Multidisciplinary approach to multiple dental anomalies in pediatric patients: a case report with 4-year follow-up.

The objective of this article is to report the clinical case and 4-year follow-up of a 5-year-old child with multiple dental anomalies, emphasizing the importance of early diagnosis and use of combined pediatric surgery and orthodontic approaches. A 5-year-old boy, accompanied by his mother, sought dental care for dental caries and tooth pain. Clinical and radiographic examinations revealed active caries, a supernumerary primary tooth in the region of the mandibular right second premolar, and severe ankylosis of the primary mandibular right second molar. The treatment plan involved extraction of the supernumerary tooth as well as sectioning and extraction of the ankylosed molar. When the patient was 6 years old, the permanent mandibular right first molar showed signs of an altered eruptive process, and orthodontic treatment was initiated. A unilateral band-and-loop space maintainer with coil springs designed to move the permanent first molar was placed on the primary first molar. A new panoramic radiograph, obtained when the patient was aged 7 years, suggested the presence of an odontoma in the apical region of the primary maxillary right canine. Surgical removal and histopathologic examination of the lesion confirmed that it was a developing odontoma. After surgery, due to occlusal anomalies that included transverse maxillary deficiency, deep overbite, and midline deviation, the patient underwent rapid maxillary expansion therapy with a Haas-type appliance. When the patient was 8 years old, orthodontic treatment continued with a removable palatal Hawley expander and a orthodontic mandibular lingual arch. Currently, at the age of 9 years, the child is still undergoing fixed orthodontic treatment after surgical exposure of the impacted permanent maxillary right canine and bonding of an orthodontic attachment to enable traction. A multidisciplinary approach to the management of dental anomalies promotes a favorable prognosis and ensures comprehensive treatment of young patients.

Large Complex Odontoma in the Angulus Mandibulae - Intraoral Enucleation as an Alternative to Mandibular Continuity Resection.

BACKGROUND: Odontomas are among the most common odontogenic tumors and are generally considered as hamartomatous odontogenic lesions. These tumors can be histopathologically divided into complex odontomas and compound odontomas based on their composition. Odontomas show a slow growing behavior and typically lack characteristic symptoms. The standard surgical treatment for large odontogenic tumors is a mandibular (continuity) resection followed by primary or secondary plastic reconstruction. CASE REPORT: A 22-year-old male presented to the Department of maxillofacial surgery with an increasing feeling of pressure in the left mandible. An orthopantomogram revealed a large complex odontoma rg 038. Instead of mandible continuity resection an alternative minimally invasive technique/approach (intraoral) with a trapezoidal bone flap for the enucleation of an odontoma of the mandibular angle with subsequent flap reimplantation and osteosynthesis was performed. CONCLUSION: Surgical enucleation of large mandibular odontoma with a continuity resection through an extraoral approach represents the surgical standard treatment of this entity. The present case report describes an alternative minimally invasive technique/approach. This technique may reduce surgical risks of the continuity resection through an extraoral approach (nerve damage, scarring) and can improve the long-term stability of the mandible by bone preservation.

Odontoma Recurrence. The Importance of Radiographic Controls: Case Report with a 7-Year Follow-Up.

Odontomas are benign tumors characterized by slow and limited growth with a rare recurrence. Odontomas are generally detected by radiographic findings in the radiopaque stage, where calcification of the tissues is observed. This article seeks to report the recurrence of a radiologically diagnosed odontoma to show the importance of radiographic controls after enucleation as a diagnostic and follow-up method. Case report: A female patient, 9 years old, attended dental care in 2020 due to malpositioned teeth. In the intraoral clinical examination, she presented stage II mixed dentition with crowding. A radiographic exam showed no associated lesions. The patient reported a history of odontoma removal and a supernumerary tooth in sextant II in 2016. Subsequently, she was referred to orthodontics, where permanent dentition with moderate anterior crowding in the maxilla and mandible was observed. The radiographic examination showed a radiopaque area compatible with odontoma, palatal to teeth 12 and 13. Conclusions: Although recurrence is rare, complete removal in the case of an odontoma is critical. This study demonstrates the importance of performing radiographic controls 5 years after enucleation of an odontoma, considering the stages of evolution.

Odontome and its pathological effect on surrounding teeth.

Odontoma is the most common odontogenic tumour derived from both epithelial and mesenchymal components of the tooth-forming apparatus. It is commonly diagnosed in the second and third decades of life when a radiograph is taken for some other purpose, as most cases are asymptomatic. This case involves a young boy, with the chief complaint of pain and swelling in the lower left back region. An intraoral examination revealed a carious and hypoplastic left permanent mandibular first molar. Although the molar was suspected as the source of his symptoms, radiographic imaging revealed multiple odontomas and missing second and third molar tooth buds. This case highlights the pathological effects of odontomas on surrounding teeth, including the malformation of the first molar and aplasia of the second and third molars. The sole management depends on the early diagnosis, histopathological examination to rule out malignancy and conservative surgical excision of these tissues.

[Komplexes Odontom im Unterkieferfrontzahnbereich bei einer 16-jährigen Patientin - Diagnostik, Therapie und Nachsorge].

Odontome gelten zusammen mit den Amelo- blastomen als die häufigsten odontogenen Tumoren. Sie entstehen während der embryo- nalen Zahnkeimentwicklung durch fehlerhaft differenziertes Keimgewebe und werden daher auch als Hamartome bezeichnet. Somit sind sie also strenggenommen keine klassischen Neoplasien.

Aggressive presentation of ameloblastic fibro-odontoma: a clinical-pathological enigma.

Ameloblastic fibro-odontoma (AFO) is a rare, gnathic, benign, mixed odontogenic tumor that commonly presents in the first or second decade of life as a unilocular and rarely multilocular radiolucency with variable amounts of calcified material. Tumor progression is typically indolent, and generally accepted treatment is surgical enucleation and curettage. This case report describes an atypical presentation in a 14-year-old male with a multilocular, aggressive AFO requiring hemimandibulectomy with immediate osseous and dental "Jaw-in-a-Day" reconstruction. This report highlights the debate regarding whether AFO is a true neoplasm or an early-stage hamartoma in the continuum of complex odontoma formation. Regardless of the pathogenesis, maxillofacial surgeons and pathologists should be cognizant of the potential for AFO to develop locally aggressive behavior with considerable morbidity.

Immunohistochemical expression of beta-catenin, BMP4 and TGF-beta in odontomas.

Changes in the expression of nuclear ß-catenin are responsible for tumorigenesis. Beta-catenin acts synergistically with the TGF-ß/BMPs pathway. This interaction leads to greater dentin deposition and may explain the differences between distinct tooth morphologies and hamartomas. The aim of this study was to investigate the role of ß-catenin, BMP4 and TGF-ß in the development of odontomas. This cross-sectional, retrospective, immunohistochemical study evaluated 30 compound odontomas, 30 complex odontomas and 17 tooth germs. The results showed that BMP4 and TGF-ß were more immunoexpressed in the ectomesenchyme of complex odontomas (median = 33.7, p < 0.001; median = 76.4, p = 0.002, respectively). Higher immunoexpression of BMP4 and TGF-ß was also observed in the epithelium of tooth germs (median = 2.0, p < 0.001; median = 120.3, p < 0.001, respectively). TGF-ß and BMP4 showed a positive and significant correlation (p < 0.001). Both TGF-ß and BMP4 were positively correlated with nuclear ß-catenin in ectomesenchyme (p = 0.047 and p = 0.023, respectively). Developing teeth exhibited higher concentrations of the proteins studied in odontogenic epithelium, especially during the bud and cap stages. Higher immunoexpression in odontomas occurred mainly in the ectomesenchyme. We therefore suggest that changes in the ectomesenchyme can lead to the development of odontomas.

[Three-dimensional radiographic features of calcifying odontogenic cyst and calcifying epithelial odontogenic tumor].

OBJECTIVE: To analyze the three-dimensional radiographic characteristics of calcifying odontogenic cyst and calcifying epithelial odontogenic tumor using spiral computed tomography (CT) and cone-beam computed tomography (CBCT). METHODS: Clinical records, histopathological reports, and CBCT or non-enhanced spiral CT images of 19 consecutive patients with calcifying odontogenic cyst (COC) and 16 consecutive patients with calcifying epithelial odontogenic tumor (CEOT) were retrospectively acquired, and radiographic features, including location, size, expansion, internal structure and calcification, were analyzed. RESULTS: Among the 19 COC cases (12 males and 7 females, with an average age of 27 years), 89.5% (17/19) of the lesions originated from the anterior and premolar areas, 100.0% of them exhibited cortex expansion, and 78.9% had discontinued cortex. Among the 16 CEOT cases (3 males and 13 females, with an average age of 36 years), 81.3% (13/16) of the lesions were in the premolar and molar areas, 56.3% of them exhibited cortex expansion, and 96.8% had discontinued cortex. According to the distribution of internal calcifications, these lesions were divided into: Ⅰ (non-calcification type): absence of calcification; Ⅱ (eccentric marginal type): multiple calcifications scattered along one side of the lesion; Ⅲ (diffused type): numerous calcifications diffusely distributed into the lesion; Ⅳ (plaque type): with a ≥ 5 mm calcified patch; Ⅴ (peri-coronal type): multiple calcifications clustered around impacted teeth. Calcifications were present in 73.7% of COC lesions, including 9 type Ⅱ, 3 type Ⅲ and 2 type Ⅳ lesions, and 42.8% of CEOT lesions had calcification images, including 2 type Ⅲ and 5 type Ⅴ lesions. Six COC lesions had odontoma-like images. Moreover, 8 of 9 type Ⅰ CEOTs were histologically Langerhans cell-rich subtype, which had a smaller size (with an average mesiodistal diameter of 17.8 mm) and were not associated with impacted teeth. CONCLUSION: COC lesions tended to originate from the anterior part of the jaw and exhibit cortex expansion, and were sometimes associated with odontoma. CEOT commonly occurred in the posterior jaw and had discontinued cortex. Two lesions had significantly different calcification map. Over 70% of COC lesions had calcification images, which were mostly scattered along one side of the cysts, far from the impacted teeth. Approximately 60% of CEOT lesions exhibited smaller size and non-calcification, and the remaining CEOT cases often had calcification images clustered around the impacted teeth.

Modifications of Gallego staining and its dental application.

BACKGROUND: The objectives of the study are to employ modified Gallego staining in pathology and to specifically evaluate the hard tissue apposed to odontogenic epithelium in order to aid in the diagnosis. METHODS: Lillie's modification of Gallego's stain was utilized as a reference to create a fresh batch of the stain. The archival and running cases of 2021-2022 was screened for odontogenic pathologies, yielding approximately 46 cases, among these four cases were selected which required evaluation of the hard tissue matrix apposed to odontogenic epithelium for its characterization. The modified Gallego staining was employed in the soft tissue sections of these cases under controlled environment. The staining results were evaluated. RESULTS: The stain was used in cases to stain dentinoid deposition as green color in hybrid ameloblastoma, archegonous cystic odontoma, dentinogenic ghost cell tumor and also in other cases like calcifying odontogenic cyst. Bone presented as green in color, cells as pink color and collagen as green-pink. This helped in the correct diagnosis of these cases facilitating the correct treatment modality. CONCLUSIONS: In the field of oral pathology, there are a multitude of lesions which are odontogenic with the diagnosis of few of them hinging on the characterization of hard tissue matrix found in close approximation to odontogenic epithelium implying an inductive capability to the odontogenic epithelium. This particular modified Gallego stain has been able to help in diagnosis of few such cases in our repertoire of cases.

A novel APC mutation associated with Gardner syndrome in a Chinese family.

Gardner syndrome (GS) is a specific form of familial adenomatous polyposis (FAP), which manifests as colorectal polyps, multiple osteomas and soft tissue tumors, and in the oral cavity as osteomas of the jaws, odontomas, and abnormal tooth counts. The underlying cause of GS is attributed to mutations in the APC gene. Mutations in this gene disrupt the normal functioning of the protein and lead to the development of GS. To further investigate GS, a family affected by the syndrome was selected from Dongguan, Guangdong Province. The family members underwent a comprehensive survey, which involved collecting clinical data and peripheral venous blood samples. The samples were then used for genetic analysis. Whole exome sequencing (WES) and Sanger sequencing techniques were utilized to screen and identify specific mutation sites in the APC gene. The clinical findings for the GS family included the presence of gastrointestinal polyps and odontomas. After analyzing the genetic sequencing results, a novel mutation site c.4266dupA on the APC gene was found in the patients, which leading to the APC protein truncation. As a result of this study, it is suggested that odontoma may be an early indicator of GS. Additionally, the identification of this novel mutation site in the APC gene expands the known spectrum of genetic mutations associated with the disease. This discovery has significant implications for the early diagnosis of GS, thus enabling timely intervention to reduce the risk of developing colon cancer and other related diseases.

Diminutive compound odontoma in a child: a case report with emphasis on early detection.

The odontoma is regarded as a hamartomatous process of the jaws. Most are discovered as an incidental radiographic finding, averaging 15 mm in size. This report describes a case of a diminutive odontoma that was surgically removed before the onset of eruptive and pathologic consequences. A compilation of documented complications and syndromes associated with odontomas is also presented.

Higher immunoexpression of CK14 from the Wnt-1/ß-catenin pathway in the development of odontomas.

Tooth development depends on a series of reciprocal signaling interactions between the oral epithelium and ectomesenchyme. This study aimed to investigate the role of CK14, a protein involved in Wnt-1/ß-catenin signaling, in odontogenesis and the development of odontomas. This cross-sectional, retrospective, immunohistochemical study analyzed 30 compound odontomas, 30 complex odontomas, and 17 tooth germs. Higher immunoexpression of CK14 was observed in odontogenic epithelial cells of tooth germs (p < 0.001) and odontogenic epithelial cells of odontomas (p < 0.001). There was higher immunoexpression of Wnt-1 and ß-catenin proteins in epithelial cells of tooth germs (p = 0.002 and p < 0.001, respectively), as well as in the ectomesenchyme of odontomas (p = 0.003 and p < 0.001, respectively). ß-Catenin was moderately and significantly correlated with CK14 in the membrane of reduced enamel epithelial cells in odontomas (p = 0.007). Higher immunoexpression of CK14 was observed in the odontogenic epithelium during the bud and cap stages and lower immunoexpression in the internal enamel epithelium during the bell stage. In odontomas, lower expression of Wnt-1/ß-catenin and higher immunoexpression of CK14 were found in odontogenic epithelial cells, especially adjacent to the mineralized material resembling the tooth formed in these lesions.

Pediatric Odontogenic Tumors.

Odontogenic tumors are rare tumors of the jaws that arise from remnants of the tooth forming apparatus. Some odontogenic tumors demonstrate strong predilection for pediatric patients including the unicystic ameloblastoma, adenomatoid odontogenic tumor, ameloblastic fibroma, ameloblastic fibro-odontoma, odontoma, and primordial odontogenic tumor. In this review, we discuss the clinical, radiographic, histopathologic, and molecular characteristics of select odontogenic tumors that demonstrate pediatric predilection and review management.

Ameloblastic fibro-dentinoma: a rare mixed odontogenic tumor case report with review of literature.

BACKGROUND: Ameloblastic fibro-dentinoma is considered a rare, benign, mixed odontogenic tumor that occurs mainly in the posterior mandible in the 1st-2nd decade of life. Although the clinical behavior of Ameloblastic fibro-dentinoma is similar to that of ameloblastic fibroma, there is a debate about whether Ameloblastic fibro-dentinoma is a developing hamartomatous odontoma or a separate neoplastic odontogenic tumor like ameloblastic fibroma. However, it is important to understand the histopathogenesis of this rare tumor. CASE PRESENTATION: A case report presenting an 11-year-old male child with a swelling in the posterior mandible. Radiographic examination revealed a multilocular lesion with mixed radiodensity related to the impacted lower left second premolar tooth. Incisional biopsy was done, and microscopic examination revealed cords and nests of odontogenic follicles lined by ameloblast-like cells and central stellate reticulum-like cells in the primitive ecto-mesenchymal stroma with areas of dentinoid material and osteodentin. The diagnosis was ameloblastic fibro-dentinoma. Surgical excision of the lesion was done, and the patient was followed up for 1 year without evidence of recurrence. CONCLUSION: Reporting such a rare entity clarifies the debate about its nature and the importance of early diagnosis of lesions that are associated with unerupted teeth showing how it is effective in early management and prognosis.

Odontogenic tumours in a Chilean population: a retrospective study of 544 cases based on 2022 WHO classification.

BACKGROUND: Odontogenic tumours are infrequent lesions. Studies on the frequency of odontogenic tumours from Latin America are scarce. This work aimed to determine the relative frequency of odontogenic tumours in a Chilean population using the 2022 World Health Organization classification. MATERIAL AND METHODS: This is a case series retrospective study. We reviewed 35,530 samples from 1975 to 2022 from the Oral Pathology Referral Institute and the Pathological Anatomy Service, Faculty of Dentistry, University of Chile. We utilized the 2022 World Health Organization classification for histological typification. RESULTS: According to 2022 World Health Organization classification, 544 odontogenic tumours were confirmed. The most frequent odontogenic tumours were: odontoma (n=241; 44.3%), ameloblastoma (n=109; 20.0%) and cemento-ossifying fibroma (n=71; 13.1%). Benign odontogenic tumours corresponded to 538 cases (98.9%) and malignant tumours were only six cases (1.1%). CONCLUSIONS: In our population, odontoma was the most frequent odontogenic tumour followed by ameloblastoma and cemento-ossifying fibroma. Malignant odontogenic tumours were very rare. The results of this study are similar to reports from America, but there are some differences concerning the data from Africa and Asia.

Revisiting the History of Odontoma, with Special Reference to Its Original Illustration.

BACKGROUND: Practically every facet of the most common odontogenic tumor, odontoma, has been covered by an extensive volume of literature. However, uncertainty about its precise history has persisted. MATERIALS AND METHODS: The historical evolution of odontoma was traced with reference to the original illustrations that accompanied European and American reports published at the beginning of the 19th century and also at the turn of the century. RESULTS: The prevailing views regarding the first description of odontoma by Oudet of Paris in 1809 and the original designation "odontome" by Broca of Paris in 1867 are not entirely accurate. Before Broca's suggested term, "exostose dentaire" (dental exostosis) and "tumeur dentaire" (dental tumor) proposed by Oudet and Forget of Paris, respectively, were popular terms adopted in France, while in Briatin the terms "warty tooth" and "supernumerary teeth" proposed by Salter and Tomes of London, respectively, were widely coined. The original illustrations of complex odontoma were published by Wedl of Vienna in 1851, and in 1862 Tomes published the first drawing of compound odontoma denticles. Before the advent of diagnostic radiography in the early 1900s, spontaneous exposure or eruption of odontoma followed by secondary infection was very common. In 1887-1888, Bland Sutton of London criticized Broca's monumental research and formulated the first modern classification which, in essence, remains valid today. At that time, large osteomas of the maxilla were inappropriately classified as odontomas by many pathologists because of Bland Sutton's influential view. Interestingly, the first radiographic evidence of odontoma was published by the American oral surgeon Gilmer in 1899. CONCLUSION: In view of their fundamental achievements, the names of Wedl, Salter, Broca and Bland Sutton have been closely associated with the true history of odontoma.

Tratamento de odontoma composto e cisto dentígero: relato de caso / Treatment of compound odontoma and dentigeral cyst: case report

Introdução: o odontoma é considerado como um frequente tumor odontogênico benigno, podendo ser classificado em tipo composto ou tipo complexo. O cisto dentígero é o mais comum entre os cistos odontogênicos de desenvolvimento, onde envolve a coroa da unidade dentária no nível da junção amelocementária. Há poucos estudos na literatura do encontro das duas lesões, acometendo o mesmo local na cavidade oral. O diagnóstico pode ser constituído por exame clínico e de imagem. Objetivo: apresentar um caso clínico de odontoma composto e cisto dentígero em região de parassínfise mandibular esquerda abordando as caraterísticas clínicas destas duas lesões e as adequadas formas de tratamento. Relato de caso: paciente do sexo masculino, 16 anos de idade, compareceu ao ambulatório do Centro Odontológico da Escola Bahiana de Medicina e Saúde Pública (Salvador, Bahia), portando encaminhamento de ortodontista, solicitando exodontia da unidade dentária 33 inclusa associada a um odontoma. Ao realizar exames físicos e imaginológicos detectou-se a hipótese diagnóstica de odontoma composto associado a unidade dentária, envolto em folículo pericoronário ou cisto dentígero. Foi realizada biópsia excisional das duas lesões e exodontia da unidade. A análise histopatológica confirmou o diagnóstico para odontoma composto associado a cisto dentígero na unidade 33. Ao acompanhamento de 03 meses, paciente apresentou neoformação óssea da região de parassínfise mandibular, mediante a análise de novos exames imaginológicos. Discussão: há poucos estudos na literatura da associação entre as duas lesões, porém relatos afirmam que o odontoma pode ser encontrado associado aos cistos odontogênicos. Por conta da falta de maiores estudos dessa associação, há escassez de recomendações terapêuticas de acordo com faixa etária e extensão do acometimento das lesões. Considerações finais: lesões comumente assintomáticas, tem o diagnóstico constituído por exame clínico e avaliação de exames de imagem(AU)

Introduction: odontoma is considered a frequent benign odontogenic tumor and can be classified as a compound or complex type. The dentigerous cyst is the most common among developmental odontogenic cysts, where it involves the crown of the dental unit at the level of the cementoenamel junction. There are few studies in the literature on the meeting of the two lesions, affecting the same site in the oral cavity. The diagnosis can be made by clinical and imaging examination. Objective: to present a clinical case of compound odontoma and dentigerous cyst in the left mandibular parasymphysis region, addressing the clinical characteristics of these two lesions and the appropriate forms of treatment. Case report: male patient, 16 years old, attended the outpatient clinic of the Centro Odontológico da Escola Bahiana de Medicina e Saúde Pública (Salvador, Bahia), having been referred by an orthodontist, requesting extraction of the included dental unit 33 associated with an odontoma. Upon physical and imaging examinations, the diagnostic hypothesis of a compound odontoma associated with a dental unit, surrounded by a pericoronal follicle or dentigerous cyst, was detected. Excisional biopsy of the two lesions and extraction of the unit were performed. The histopathological analysis confirmed the diagnosis of compound odontoma associated with dentigerous cyst in unit 33. At the 03-month follow-up, the patient presented bone neoformation in the mandibular parasymphysis region, through the analysis of new imaging exams. Discussion: there are few studies in the literature on the association between the two lesions, but reports state that odontoma can be found associated with odontogenic cysts. Due to the lack of further studies on this association, there is a lack of therapeutic recommendations according to age group and extent of lesion involvement. Final considerations: commonly asymptomatic lesions, the diagnosis consists of clinical examination and evaluation of imaging tests(AU)

Odontoma and other congenital dental anomalies: Implications for forensic identification.

Forensic odontology plays a crucial role in establishing the identity in mass disasters and criminal cases with high accuracy. Dental anomalies and features help in such situations. Congenital and developmental dental anomalies can be easily documented to establish distinctive and individualistic characteristics of an individual. The location, number of teeth involved, and the type of anomaly vary between individuals. Similarly, dental malformations also assist greatly in the identification process. Many types of dental anomalies have been studied in the past for their individualistic characteristics in forensic examinations. One such dental anomaly is odontoma, which is a benign odontogenic malformation. This malformation may also help in the identification of the deceased, when recorded and examined accurately. An odontome is a malformed teeth-like structures consisting of enamel, dentin, and pulpal tissue, formed due to the growth of completely differentiated epithelial and mesenchymal cells. If antemortem (AM) dental records incorporate information regarding odontomes and other dental anomalies, including in radiographs, orthopantomograms or microradiographs, positive identification may be established by comparison of these records with postmortem (PM) records. In the present communication, a rare case of compound composite odontoma in the anterior mandible with multiple denticles has been discussed with a brief overview of congenital and developmental dental anomalies. The authors emphasize the importance of such rare dental anomalies and malformations which may be used for identifying the deceased in mass disasters and forensic identification.

Dentinogenic ghost cell tumor associated with odontoma: report of a rare case and review of literature.

Dentinogenic ghost cell tumor (DGCT) is an uncommon odontogenic tumor considered to be the solid, tumorous variant of calcifying odontogenic cyst. DGCT is characterized by islands of ameloblastoma-like epithelial cells that resemble the enamel organ, the presence of ghost cells, and dentinoid material. This article reports a rare case of dentinogenic ghost cell tumor associated with an odontoma in an adult patient, with a review of the literature. To the best of the authors' knowledge, there have only been four case reports of DGCT associated with odontoma, all of which occurred in children and adults younger than 30 years old.