Adenomatoid Tumor Research Articles

Prevalence of Odontogenic Tumours: A Study of 122 Cases among Karnataka Population

Introduction: Tumours arising from odontogenic tissues are rare and constitute a heterogeneous group of interesting lesions. Studies on incidence of Odontogenic Tumours (OT) published from many countries show a distinct geographic variation. Aim: The aim of present study was to compare and correlate the frequency of individual OT in India as well as the other parts of the world based on 2005 World Health Organization (WHO) classification. Materials and Methods: This institution-based retrospective study conducted in Department of Oral Pathology and Microbiology at Government Dental College and Research Institute, Bengaluru, Karnataka, India, from April 2015 to September 2016. Total of 122 cases of odontogenic tumours documented in duration of 10 years (2005 to 2014) were categorised based on World Health Organization 2005 classification. The histological type, age, gender, anatomical site and symptoms were systematically tabulated. Results: Odontogenic tumours constituted 9.5% (122 cases) of the total oral biopsies (1284 cases) received in a 10 year period. Among these, 95.9% of the tumours were benign and 4.1% were malignant. The most frequent tumour being ameloblastomas 39.3% followed by keratocystic odontogenic tumour 35.2%, odontome 4.9%, calcifying cystic odontogenic tumour 4.9% and adenomatoid odontogenic tumour 4%. While odontome, generally regarded as the most frequent OT in American and European countries, only accounted for 4.9%, the third most common tumour in the present study. The mean age of patients was 32.37 years (range: 9-70 years). Their exist a slight male predominance, more predilection to posterior region of mandible and majority of tumours presented with pain and swelling. Conclusion: The relative frequency of Odontogenic tumours reported in our institution was similar to the studies reported in India, Asian and African population but differs from that of European and American countries. There is a definite geographic variation in the prevalence of OT published worldwide.

An unusual presentation of aneurysmal bone cyst of the maxillary sinus in a child: A rare case report

ABSTRACT Aneurysmal bone cyst(ABC) is a rare vascular tumor arising usually from long bone and vertebrae,very rarely involves the skull and facial bone. Here, we present a case of a 9-year-old child presented with progressive proptosis and strabismus of the left eye associated with nasal obstruction and epistaxis. Computed tomography was done which gave us an insight into the possibilities of ABC, giant cell tumor, adenomatoid odontogenic tumor, etc., Biopsy was taken for histopathological confirmation. Usually, ABC of the maxillary sinus presents with pain, cheek swelling, and nasal obstruction. Our patient presented with ocular findings which are rare among maxillary bone ABCs.

Germ cell neoplasms of the testis: Update for 2022.

Germ cell neoplasia in situ (GCNIS) is the precursor of both seminomatous and non-seminomatous germ cell tumors. It consists of distended tubules that may have either intratubular seminoma or intratubular embryonal carcinoma cells. Many invasive non-seminomatous tumors contain a mixture of tumor types, which are reviewed here. Morphology, aided by a panel of immunostains, can determine the presence and percent of embryonal carcinoma, yolk sac tumor, choriocarcinoma, or teratoma in such tumors. Use of immunostains, required for diagnosis in perhaps 25% of testicular neoplasms, is reviewed. Changes of classification in the AJCC (8th edition) in 2016 are discussed, including the partitioning of two tumor types: the central role of chromosome 12p amplification allows both teratoma and yolk sac tumor to be divided into prepubertal types (lacking amplification) and post-pubertal types. Occasionally, sex cord-stromal tumors, hematolymphoid tumors, or epididymal adenomatoid tumors enter the differential diagnosis of germ cell neoplasms.

Uterine Adenomatoid Tumor: A Great Imitator of Leiomyoma under Laparoscopy.

Uterine Adenomatoid Tumor: A Great Imitator of Leiomyoma under Laparoscopy.

Adenomatoid tumors of ovary mimicking malignancy: report of 2 cases and literature review

BackgroundAdenomatoid tumors (ATs) are benign tumors originating from the mesothelium. ATs of the ovary are rare, and can easily be confused with malignancy due to the histomorphological diversity. Thus, it is difficult in histopathological and differential diagnosis, especially during intraoperative frozen pathological diagnosis, which directly affects the resection scope of surgery.Case presentationIn this study, we reported two patients (58 and 41 year old) with ovarian ATs. AT of patient 1 occurred in both ovaries at different time points and she had been diagnosed with Hashimoto's thyroiditis. AT of patient 2 occurred in right ovary. Intraoperative frozen pathological diagnosis was performed in both cases and laparoscopic salpingo-oophorectomy was undergone on the lesion side according to benign freezing diagnostic result. Ovarian ATs, the final diagnoses of the 2 cases were concluded after histological, extensive immunohistochemical (IHC), histochemical, and fluorescence in situ hybridization analyses.ConclusionsOur results show that ovarian ATs may not be related to BAP1 or CDKN2A/p16 mutations. In addition, the case 1 suggests that ATs may be associated with immune dysregulation. When encountering such similar lessions, we recommend that a series of immunohistochemical, histochemical and molecular biological techniques should be used for diagnosis and differential diagnosis to avoid misdiagnosis. Improving understanding of the rare ovarian ATs which mimic malignancy is necessary to prevent overresection.

Expression of Fascin and SALL4 in odontogenic cysts and tumors: an immunohistochemical appraisal.

Background: Various stemness markers (SOX2, OCT4, and NANOG) have been studied in odontogenic cysts and tumors. However, studies on SALL4 having similar properties of stemness has not been documented. Additionally, insight into fascin as a migratory molecule is less explored. In this study, the expression of SALL4 and fascin were evaluated in ameloblastoma, adenomatoid odontogenic tumor (AOT), odontogenic keratocyst (OKC), dentigerous cyst (DC), radicular cyst (RC), and calcifying odontogenic cyst (COC). Methods: Semi-quantitative analysis of fascin and SALL4 immuno-positive cells was done in a total of 40 cases of ameloblastoma (11 plexiform, 12 follicular, 12 unicystic, and 5 desmoplastic) variants, 6 cases of AOT, 15 each of OKC, DC, RC and 5 of COC. Chi-square test was applied to evaluate the association between SALL4 and fascin expression in odontogenic cysts and tumors. Results: Fascin immunopositivity was observed in peripheral ameloblast-like cells, and weak or absent in stellate reticulum-like cells. A moderate to weak immune-reactivity to SALL4 was observed in the cytoplasm of ameloblastoma, epithelial cells of dentigerous and radicular cysts, having a marked inflammatory infiltrate, which is an interesting observation. COC and AOT had negative to weak expressions. No recurrence has been reported. Conclusions: Expression of fascin in ameloblastomas elucidate their role in motility and localized invasion. Its expression in less aggressive lesions like DC, COC, AOT will incite to explore the other functional properties of fascin. SALL4 expression in the cytoplasm of odontogenic cysts and tumors may represent inactive or mutant forms which requires further validation.

Adenomatoid odontogenic tumour- revisted

Adenomatoid odontogenic tumor, fourth most common odontogenic tumor in the Indian population. It is the most common odontogenic tumor associated with impacted teeth. The diagnosis stands important as it is most commonly associated with an impacted maxillary canine. An adenomatoid odontogenic tumor (AOT) is a tumor of epithelial origin that shows duct-like structures and the epithelium forms whorled masses of cells in a scant fibrous stroma. AOT is not a common odontogenic neoplasm and it was first described by Steensland in the year 1905. Initially, AOT was described as pseudoadenoameloblastoma by Dreibladt in the year 1907 and it was considered a distinct entity and some of them believed it to be a variant of ameloblastoma by Stafne in the year 1948.

An Unusual Presentation of “Two-Thirds Tumor” in the Mandible – A Diagnostic Quandary with a Review of the Literature

An adenomatoid odontogenic tumor (AOT) is an uncommon benign tumor of the oral cavity commonly found in the maxillary anterior region and is associated with impacted canines in young females. It rarely occurs in the mandibular region with no impacted or missing teeth. A 21-year-old female reported to the clinic with swelling on the right side of the face for the past six months with no history of pain. Radiographic features such as unilocular radiolucency with thinning of the cortical borders and considerable buccal cortical expansion, as well as some evidence of radiopaque specks were noted. Histopathological examination revealed cells with hyperchromatic nuclei, rosette-like structures with focal areas of calcified mass, and concentric rings resembling Liesegang rings, suggestive of AOT. The tumor was treated surgically by enucleation and cauterization. Although follicular type is a common variant, the tumor presented in this case was of extrafollicular type noted in the mandibular canine and premolar regions of a young female patient with no related impacted tooth.

Immunohistochemical expression of Ki-67 and Glypican-3 to distinguish aggressive from nonaggressive benign odontogenic tumors.

The benign neoplasms are normally slow growing, indolent with no invasive potential. However, there exist a few locally aggressive benign odontogenic tumors that have a tendency to invade and deform the surrounding structures. The exact reason for the aggressiveness of these benign neoplasms remained an enigma. Their biology and clinical expression can often be destructive and ominous. An appropriate treatment protocol needs to be followed to combat the high recurrence rate and aggressiveness of these entities. Aggressive and noniaggressive epithelial odontogenic tumors were analyzed immunohistochemically with Ki-67 and glypican 3 (GPC3). Fifty-nine cases of tumors were divided into aggressive odontogenic tumors (20 solid ameloblastomas, four unicystic ameloblastoma, and 28 keratocystic odontogenic tumors) and nonaggressive odontogenic tumors (five adenomatoid odontogenic tumors and two calcifying cystic odontogenic itumors). Statistical analysis using Pearson correlation showed Ki-67 to be a better marker for differentiating aggressive from nonaggressive odontogenic tumor as compared to GPC3 (P < 0.001, highly significant), whereas among aggressive tumors, GPC3 turned out to be more useful as compared to Ki-67 (P < 0.001, highly significant). The present study provides an insight into the different biological behavior of odontogenic tumors, which can thus be helpful in determining the therapy strategies for more aggressive odontogenic tumors.

Epididymal adenomatoid tumor: a case report and literature review

BackgroundAdenomatoid tumor is a very rare benign neoplasm of mesothelial origin affecting mainly female and male genital tracts. The diagnosis is challenging as this tumor mimics many differential diagnoses. The current literature offers only some case reports and short series of adenomatoid tumors.Case presentationA 47-year-old patient with unremarkable medical history presented for chronic mild pain of the right testis evolving for months. The physical examination shows a palpable right intrascrotal nodule of 10 mm in greatest diameter. The nodule was painful, mobile with firm consistency. The laboratory investigations were within normal limits, the scrotal ultrasonography showed a well-circumscribed predominantly hyperechoic intrascrotal nodule in the right epididymal head with heterogeneous echostructure. Excisional biopsy of the lesion was performed and the histopathological analysis showed a well-circumscribed tumor with microcystic and trabecular architecture made of small interconnected tubules and cysts lined by flattened cells with prominent vacuolization and thread-like bridging strands, consistent with an epididymal adenomatoid tumor. The postoperative course was uneventful and the patient was discharged. Four months after surgical treatment, the patient has no sign of the disease.ConclusionTesticular adenomatoid tumors are uncommon benign neoplasms with diagnostic challenge. Adenomatoid tumors arising in epididymis are managed by excisional biopsy with testis-sparing surgery avoiding unnecessary orchidectomy.

CDC7 Expression in Selected Odontogenic Tumors.

CDC7 is a serine-threonine kinase that plays a key role in initiating DNA replication. It has been implicated in the growth and invasion of many pathologic lesions and suggested as a diagnostic marker. The aim of this study was to evaluate CDC7 in some odontogenic tumors. In this cross-sectional study, 45 cases, including 19 ameloblastomas, 15 dentigerous cysts, 7 ameloblastic fibromas, and 4 adenomatoid odontogenic tumors (AOT), were studied immunohistochemically. ANOVA and post hoc methods were used for statistical analysis. CDC7 expression was observed in 93% of tumors and all dentigerous cysts. The expression rate was low. The results showed a higher expression rate of CDC7 in ameloblastoma and ameloblastic fibroma compared to AOT (p=0.009 and p=0.048, respectively). Ameloblastoma and ameloblastic fibroma were not significantly different in CDC7 expression (p=0.6). According to the results, the expression of the CDC7 protein in odontogenic tumors is low. The higher expression of CDC7 in ameloblastoma and ameloblastic fibroma in comparison with AOT confirms the hamartomatous growth of the latter, so it can be considered as a potential diagnostic marker. Future studies with a larger sample size are suggested to obtain a cut-off point for diagnostic purposes.

Molecular diagnostics in odontogenic tumors.

Odontogenic tumors (OTs) are rare, with an estimated incidence rate of less than 0.5cases per 100,000 per year. The causes of OTs remain unclear. Nonetheless, the majority of OTs seem to arise de novo, without an apparent causative factor. Although the etiopathogenesis of most OTs remains unclear, there have been some recent advances in understanding the genetic basis relating to specific histologies and clinical features. Molecular analyses performed by different techniques, including Sanger sequencing, next-generation sequencing, and allele-specific PCR, have uncovered mutations in genes related to the oncogenic MAPK/ERK signaling pathway. Genetic mutations in these pathway genes have been reported in epithelial and mixed OTs, in addition to odontogenic carcinomas and sarcomas. Notably, B‑RAF proto-oncogene serine/threonine kinase (BRAF) and KRAS proto-oncogene GTPase (KRAS) pathogenic mutations have been reported in ahigh proportion of ameloblastoma and ameloblastoma-related tumors and adenomatoid odontogenic tumors, respectively. To discuss how molecular profiling aids in diagnostic classification of odontogenic tumors. Molecular profiling of odontogenic tumors helps to identify patients for neoadjuvant therapies and reduces postoperative morbidity.

Peritoneal Tumors of Mullerian Origin: A Case Report and Review of the Literature

Primary peritoneal tumors share a common anatomical site and therefore their clinical manifestations are very similar. When peritoneal masses are detected, the tentative initial diagnosis is secondary to metastatic disease, which has been determined most frequently in the peritoneal cavity. However, primary peritoneal tumors should always be included in the differential diagnosis. Among the primary malignant peritoneal neoplasms, mesothelial tumors have been identified (malignant mesothelioma, well-differentiated papillary mesothelioma, benign cystic mesothelioma, adenomatoid tumor of the peritoneum), epithelial-type tumors (primary peritoneal serous carcinoma, primary peritoneal serous borderline tumor), tumors of the smooth muscle (disseminated peritoneal leiomyomatosis) and tumors of uncertain or undetermined origin (desmoplastic small round cell tumor, peritoneal solitary fibrous tumor).

Homeobox Genes in Odontogenic Lesions: A Scoping Review.

Homeobox genes play crucial roles in tooth morphogenesis and development and thus mutations in homeobox genes cause developmental disorders such as odontogenic lesions. The aim of this scoping review is to identify and compile available data from the literatures on the topic of homeobox gene expression in odontogenic lesions. An electronic search to collate all the information on studies on homeobox gene expression in odontogenic lesions was carried out in four databases (PubMed, EBSCO host, Web of Science and Cochrane Library) with selected keywords. All papers which reported expression of homeobox genes in odontogenic lesions were considered. A total of eleven (11) papers describing expression of homeobox genes in odontogenic lesions were identified. Methods of studies included next generation sequencing, microarray analysis, RT-PCR, Western blotting, in situ hybridization, and immunohistochemistry. The homeobox reported in odontogenic lesions includes LHX8 and DLX3 in odontoma; PITX2, MSX1, MSX2, DLX, DLX2, DLX3, DLX4, DLX5, DLX6, ISL1, OCT4 and HOX C in ameloblastoma; OCT4 in adenomatoid odontogenic tumour; PITX2 and MSX2 in primordial odontogenic tumour; PAX9 and BARX1 in odontogenic keratocyst; PITX2, ZEB1 and MEIS2 in ameloblastic carcinoma while there is absence of DLX2, DLX3 and MSX2 in clear cell odontogenic carcinoma. This paper summarized and reviews the possible link between homeobox gene expression in odontogenic lesions. Based on the current available data, there are insufficient evidence to support any definite role of homeobox gene in odontogenic lesions.

Immunohistochemical analysis of SHH, SMO and GLI-1 proteins in epithelial odontogenic lesions.

The present study analyzed the expression of proteins involved in the sonic hedgehog signaling pathway (SHH, SMO, and GLI-1) in benign epithelial odontogenic lesions (odontogenic keratocyst - OKC, ameloblastoma - AB, and adenomatoid odontogenic tumor - AOT) in order to identify the role of these proteins in the pathogenesis of these lesions. The sample consisted of 20 OKCs, 20 ABs, and 10 AOTs. The Kruskal-Wallis, Mann-Whitney U, and Spearman's (r) tests were used for statistical analysis, with the level of significance set at 5% (p < 0.05). The membrane/cytoplasmic expression of SHH was significantly higher in AB compared to AOT (p = 0.022) and OKC (p = 0.02). No differences were found in the membrane/cytoplasmic expression of SMO between the lesions studied. Regarding GLI-1, significant differences were observed at the nuclear level for AB and OKC compared to AOT (p < 0.0001). In addition, significant positive correlations were found between cytoplasmic and nuclear GLI-1 in AB (r = 0.482; p = 0.031) and OKC (r = 0.865; p < 0.0001), and between membrane/cytoplasmic SMO and cytoplasmic GLI-1 in AOT (r = 0.667; p = 0.035) and OKC (r = 0.535; p = 0.015). The results of this study confirm the participation of the sonic hedgehog signaling pathway in the pathogenesis of the lesions studied. Overexpression of SHH in ABs and nuclear expression of GLI-1 in ABs and OKCs indicate that these proteins contribute to the more aggressive behavior of these two lesions when compared to AOT.

EP42.08: Rare case of adenomatoid tumour of the uterus: diagnosis and treatment

EP42.08: Rare case of adenomatoid tumour of the uterus: diagnosis and treatment

ADENOID AMELOBLASTOMA: A CASE REPORT

Adenoid ameloblastoma is a rare variant of ameloblastoma that exhibits adenoid characteristics, resembling an adenomatoid odontogenic tumor. Despite few studies, the clinical, histopathologic, and behavioral aspects of this lesion are still unknown. A 57-year-old White female patient presented with a well-defined unilocular radiolucency between the roots of the right mandibular premolars, without intraoral changes. After an excisional biopsy, microscopic examination revealed islands of odontogenic epithelium surrounded by fibrous stroma. The peripheral columnar cells showed ameloblast-like differentiation with nuclear palisading and reverse polarization. A cribriform pattern was observed, as well as large duct-like spaces lined by columnar or cuboid cells and filled with eosinophilic mucoid material. A diagnosis of adenoid ameloblastoma was made and the patient is currently being followed up. Adenoid ameloblastoma is a rare variant of ameloblastoma that exhibits adenoid characteristics, resembling an adenomatoid odontogenic tumor. Despite few studies, the clinical, histopathologic, and behavioral aspects of this lesion are still unknown. A 57-year-old White female patient presented with a well-defined unilocular radiolucency between the roots of the right mandibular premolars, without intraoral changes. After an excisional biopsy, microscopic examination revealed islands of odontogenic epithelium surrounded by fibrous stroma. The peripheral columnar cells showed ameloblast-like differentiation with nuclear palisading and reverse polarization. A cribriform pattern was observed, as well as large duct-like spaces lined by columnar or cuboid cells and filled with eosinophilic mucoid material. A diagnosis of adenoid ameloblastoma was made and the patient is currently being followed up.

ADENOID AMELOBLASTOMA WITH DENTINOID: A CASE REPORT

A 58-year-old afrodescendant man presented a chief complaint of pain in tooth 48. Clinical examination revealed mobility, a deep periodontal pocket, and purulent secretion in teeth 48, 47, and 46. Cone beam computed tomography revealed a multilocular osteolytic lesion involving teeth 46, 47, and 48, promoting root resorption, with involvement of the ascending branch of the mandible. The diagnostic hypotheses were infected ameloblastoma or odontogenic keratocyst. Incisional biopsy revealed areas of typical ameloblastoma, in addition to epithelial spirals, duct-like microcysts, and the deposition of extracellular material consistent with dentinoid. The histopathologic diagnosis was adenoid ameloblastoma with dentinoid (AAD). The patient is in preoperative preparation for further definitive surgical procedure. AAD is an extremely rare odontogenic tumor with histologic features of ameloblastoma and adenomatoid odontogenic tumor and formation of hard tissue. Oral pathologists should have a thorough understanding of this histologic spectrum of ameloblastoma to avoid misdiagnosis. A 58-year-old afrodescendant man presented a chief complaint of pain in tooth 48. Clinical examination revealed mobility, a deep periodontal pocket, and purulent secretion in teeth 48, 47, and 46. Cone beam computed tomography revealed a multilocular osteolytic lesion involving teeth 46, 47, and 48, promoting root resorption, with involvement of the ascending branch of the mandible. The diagnostic hypotheses were infected ameloblastoma or odontogenic keratocyst. Incisional biopsy revealed areas of typical ameloblastoma, in addition to epithelial spirals, duct-like microcysts, and the deposition of extracellular material consistent with dentinoid. The histopathologic diagnosis was adenoid ameloblastoma with dentinoid (AAD). The patient is in preoperative preparation for further definitive surgical procedure. AAD is an extremely rare odontogenic tumor with histologic features of ameloblastoma and adenomatoid odontogenic tumor and formation of hard tissue. Oral pathologists should have a thorough understanding of this histologic spectrum of ameloblastoma to avoid misdiagnosis.

PERIPHERAL ADENOMATOID ODONTOGENIC TUMOR IN MAXILLA: A CASE REPORT

A 26-year-old female patient was referred to the stomatology clinic with a complaint of "lump in the gums." Physical examination revealed a discrete swelling laterally to the upper lip on the left side. On intraoral examination, a dome-shaped swelling that was sessile, normochromic, with an intact surface, painless and firm in the alveolar mucosa, above the crowns of teeth 23 and 24, and with a year of evolution was noted. Periapical and panoramic radiographic revealed a single radiolucent image with sclerotic borders limited by the adjacent bone and displacement of the premolar roots. Excisional biopsy was performed during the surgery, and upon detaching the buccal mucosa, it was noted that the lesion was located peripherally and that the cortical bone was intact, showing only an erosion where the lesion was lodged. The diagnostic hypotheses were odontogenic lesions with peripheral variant and other reactive lesions. Histopathologic analysis was conclusive to adenomatoid odontogenic tumor. The patient is being followed up with no recurrence.

ADENOMATOID ODONTOGENIC TUMOR OF THE JAW: CASE REPORT

A 35-year-old woman was referred to the oral and maxillofacial surgery service for excision of a benign intra-osseous lesion with histopathologic report of adenomatoid odontogenic tumor. On intraoral examination, the presence of a bulge along the bottom of the left mandibular body vestibule, with normal color and a smooth surface, was noted. Cone beam computed tomography showed an extensive hypodense image, with defined limits, comprising the region between teeth 31 and 35, expanding the buccal and lingual cortical bones with displacement of adjacent teeth and resorption of the 33-tooth apex root. Complete surgical excision of the lesion was performed under general anesthesia. At 1 year postoperative follow-up, the surgical site region showed a completely healed mucosa with no signs of recurrence and complete bone repair on the control radiograph.