Dentinoid Formation Research Articles

Developmental Anomalies in Human Teeth: Odontoblastic Differentiation in Hamartomatous Calcifying Hyperplastic Dental Follicles Presenting with DSP, Nestin, and HES1.

Hyperplastic dental follicles (HDFs) represent odontogenic hamartomatous lesions originating from the pericoronal tissues and are often associated with impacted or embedded teeth. These lesions may occasionally feature unique calcifying bodies, known as calcifying whorled nodules (CWNs), characterized by stromal cells arranged in a whorled or spiral fashion. CWNs are typically observed in multiple calcifying hyperplastic dental follicles or regional odontodysplasia. In our study, we examined 40 cases of HDFs, including nine instances with characteristics of CWNs, referred to as calcifying hyperplastic dental follicles (CHDFs), which are infrequently accompanied by odontodysplasia. The median ages of the HDFs and CHDFs were 16 (ranging from 3 to 66) and 15 (ranging from 11 to 50) years, respectively. The lower third molars were the most frequently affected by HDSFs and CHDFs, followed by the upper canines. A histological examination was conducted on all 40 cases, with an immunohistochemical analysis performed on 21 of them. Among the cases with CWN, nine affected a single embedded tooth, with one exception. CWNs exhibited diverse calcifications featuring sparse or entirely deposited psammoma bodies, and some displayed dentinoid formation. Immunohistochemically, the stromal cells of HDFs were frequently positive for CD56 and nestin. By contrast, CWNs were negative for CD56 but positive for nestin as well as hairy and enhancer split 1 (HES1), with a few dentin sialoprotein (DSP)-positive calcified bodies. Our results revealed that hamartomatous CHDFs can impact multiple and single-embedded teeth. CWNs composed of nestin and HES1-positive ectomesenchymal cells demonstrated the potential to differentiate into odontoblasts and contribute to dentin matrix formation under the influence of HES1. This study is the first report documenting odontoblastic differentiation in HDFs. The rare occurrence of HDFs and CHDFs contributes to limited comprehension. To prevent misdiagnosis, a better understanding of these conditions is necessary.

Odontogenic Carcinoma with Dentinoid of the Left Maxilla

Abstract Introduction/Objective Odontogenic carcinoma with dentinoid is a very rare dentinoid producing odontogenic tumor with 10 cases reported in literature, and it is not currently defined by the World Health Organization. It has been described to have a locally aggressive and non-metastatic course with high level of recurrence after excision. Methods/Case Report We report a case of odontogenic carcinoma with dentinoid located in the left maxilla of an otherwise healthy, 22-year-old male. The patient initially endorsed left upper molar tooth pain for 2 months that led him to receive medical attention. Initial examination revealed a 3 x 2 cm firm, non-mobile and non-pulsatile lesion on buccal and lingual surface of the left maxilla with imaging revealing a 2.9 x 2.7 x 2.7 cm mixed radiolucent/radiopaque lesion in the posterior left maxilla with expansion on the buccal and palatal surfaces. Incisional biopsy of the left maxilla (soft tissue) was initially diagnosed as suspicious for osteosarcoma, which was later changed to odontogenic carcinoma with dentinoid. One year later, the mass had increased to 4.5 x 3 x 4 cm in size. Left hemi maxillectomy was performed, and the specimen was diagnosed with odontogenic carcinoma with dentinoid. Previously described histological features of clear cell change, low grade appearance, focal calcification, and dentinoid formation are present in our case. Our case is the first to describe glandular formation of the tumor cells. Results (if a Case Study enter NA) NA Conclusion The histopathological changes of our case reflect previously reported microscopic descriptions for this exceedingly rare tumor that is unclassified by the World Health Organization. It has been reported to occur in both male and female patients without predilection and a wide age range from 14-61 years. To our knowledge, this is the 11th reported case, and the 5th case described in the maxilla. Further studies, including NGS sequencing to determine genetic alterations, will be performed on our case to further elucidate pathophysiological mechanisms. Long term follow up with our patient will be attempted to determine tumor behavior.

Hemangiomatous Ameloblastoma with Dentinoid Formation: A Sui Generis Case Report

Hemangiomatous ameloblastoma has long been explicated as a divergent of solid multicystic ameloblastoma (SMA). Described first by Kuhn in 1932, it is histologically distinct from a conventional ameloblastoma by the presence of spaces filled with blood or large endothelial lined capillaries. Less than 12 cases have been reported till date and mostly in the middle-aged population with a predilection for the mandibular posterior region. The unlikeness of the case being reported here is the fact that it was manifested in a 16-year-old teenage girl. Being an extraordinary version of SMA, very little is known about its behavior and prognosis. In the following case report, we try to annotate the clinical, radiological, and the histological features that lead us to the diagnosis of hemangiomatous ameloblastoma with dentinoid formation.

ADENOID AMELOBLASTOMA WITH DENTINOID: A CASE REPORT

Adenoid ameloblastoma with dentinoid (AAD) has been considered a very rare variant of ameloblastoma showing histopathologic features similar to adenomatoid odontogenic tumor (AOT) along with apparent dentinoid formation. Since the first use of this term by Brannon in 1994, however, there has been no official recognition of this entity as shown in both the 3rd and 4th edition of WHO classification of odontogenic tumors in 2005 and 2017. Because less than 20 cases of AAD have been reported to date, clinical behavior and optimal treatment modalities of AAD are still uncertain. Here we present an additional case of AAD with recurrence 10 years after the initial treatment. A 39-year-old male was referred to department of oral and maxillofacial surgery, complaining of pain and mobility of teeth in the right posterior maxilla. Panoramic radiograph revealed a unilocular radiolucency with relatively well-defined borders extending from the second premolar to the second molar. Root resorptions of the affected teeth were found. Mass excision was performed and the diagnosis of epithelial odontogenic ghost cell tumor was made. Ten years later, he presented with the recurrent lesion at the same area. CT view showed destructive enhancing mass suspicious for malignancy at the right posterior maxilla. Radically resected mass was diagnosed as adenoid ameloblastoma with dentinoid/osteodentin as it showed lots of duct-like structures with ameloblastoma-like features along with numerous dentinoid formation, but there were no ghost cells.

Odontogenic keratocyst with granular cell changes: A distinctive finding

Odontogenic keratocyst (OKC) originates from the dental lamina and is more commonly seen in the posterior mandible than in the maxilla. OKC is the most aggressive cyst of the oral cavity and is known for its rapid growth and its tendency to invade bone of the adjacent tissues. The recurrence rate of OKC is very high due to various reasons debated upon. Cases of OKC have shown the presence of calcifications, dentinoid formation and ossification. Here, we report the first case of OKC in a 27-year-old male showing granular cell changes.

Odontogenic keratocyst with dentinoid formation

Odontogenic keratocyst with dentinoid formation

Mural calcifications in keratocystic odontogenic tumor: Report of a case with a brief review

Hard tissue deposits or mineralization in the wall of the keratocystic odontogenic tumor (KCOT) are infrequent and unusual. They are usually of dystrophic calcification or cartilage type, and dentinoid formation, although reported, is a rare phenomenon. Here, a rare case of extensive mural calcifications in the wall of the KCOT involving maxillary premolar-molar area, in a 14-year-old boy is described. These calcifications were found to be dentinoid, confirmed with the use of special stains.

Update of odontogenic tumors

The latest WHO classification of odontogenic tumours was published in 2005. The classification has been used as a global standard for last 10 years but our knowledge of odontogenic tumors continues to evolve. At biennial meetings of International Association of Oral and Maxillofacial pathologists (IAOP), we have started discussions on classification of odontogenic tumors toward the next WHO Classification. I would like to introduce some subjects of discussion including cyst-tumor interface (odontogenic keratocyst vs keratocystic odontogenic tumor, glandular odontogenic cyst vs central mucoepidermoid carcinoma, calcifying odontogenic cyst vs ghost cell lesions); placement of adenomatoid odontogenic tumor (hamartoma vs neoplasia, epithelial tumor vs mixed tumor with dentin formation), clear cell odontogenic carcinoma with dentinoid formation; primary intraosseous squamous cell carcinoma vs sclerosing odontogenic carcinoma; classification based on inductive phenomenon of odontogenesis vs clinical behavior and others. Recent advances in molecular analyses of odontogenic tumors will be presented. I expect to exchange opinions on the new classification of odontogenic tumors.

Adenoid ameloblastoma with dentinoid

Ameloblastomas seldom cause diagnostic difficulties due to classic histopathological presentations. Adenoid ameloblastoma is a rare variant in this category which can cause problem in diagnosis due to the presence of areas resembling adenomatoid odontogenic tumor (AOT) and occurrence of varying degrees of dentinoid formation. In this article, we report a case of adenoid ameloblastoma with dentinoid, which was diagnosed accurately after the third recurrence. To the best of our knowledge, so far, only 13 cases have been reported of tumors that histologically show features of amelobalstoma and AOT with hard tissue formation. The recurrences were due to under diagnosis of the lesion followed by a conservative treatment.

A Rare Case of Gorlin Cyst in the Maxillary Anterior Edentulous Region

ABSTRACT The Gorlin cyst (calcifying epithelial odontogenic cyst) is a rare lesion. Its clinical and radiological features are not pathognomonic, and is characterized by histological diversity. Radiographically, it presents as a unilocular or, occasionally, multilocular radiolucency that may contain calcified radiopacities or tooth-like densities. Microscopic appearance of odontogenic epithelium admixed with focal areas of dentinoid formation or calcification along with sheets of ghost cells gives the definitive diagnosis of Gorlin cyst. A rare case of Gorlin cyst, which present as swelling in the maxillary anterior edentulous region along with emphasis on review of literature, is presented in this paper.

Dentinogenic Ghost Cell Tumor of the Peripheral Variant Mimicking Epulis

Dentinogenic ghost cell tumor (DGCT) is an uncommon locally invasive odontogenic tumor regarded by many as a variant of calcifying odontogenic cyst. The peripheral variant of this clinical rarity appears as a well-circumscribed mass mimicking a nonspecific gingival enlargement. Microscopic appearance of odontogenic epithelium admixed with focal areas of dentinoid formation and sheets of ghost cells giving the definitive diagnosis of dentinogenic ghost cell tumor imply that microscopic examination is compulsory for any gingival mass. Van Gieson histochemical stain further confirmed the nature of dentinoid-like material. A complete workup of a case of peripheral dentinogenic ghost cell tumor is presented in this paper and the current concept as well as the appraisal of literature is presented.

Morphometric analysis of epithelial components and dentinoid formation in non-neoplastic calcifying odontogenic cyst.

Calcifying odontogenic cysts (COCs) represent a group of lesions that may be broadly classified into two main entities: cysts and neoplasms. In the present study 30 non-neoplastic cystic COCs were examined by a quantitative histological method in an attempt to calibrate the relative distribution of the type of epithelial lining, intensity of ghost cell formation and the amount of dentinoid present. The results showed that there are two main types of cystic COC: an odontoma-producing type and a non-odontoma-producing variant. Morphologically, tooth-like structures were a valid distinguishing feature, while morphometrically the odontoma-producing variant showed a greater amount of luminal and mural dentinoid as well as luminal ghost cells. Demographic analysis also revealed that the odontoma-producing COC occurred in younger patients and showed an even sex distribution, whereas the non-odontoma-producing type was seen in older patients and showed a predilection for females. Both subtypes were more prevalent in the Chinese population and occurred preferentially in the maxilla.

類象牙質の形成を伴った腺様歯原性腫ようの１例

The adenomatoid odontogenic tumor (AOT) is a rare benign tumor. In this paper, a case of AOT which was histologically different from the previously reported cases was described.A 34 year-old male was referred for evaluation of a swelling in the mandible. The patient had been aware of the swelling for several months but never felt pain or paresthesia. The patient's past medical history was noncontributory. Local examinations disclosed an expansion of the cortices of the lower anterior region. The overlying mucosa was normal in color and texture. Radiographs revealed a radiolucent monolocular lesion in the anteriorpremolar area of the mandible. The lesion was interpreted as a benign tumor of the mandible. Under general anesthesia, surgical enucleation and curettage were carried out. The teeth from the right central incisor to the left premolar were extracted. Pathological diagnosis of the surgical specimen was an adenomatoid odontogenic tumor associated with dentinoid formation. The patient has been free of disease for two years after the surgery. From the following analysis of the previously reported cases, the current case was characterized by several unusual features, such as sex (male) and age (34 years old) of the patient, location of the lesion (mandible), absence of the impacted teeth and presence of dentinoid formation.From a search of the Japanese literature, the authors chose 84 well-documented cases of AOT including our own case to characterize clinical features of AOT among the Japanese. The following analysis is based on our study.1. AOT occurs most frequently in the second decade of life, though cases have been reported in the range of 3-46 years of age. The mean age is 18. 2 years.2. AOT affects females with greater frequency than males, a ratio of 3. 4: 1.3. The maxilla is involved more frequently than the mandible, a ratio of 1. 3: 1.4. Sixty-seven percent of AOT is associated with the impacted teeth and the canine is most frequently affected (58.9%).5. AOT is usually asymptomatic and causes jaw enlargement.6. AOT often resembles a follicular cyst radiographically.7. The size of AOT is less than 3.0 cm in diameter.8. Calcified material is usually present within AOT (96.4%).9. AOT does not show invasive tendency, metastasis and malignant transformation.10. Enucleation is a curative treatment for AOT.

Ameloblastic fibroma with dentinoid formation/immature dentinoma. A microscopic and ultrastructural study of the epithelial-connective tissue interface.

The epithelial-connective tissue interface in an ameloblastic fibroma with dentinoid formation shows four characteristic features: epithelium which is surrounded by a cellular stroma, by a cell-free zone, by a cell-free zone consisting of an amorphous hyaline-like material, and by a dentinoid material. The ultrastructure of these four characteristics resembles stages of the developing tooth. The cellular epithelial-connective tissue interface corresponds to early development before the "bud cap" stage described by Sisca and Provenza (1972). The cell-free zone resembles the "bud cap" stage, and the amorphous layer the "intermediate bell" stage, whereas the interface with dentinoid material has all the features of the "late bell" stage, except for the absence of calcification.