clinical Pediatric Dentistry

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Regional odontodysplasia (RO) is a rare nonhereditary developmental condition usually affecting maxilla and female patients. A few number of articles have used the cone beam computed tomography (CBCT) to describe imaging findings in RO. Thus, this article aims to report unusual imaging findings in a Brazilian child who presented with mandibular RO. A healthy 8-year-old boy was referred for evaluation of an anterior mandibular gingival enlargement. Radiographically, all right mandibular teeth showed wide pulp chambers and shortened roots with thin dentinal walls and open apices, featuring a “ghost teeth“ appearance, which indicated a diagnosis of RO. Missing teeth and altered radiopacity were observed. Cone beam computed tomography provided great insight into RO, allowing to identify gubernaculum tracts (GTs) associated with RO teeth. Fractal analysis of the bone surrounding RO teeth revealed an altered trabecular pattern.Clinical significanceThis case describes a rare developmental teeth-related pathology in a boy who showed unusual features on imaging exams. The CBCT provided the observation of RO tooth-related GTs, probably a new finding added to the international literature regarding RO, as well as the pulp chamber volume characterization of an affected tooth not published yet. In addition, it was observed an altered fractal pattern of the mandibular bone adjacent to RO teeth, which has not been described to date.How to cite this article: de Sa Cavalcante D, Fonteles CSR, Ribeiro TR, Kurita LM, Pimenta AVM, Carvalho FSR, Costa FWG. Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern. Int J Clin Pediatr Dent 2018;11(2):128-134.

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Section: Discussionmentioning

confidence: 88%

“…In RO, CT is a useful exam to provide a tri-dimensional tooth morphology, assessment of the pulp chamber volume, tissue density, and localization of dysplasia and hypocal-cification. 21 However, there is only one published case of RO evaluated on CBCT to date. 8 …”

Section: Introductionmentioning

confidence: 99%

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Cavalcante

Fonteles

Ribeiro

et al. 2018

International Journal of Clinical Pediatric Dentistry

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“…Activation of latent viruses in the odontogenic epithelium, and disorders of neural crest cell migration also have been suggested (Dahllöf et al, 1987, Nagai et al, 1963, Sabah et al, 1992). All authors agree that this is a non-hereditary condition, however the distribution of affected teeth in all four quadrants in some cases makes some authors suggest that somatic mutation may be the causative factor (Grover, 2014, Matsuyama et al, 2014, Sabah et al, 1992).…”

Section: Introductionmentioning

confidence: 99%

Regional odontodysplasia: An analysis of 161 cases from 1953 to 2017

Alotaibi

Alfawaz

2019

The Saudi Dental Journal

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Background and objectiveRegional odontodysplasia (RO) is a localized developmental anomaly involving both dentitions. The characteristic clinical findings are soft discolored several contiguous teeth with atypical morphology associated with swelling or abscess formation. Radiographic images of the involved teeth show a ghost-like appearance because enamel and dentin are hypoplastic and hypocalcified. Although RO is known for more than seven decades, the literature on RO is mostly limited to case reports.The objective of this investigation was to provide an updated review of the literature with regards to clinical presentation and epidemiology of RO. Because of its rarity, retrospective analysis of the published reports across the world is considered one of the most appropriate available methods to analyze epidemiological and clinical data of RO. This review was reported and conducted according to the PRISMA guidelines.ResultsBetween 1953 and 2017, 161 cases were reported in literatures written in English. At the time of diagnosis, the age of the patients ranged between 1 and 25 years. There was slight female predilection with a ratio of 1.37: 1. When examined collectively, the maxilla was commonly affected compared to the mandible. RO occurred in both dentitions in 75 reported cases. While it affects the deciduous teeth alone in 19.3% and the permanent teeth in 34.2% of the cases. Failure of tooth eruption was the most common presenting finding (41.6%) followed by swelling of the affected area (32.3%).ConclusionAlthough RO was first described almost 80 years ago and the total subsequent case reports were over 160 cases, there is no consensus on its pathogenesis and how it should be treated. The descriptive nature of the examined case reports reveals the limited information on this condition. Further experimental and genetic studies are needed.

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“…In any case, early diagnosis is necessary. 3D technology is a novel modern instrument for studying the structure of the tissues surrounding the tooth [14]. Knowledge on the etiology and genetics of RO facilitates the early diagnosing of the condition, which again is essential for treatment planning.…”

Section: Discussionmentioning

confidence: 99%

Familial oligodontia and regional odontodysplasia associated with a PAX9 initiation codon mutation

et al. 2019

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Objective Tooth agenesis is one of the most common craniofacial developmental anomalies. In hypodontia, one to five teeth are missing, whereas oligodontia refers to the absence of at least six teeth, excluding the third molars. Mutations in several genes including MSX1, PAX9, AXIN2, and WNT10A have been shown to cause non-syndromic tooth agenesis. Regional odontodysplasia (RO), also known as Bghost teeth,^is a rare developmental anomaly of tooth formation affecting both dentitions. Some possible causes of RO have been suggested, yet the etiology remains unknown. Because the phenotypes of both oligodontia and RO co-occur in one Finnish family, the aim here was to investigate the genetic etiology of the two conditions. Materials and methods A mutation screening of the genes MSX1, PAX9, AXIN2, and WNT10A was performed for the family members of a RO patient and family history of oligodontia. Results An initiation codon mutation of the PAX9 gene was found in the proband and segregating with oligodontia in the family. Conclusions The etiology of regional odontodysplasia (RO) may be genetic and the same genes can be involved both in RO and tooth agenesis. Clinical relevance Our results give new insights into the etiology of regional odontodysplasia, yet further results are needed.

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Clinical and Radiographic Findings and Usefulness of Computed Tomographic Assessment in Two Children with Regional Odontodysplasia

Cited by 9 publications

References 11 publications

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Mandibular Regional Odontodysplasia in an 8-year-old Boy showing Teeth Disorders, Gubernaculum Tracts, and Altered Bone Fractal Pattern

Regional odontodysplasia: An analysis of 161 cases from 1953 to 2017

Familial oligodontia and regional odontodysplasia associated with a PAX9 initiation codon mutation

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