Search

Background: Intraosseous myofibroma of the jaw is a rare neoplasm of mesenchymal origin with limited comprehensive understanding. It typically affects patients in the first two decades of life with a male predilection., Case Presentation: This study presents a rare case of myofibroma mimicking an odontogenic lesion in a 2-year-old boy. The patient presented with an incidental finding of a painless swelling of the right mandibular ramus of unknown etiology. Imaging analysis revealed a solid, expansile lesion adjacent to the germinal zone of the right mandibular first molar. Histopathologic analysis and immunohistochemistry after incisional biopsy suggested a possible central odontogenic fibroma, and the patient underwent total enucleation, leading to the final diagnosis of intraosseous myofibroma. Follow-up examinations showed no evidence of recurrence., Conclusions: This report contributes to the understanding of myofibroma in pediatric patients and underscores the critical role of meticulous histopathologic examination for effective surgical planning and optimal patient outcomes., (© 2024. The Author(s).)

Background: Benign odontogenic lesions (BOLs) can cause severe jaw bone defects and compromise the quality of life of patients. Extracellular vesicles (EVs) are well-established and versatile players in mediating pathophysiological events. EVs in the interstitial space (tissue-derived EVs or Ti-EVs) possess higher specificity and sensitivity in disease-related biomarker discovery. However, the role of Ti-EV-loaded proteins in mediating the development of BOLs has remained untapped. Herein, we aim to explore the contribution of Ti-EV-loaded proteins to the development of BOLs., Methods: Samples were obtained from 3 with dental follicle, 3 with dentigerous cyst (DC), 7 with odontogenic keratocyst (OKC), and 3 patients with ameloblastoma (AM). Tissue-derived EVs were then extracted, purified, and validated using ultracentrifugation, transmission electron microscopy, and western blotting. Proteins from Ti-EVs were analyzed using LC-ESI tandem mass spectroscopy and differentially expressed proteins were screened, which was then validated by immunohistochemistry and immunofluorescence assays., Results: The protein profile of Ti-EVs in each group was mapped by LC-MS analysis. The top 10 abundant proteins in BOL-derived Ti-EVs were COL6A3, COL6A1, ALB, HIST1H4A, HBB, ACTB, HIST1H2BD, ANXA2, COL6A2 and FBN1. Additionally, unique proteins in the Ti-EVs from various lesions were identified. Moreover, focal adhesion kinase (FAK) and myeloid differentiation primary response 88 (MyD88) showed higher expressions in Ti-EVs derived from OKC and AM, which were confirmed by immunohistochemistry and immunofluorescence staining., Conclusions: Ti-EVs containing FAK and MyD88 might be related to the development of OKC and AM, which can be potential therapeutic targets., Competing Interests: Declaration of competing interests The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 Elsevier Masson SAS. All rights reserved.)

Objective: The present study aims to identify adenoid ameloblastoma (AdAM) from previously diagnosed cases of dentinogenic ghost cell tumor (DGCT), and gain insight to the possible relationship between AdAM and DGCT., Methods: DGCT cases diagnosed between 2006 and 2022 were re-examined with focus on the AdAM-like features., Results: A total of nine patients were included. Seven patients were males and two were females. The mean age was 38.0 ± 16.0 years. Five tumors occurred in the maxilla and four in the mandible, with a remarkable predilection for the posterior regions of both jaws. Microscopically, dentinoid material deposition was present in all cases. The ghost cells were absent in two cases. Rare ghost cells (<1%) were observed in three cases, and a higher proportion of ghost cells (5%-20%) were present in the remaining four cases. All cases showed prominent AdAM-like features, including duct-like structures, whorls/morules, and cribriform architecture. According to the diagnostic criteria proposed by the 2022 WHO classification, five cases without or with rare ghost cells were reclassified as AdAM. The other four cases including a higher proportion of ghost cells consisted of a mixture of DGCT and AdAM histopathologic patterns., Conclusion: Our results confirmed that the AdAM-like features had been largely overlooked in the diagnosis of DGCT at our institution in the past. Whilst a subset can now be more accurately classified as AdAM, some tumors showed overlapping morphological features between AdAM and DGCT, suggesting that the two may represent a spectrum of the same entity., Competing Interests: Declaration of Competing Interest The authors declare no conflicts of interest., (Copyright © 2023 Elsevier Masson SAS. All rights reserved.)

Pediatric odontogenic cysts and tumors are rare and often associated with developing or impacted teeth. Odontogenic cysts are broadly categorized as inflammatory or developmental while odontogenic tumors are classified histologically as epithelial, mesenchymal, or mixed tumors. This article will discuss the presentation, diagnosis, and treatment of odontogenic cysts and tumors in the pediatric population., Competing Interests: Disclosure The authors have nothing to disclose., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Odontogenic carcinoma with dentinoid (OCD) is a rare and controversial entity, which has not yet been included in the current World Health Organization classification of odontogenic lesions. Owing to the small number of reported cases, the clinicopathological characteristics, biological behavior, prognosis, and appropriate treatment strategies for OCD remain to be defined. Herein, we present an additional case of OCD with a focus on the differential diagnosis and review of the pertinent literature, in order to enable better recognition by oral clinicians and pathologists and further characterization of this entity., Case Presentation: This paper reports a case of OCD in the posterior mandible of a 22-year-old female. Radiography showed a well-defined unilocular radiolucency with radiopaque materials. The intraoperative frozen section pathology gave a non-committed diagnosis of odontogenic neoplasm with uncertain malignant potential. Then a partial mandibulectomy with free iliac crest bone graft and titanium implants was performed. Microscopically, the tumor consisted of sheets, islands, and cords of round to polygonal epithelial cells associated with an abundant dentinoid matrix. Immunohistochemically, the tumor cells were diffusely positive for CK19, p63, and β-catenin (cytoplasmic and nuclear). No rearrangement of the EWSR1 gene was detected. The final diagnosis was OCD. There has been no evidence of recurrence or metastasis for 58 months after surgery. We also provide a literature review of OCD cases, including one case previously reported as ghost cell odontogenic carcinoma from our hospital., Conclusions: OCD is a locally aggressive low grade malignancy without apparent metastatic potential. Wide surgical excision with clear margins and long-term period follow-up to identify any possible recurrence or metastases are recommended. Histopathological examination is essential to conclude the diagnosis. Special care must be taken to distinguish OCD from ghost cell odontogenic carcinoma and clear cell odontogenic carcinoma, as misdiagnosis might lead to unnecessary overtreatment. Study of additional cases is required to further characterize the clinicopathological features and clarify the nosologic status and biological behavior of this tumor., (© 2024. The Author(s).)

Background: Ghost cell odontogenic carcinoma (GCOC) is a rare malignancy characterized by the presence of ghost cells, preferably in the maxilla. Only slightly more than 50 case reports of GCOC have been documented to date. Due to the rarity of this tumor and its nonspecific clinical criteria, there is a heightened risk of misdiagnosis in clinical examination, imaging findings, and pathology interpretation., Case Presentation: A 50-year-old male patient presented to the hospital due to experiencing pain in his lower front teeth while eating for the past 2 months. Upon examination, a red, hard, painless mass was found in his left lower jaw, measuring approximately 4.0 cm × 3.5 cm. Based on the malignant histological morphology of the tumor and the abundant red-stained keratinized material, the preoperative frozen section pathology misdiagnosed it as squamous cell carcinoma (SCC). The surgical resection specimen pathology via paraffin section revealed that the tumor was characterized by round-like epithelial islands within the fibrous interstitium, accompanied by a large number of ghost cells and some dysplastic dentin with infiltrative growth. The malignant components displayed marked heterogeneity and mitotic activity. Additionally, a calcified cystic tumor component of odontogenic origin was observed. Hemorrhage, necrosis, and calcifications were present, with a foreign body reaction around ghost cells. Immunoreactivity for β-catenin showed strong nuclear positivity in tumor cells, while immunostaining was completely negative for p53. The Ki67 proliferation index was approximately 30-40%. The tumor cells exhibited diffuse CK5/6, p63, and p40 immunoreactivity, with varying immunopositivity for EMA. Furthermore, no BRAF V600E mutation was identified by ARMS-PCR. The final pathology confirmed that the tumor was a mandible GCOC., Conclusion: We have reported and summarized for the first time the specific manifestations of GCOC in frozen section pathology and possible pitfalls in misdiagnosis. We also reviewed and summarized the etiology, pathological features, molecular characteristics, differential diagnosis, imaging features, and current main treatment options for GCOC. Due to its rarity, the diagnosis and treatment of this disease still face certain challenges. A correct understanding of the pathological morphology of GCOC, distinguishing the ghost cells and the secondary stromal reaction around them, is crucial for reducing misdiagnosis rates., (© 2024. The Author(s).)

Dentinogenic ghost cell tumors are rare tumors, and few cases of them were reported in the literature. The presence of pigment in odontogenic lesions is a rare unexplained histological finding. In this report, we describe a unique case of a 7-year-old girl that was referred to the Department of Oral and Maxillofacial Surgery complaining of a left mandibular swelling. Clinical examination revealed a huge, ulcerated mass. Both incisional and excisional biopsies revealed a benign infiltrative odontogenic tumor with admixed ameloblast-like cells and pigmented ghost cells, consistent with a pigmented dentinogenic ghost cell tumor. To the best of our knowledge, this is the youngest case of intraosseous dentinogenic ghost cell tumor reported in the English literature and the second report of a pigmented variant. This rare variant should be included in the differential of pigmented odontogenic lesions to avoid misinterpretation, especially in small biopsies., (© 2024. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Background: Adenoid ameloblastoma with dentinoid (AAD) is a hybrid odontogenic tumor comprising histopathological presentation of ameloblastoma (AM) and adenomatoid odontogenic tumor (AOT) along with extracellular dentinoid material., Case Presentation: A 35-year-old female reported an asymptomatic swelling in the left mandibular posterior region. Histopathological examination revealed composite features of AM with AOT along with dentinoid material, which stained positively with Van Gieson and trichrome stains., Conclusion: The present case report serves to add further to the modicum of literature reports pertaining to AAD, which may gain recognition as a distinct entity in future World Health Organization (WHO) classification of odontogenic tumors., (Copyright © 2023 Copyright: © 2023 Indian Journal of Pathology and Microbiology.)

Odontogenic keratocysts (OKCs) are aggressive cystic jaw lesions with a high epithelial turnover rate and increased propensity for recurrence. Sometimes, the characteristic histopathological features of OKCs are either completely lost or seen focally due to previous marsupialization or inflammation. This research aimed to determine whether specific patterns of CK14 and Bcl-2 staining could assist in diagnosing OKCs with altered epithelial features and provide clues in elucidating their aggressive nature. CK14 expression was restricted to basal and suprabasal layers near satellite cysts and in areas showing subepithelial split. The entire epithelial lining showed CK14 expression in areas of inflammation and after marsupialization. The typical basal/suprabasal staining of Bcl-2 was lost in areas of inflammation and intensity is decreased in OKCs after marsupialization. These new findings could offer a hint into the biological nature and pathogenesis of OKCs. Because of its therapeutic consequences and high recurrence rate, proper recognition and diagnosis are essential for treatment planning., Competing Interests: The authors declare no conflict of interest., (Copyright © 2023 Wolters Kluwer Health, Inc. All rights reserved.)

Objective: Differential diagnosis between the non-calcifying variant of calcifying epithelial odontogenic tumor (NCLC-CEOT) and amyloid-rich central odontogenic fibroma (AR-COdF) has become a debate, particularly regarding the frequency of CD1a positivity in both entities. This has led to the growing consensus that CD1a-positive staining in AR-NC lesions confirms the diagnosis of AR-COdF. Here, we assess the validity of this consensus., Study Design: We collected the data of a case series of histopathologically distinct CEOTs, NCLC-CEOTs, and COdFs and stained them for CD1a and amyloid. Of the 9 CEOTs and NCLC-CEOTs, we diagnosed 4 as classic, 3 as associated with a dentigerous cyst, and 2 as combined CEOT/adenomatoid odontogenic tumors. Of the 9 COdFs, we diagnosed 3 as epithelial poor, 3 as epithelial rich (lacking amyloid), 2 as hyalinized with amyloid, and 1 as hyalinized without amyloid and assessed the staining results., Results: Of the 9 CEOTs and NCLC-CEOTs, 7 stained positively for CD1a, 5 diffusely and 2 focally. Notably, 2 classic NCLC-CEOTs stained strongly CD1a positive. All 3 of the epithelial-poor COdFs were predictably CD1a negative. Of the 6 remaining COdFs, 2 were CD1a positive, 1 hyalinized-with-amyloid COdF diffusely and 1 epithelial-rich-without amyloid focally., Conclusions: CD1a positivity, which occurs in classic CEOT and NCLC-CEOT, does not help distinguish between NCLC-CEOT and AR-COdF and is inconsistent in all AR-COdFs. The diagnosis of CEOT and AR-COdF should be guided by appropriate histopathologic criteria irrespective of CD1a staining or the presence of amyloid or calcifications., Competing Interests: Declarations of interest None., (Copyright © 2023 Elsevier Inc. All rights reserved.)

Medical records of dogs with an initial histopathological diagnosis of odontogenic sarcoma were reviewed for information on signalment, body weight, medical history, clinical signs, physical examination and diagnostic imaging findings, surgical procedure performed, and histologic characteristics. Twenty dogs were deemed to fit the criteria following the rigorous histology review process. These tumors were characterized by hypercellularity of the spindle cell component with less intervening stroma, mitotic activity, and variable presence of odontogenic epithelium and hard substance (cementum/dentin/bone). Non-invasive or locally invasive hypercellular peripheral odontogenic fibroma is suggested by the authors to describe these tumors. There were no signs of recurrence of any of the masses following surgical excisions with varying margins., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Background: Clinico-histopathologic assessment of patients with ameloblastoma and ameloblastic carcinoma remains the best diagnostic modality for the tumors. However, in cases where the criteria for arriving at a definitive diagnosis are not clearcut, the pathologist is faced with a dilemma and thus an imperative need for adjunct diagnostic methods., Objectives: To evaluate/compare the immunohistochemical expression of NM23 in classical, borderline (atypical) ameloblastoma and ameloblastic carcinoma and to assess usefulness of NM23 in closing diagnostic gaps between ameloblastoma and ameloblastic carcinoma., Methods: Twenty-four (24) cases of ameloblastoma, 10 ameloblastoma with classical histopathologic features, 8 with nonclassical histopathology [atypical], and 6 cases of ameloblastic carcinoma were selected from cases seen at the Oral Pathology Laboratory of the Lagos State University College of Medicine, Nigeria. NM23 immunostaining protocol was done on the selected tissue blocks and evaluated using Sinicrope method. Analysis was done using R language., Results: Positive NM23 staining was observed in all cases of ameloblastoma and ameloblastic carcinoma, with more intense staining observed in the stellate reticulum-like areas than in the ameloblast-like areas. Ameloblastic carcinoma stained intensely with NM23 (100%) compared with atypical cases (37.5%) and ameloblastoma (20.0%; p = 0.04). The mean aggregate score was also significantly higher in AC (11 ± 2.4; p = 0.01). The mean aggregate score was also significant amongst growth pattern of ameloblastoma (p = 0 0.02)., Conclusions: The findings in this study reveal the usefulness of NM23 in differentiating ameloblastoma from ameloblastic carcinoma; a more comprehensive study with a larger sample size is recommended to corroborate or refute the findings in this study., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 Elsevier Masson SAS. All rights reserved.)

The odontogenic keratocyst (OKC) is a common cystic lesion of the maxilla and mandible. Squamous cell carcinoma (SCC) arising from OKC or dysplasia occurring in OKC is rare. This study aimed to explore the incidence and clinical features of the dysplasia and malignant transformation of OKC. In this study, 544 patients diagnosed with OKC were collected. Among them, 3 patients were diagnosed as SCC arising from OKC, and 12 patients were diagnosed as OKC with dysplasia. The incidence was calculated. Clinical features were analyzed by chi-square test. In addition, a representative case reconstructing mandible with vascularized fibula flap under general anesthesia was reported. And cases reported before were reviewed. The incidence of the dysplasia and malignant transformation of OKC, which are highly associated with the clinical features of swelling and chronic inflammation, is about 2.76%. But the relevance between the dysplasia and malignant transformation and age, gender together with pain is not statistically high. All in all, the clinical features of swelling and chronic inflammation can be considered as characteristics of the dysplasia and malignant transformation of OKC. Although the pain isn't statistically relevant, it may be a dangerous clew. Also, combined with earlier literatures, the dysplasia and malignant transformation of OKC shows unique features of radiographs and histopathology., Competing Interests: Declaration of Competing Interest None of the authors have any conflicts of interest to disclose with regard to the preparation and publication of this article., (Copyright © 2023 Elsevier Masson SAS. All rights reserved.)

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., Competing Interests: Declaration of Conflicting InterestsThe authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Rationale: Ghost cell odontogenic carcinoma is a rare malignant odontogenic carcinoma characterized by the presence of ghost cells. It has a nonspecific clinical and radiographic presentation and can be locally destructive and invasive, sometimes with distant metastases. However, no effective systemic therapy is currently recommended for such patients., Patient Concerns: The patient has been unable to undergo surgery or radiotherapy again. Therefore, he was referred to our department for a more aggressive, multimodal systematic treatment program., Diagnoses: The histopathological examination was morphologically suggestive of ghost cell odontogenic carcinomas., Interventions: We report a case of locally invasive primary inoperable odontogenic shadow cell carcinoma in a 31-year-old Chinese man who achieved treatment with Toripalimab and chemotherapy, followed by Toripalimab maintenance therapy after 6 cycles., Outcomes: He achieved partial remission after treatment. The quality of life significantly improved after treatment. There were no grade 3/4 treatment-related adverse events during treatment., Lessons: This case presented that Toripalimab and chemotherapy may be a safe and effective systemic therapy for ghost cell odontogenic carcinoma., Competing Interests: The authors have no funding and conflicts of interest to disclose., (Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.)

Background: Ameloblastic carcinoma and metastasising ameloblastoma are rare epithelial odontogenic tumours with aggressive features. Distinguishing between these two lesions is often clinically difficult but necessary to predict tumour behaviour or to plan future therapy. Here, we provide a brief review of the literature available on these two types of lesions and present a new case report of a young man with an ameloblastoma displaying metastatic features. We also use this case to illustrate the similarities and differences between these two types of tumours and the difficulties of their differential diagnosis., Case Presentation: Our histopathological analyses uncovered a metastasising tumour with features of ameloblastic carcinoma, which developed from the ameloblastoma. We profiled the gene expression of Wnt pathway members in ameloblastoma sample of this patient, because multiple molecules of this pathway are involved in the establishing of cell polarity, cell migration or for epithelial-mesenchymal transition during tumour metastasis to evaluate features of tumor behaviour. Indeed, we found upregulation of several cell migration-related genes in our patient. Moreover, we uncovered somatic mutation BRAF p.V600E with known pathological role in cancerogenesis and germline heterozygous FANCA p.S858R mutation, whose interpretation in this context has not been discussed yet., Conclusions: In conclusion, we have uncovered a unique case of ameloblastic carcinoma associated with an alteration of Wnt signalling and the presence of BRAF mutation. Development of harmful state of our patient might be also supported by the germline mutation in one FANCA allele, however this has to be confirmed by further analyses., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Background: The World Health Organization's (WHO) chapter on odontogenic and maxillofacial bone tumors provides a global reference for diagnosis of these tumors. In the fifth edition, the inclusion of consensus definitions and development of essential and desirable diagnostic criteria help improve recognition of distinct entities. These are key enhancements since the diagnosis of odontogenic tumors is largely based on histomorphology which is taken in combination with clinical and radiographic appearances., Methods: Review., Results: Despite delineation of diagnostic criteria for ameloblastoma, adenoid ameloblastoma, and dentinogenic ghost cell tumor, a subset of these tumors continues to show overlapping histological features that can potentially lead to misdiagnosis. Accurate classification may be challenging on small biopsies, but potentially enhanced by refining existing diagnostic criteria and utilization of immunohistochemistry and/or molecular techniques in a specific cases. It has become clear that the clinical and histologic features of the non-calcifying Langerhans cell-rich subtype of calcifying epithelial odontogenic tumor and the amyloid-rich variant of odontogenic fibroma converge into a single tumor description. In addition, this tumor shows remarkable clinical, histological overlap with a subset of sclerosing odontogenic carcinoma located in the maxilla. Benign perineural involvement vs perineural invasion is an underexplored concept in odontogenic neoplasia and warrants clarification to reduce diagnostic confusion with sclerosing odontogenic carcinoma., Conclusion: While controversial issues surrounding classification and discrete tumor entities are addressed in the WHO chapter, ambiguities inevitably remain. This review will examine several groups of odontogenic tumors to highlight persistent knowledge gaps, unmet needs and unresolved controversies., (© 2023. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.)

Background and Aim: The purpose of this study was to determine the clinical and histological features and treatment of peripheral ameloblastoma. Peripheral Ameloblastoma is a rare benign odontogenic tumor that concerns soft tissue and have a typical extraosseous localization., Methods: Aim of this work is to show its clinical and histological characters, in order to define some useful information for differential diagnosis with other oral neoformations, comparing literature with our data, collected in ten years of clinical activity of Oral and Maxillofacial Surgery Unit of Policlinico Tor Vergata in Rome., Results: Prognosis of PA is certainly favourable, with a restitutio ad integrum close to 100%. In the period between October 2011 and November 2021, we reported 8 diagnoses of P.A. Medium age of the group with diagnosis of PA was 71,4 y with a SD: 3,65. P.A.'s incidence in our sample of patients was 0,26%., Conclusions: PA is a benign odontogenic tumor that requires a careful diagnosis, a complete surgical eradication and adequate follow up, because malignant evolution is rare but possible.

Purpose The purpose of this article is to present a case report of a periapical lesion found in a 24-year-old female who presented to a university dental hygiene clinic with a chief complaint of vestibular pain around tooth #22. Radiographically, the lesion appeared radiolucent and unilocular with well-defined borders and significant bone resorption. The area of radiolucency increased in size over a period of five weeks indicating an aggressive pathosis. Given the clinical and radiographic presentations, the differential diagnosis included an odontogenic keratocystic tumor (odontogenic keratocyst), central giant cell granuloma, periapical cyst, and squamous cell carcinoma. Biopsy revealed the diagnosis of a focal abscess. The patient's presenting signs and symptoms are reviewed. Management and contributing factors are discussed., (Copyright © 2023 The American Dental Hygienists’ Association.)

Adenomatoid odontogenic tumour is a rare benign, odontogenic tumour with uncertain histogenesis. Whether it is a hamartoma or a neoplasm is still a controversial topic. It is usually associated with an unerupted maxillary canine. Here, we discuss a follicular adenomatoid odontogenic tumour in a young girl with uncommon features such as it arose from two unerupted teeth and partial resorption of the roots of other normal teeth. The tumour was large enough to completely occupy the maxillary sinus. It was treated with enucleation and curettage by lateral rhinotomy approach., Keywords: adenomatoid tumor; case reports; hamartoma; odontogenic cysts.

Cemento-ossifying fibroma (COF) is a benign mesenchymal odontogenic tumor that commonly occurs in the tooth-bearing areas of the maxilla and mandible. This study reports a COF case located under the left buccal mucosa. The classification and differential diagnosis of this COF case were discussed based on the diagnosis and treatment of this case and previous literature.

It was to report a rare case of peripheral odontogenic myxoma removed with high-power diode laser and to do an extensive review of studies of odontogenic cysts and tumors treated with high-power laser (HPL). This is a rare case of a 63-year-old male patient with a peripheral odontogenic myxoma measuring approximately 10 cm in the attached gingiva region of tooth 16 removed with a high-power diode laser (808 nm, 3 W, in continuous mode, under constant suction, with 400-µm optical fiber). A literature review was also carried out looking for articles that involved the use of HPL in the treatment of odontogenic cysts and tumors, without restriction of year or language. In the present case, there was no need for suturing, no postoperative discomfort, and minimal bleeding during the procedure. In a 12-month follow-up period, there were no signs of recurrence. Only two cases of intra-osseous odontogenic myxomas treated with HPL and 10 cases involving other odontogenic cysts and tumors were found. All studies showing HPL to be effective in treating these lesions. Despite the different types of lasers used and different parameters, it is observed that lasers are effective in the treatment of odontogenic lesions., (© 2023. The Author(s), under exclusive licence to Springer-Verlag London Ltd., part of Springer Nature.)

Background: Orthokeratinized odontogenic cyst (OOC) is a rare developmental odontogenic cyst of the jaw. It was originally believed to be a variant of odontogenic keratocyst (OKC) but is now considered to be a distinct entity. OOC usually presents as a single lesion and recurs infrequently. On the other hand, OKC often presents with multiple lesions and displays locally aggressive behavior and a high recurrence rate associated with the protein patched homolog 1 (PTCH1) gene mutation. Multiple OOC cases are extremely rare and seem to be aggressive, but their pathogenesis is not fully understood. This study aimed to determine the clinical, pathological, and genetic characteristics of multiple OCC., Methods: Three cases of multiple OOC were evaluated for clinical and histological findings, and immunohistochemical expression of Ki-67 and Bcl-2. Furthermore, PTCH1 mutations were analyzed by next-generation sequencing using a custom panel to cover the entire exon of PTCH1., Results: The three cases of multiple OOC included two men and one woman with a mean age of 25.3 years old (range, 18-38 years old). Each case had two or three OOCs (total of seven OOCs), all of which were simultaneously detected. Of the seven OOCs that manifested as multiple jaw cysts, seven (100%) occurred in the posterior regions, four (57.1%) occurred in the mandible, and four (57.1%) were associated with an impacted tooth. Histological examination revealed cysts lined by orthokeratinized stratified squamous epithelium. Immunohistochemistry showed a low Ki-67 labeling index and no Bcl-2 expression in the seven OOCs. No pathogenic PTCH1 mutations were detected in any of the seven OOCs. None of the patients had any other symptoms or signs of recurrence at the last follow-up (6-60 months)., Conclusion: Multiple OOCs appeared to occur more often in younger patients than solitary OOC. Both multiple and solitary OOCs may be related diseases within the entity of odontogenic cysts. Multiple OOCs are clinicopathologically and genetically distinct from OKC., (© 2022. The Author(s).)

Context: Ameloblastoma is a benign epithelial odontogenic tumor with a tendency for recurrence. Some recurrent tumors could behave unpredictably with atypical microscopic changes., Aim: To study the clinicopathologic features and diagnoses of recurrent tumors of ameloblastoma., Settings and Design: This is a 5-year (2012-2017) retrospective study of 17 consecutive patients with recurrent tumors of ameloblastoma in a Teaching Hospital in Enugu., Methods and Material: The relevant clinicopathologic information, histology slides, and blocks were retrieved and reviewed. Descriptive analysis was used to determine the frequency, tables for categorical variables, and a Chi-square test was used to determine the statistical significance., Result: Recurrent tumors constituted 33.3% (17/51) of all confirmed diagnoses of ameloblastoma. The histopathologic diagnosis of the recurrent tumors includes conventional ameloblastoma 58.8% (10/17), unicystic ameloblastoma 5.9% (1/17), and ameloblastic carcinoma 35.3% (6/17). There was bilateral mandibular involvement in 60.0%, pain 58.8%, ulceration 29.4%, and matted lymph nodes 5.9%. Tumors with positive fluid aspirates 82.4% (14/17) yielded dark-brown fluids in 90.0% (9/10) of recurrent ameloblastomas and in 66.7% (2/3) of ameloblastic carcinomas., Conclusion: There was a high recurrence rate of recurrent tumors of ameloblastoma demonstrated in the present study, with a malignant presentation in some cases., Competing Interests: None

BACKGROUND Hypoproteinemia is caused by a decrease in protein level in the blood. This report describes 2 cases of hypoproteinemia associated with a gigantic odontogenic tumor. CASE REPORT Case 1, a 65-year-old man, visited our hospital with the chief concern of swelling in the right mandible, approximately 100 mm in diameter, and ameloblastoma was diagnosed. Abscess drainage was observed in the fistula of the tumors. Total protein and albumin levels were low before surgery. Hemimandibulectomy was performed under general anesthesia. The final pathological diagnosis based on the specimen was ameloblastic carcinoma. After surgery, the total protein and albumin levels improved and remained stable 6 months after the operation. At 21 months after surgery, there were no signs of recurrence. Case 2, a 60-year-old woman, visited our hospital with a chief concern of swelling in the left mandible, approximately 100 mm in diameter, and ameloblastoma was diagnosed. Abscess drainage was observed in the fistula of the tumors. The patient had a history of hypoproteinemia; preoperative levels of total protein and albumin were low, and edema of the body was observed before surgery. Hemimandibulectomy was performed under general anesthesia. The final pathological diagnosis based on the specimen was ameloblastoma. After surgery, the total protein and albumin levels improved, and remained stable 6 weeks after surgery. There were no signs of recurrence 9 months after surgery. CONCLUSIONS These 2 cases indicate the possibility that hypoproteinemia can be caused by plasma leakage from fistulas associated with gigantic odontogenic tumors.

Background: Ameloblastoma is a benign epithelial odontogenic tumor with a tendency for recurrence. The recurrent tumors behave unpredictably with atypical microscopic changes and likelihood of malignant transformation., Aims: To study the clinicopathologic features and diagnostic outcome of recurrent tumors of ameloblastoma in Enugu. This is a six-year (2012-2017) retrospective study of 17 consecutive patients with recurrent tumors of ameloblastoma in a Teaching Hospital in Nigeria., Materials and Methods: The relevant clinicopathologic information, histology slides, and blocks were retrieved and reviewed. Descriptive analysis was used to determine the frequency, tables for categorical variables, and a Chi-square test was used to determine the statistical significance., Result: Recurrent tumors constituted 33.3% (17/51) of all confirmed diagnoses of ameloblastoma. The diagnostic outcome of the recurrent tumors was conventional ameloblastoma 58.8% (10), unicystic ameloblastoma 5.9% (1), and ameloblastic carcinoma 35.3% (6). There was bilateral mandibular extension in 60.0% (9), pain 58.8% (10), ulceration 29.4% (5), and matted lymph nodes 5.9% (1). Tumors with positive fluid aspirates 82.4% (14) yielded dark-brown fluids in 90.0% (9) of recurrent ameloblastomas and in 66.7% (2) of ameloblastic carcinomas. Atypical peripheral hyperplasia, nuclear hyperchromatism, and increased vascularization were commonly observed in benign recurrences. The frequency of recurrence is significantly associated with the biological behavior of ameloblastoma P = 0.03., Conclusion: Recurrent tumors of ameloblastoma presented atypical features and malignant transformation., Competing Interests: None

Clear cell odontogenic carcinoma (CCOC) is a rare malignant odontogenic tumor. It is characterized by showing, on histopathological examination, clusters of vacuolated and clear tumor cells with epithelial differentiation surrounded by fibrocollagenous stroma and fibroblasts. The present study presents a rare clinical case of mandibular CCOC associated with an impacted tooth in a 26-year-old woman surgically treated with mandibulectomy and reconstruction with iliac crest bone graft. The patient has been followed up for 22 months without signs of recurrence. A search for case report/case series was carried out in the PUBMED database, as well as in the references of relevant previously published literature reviews. Ninety-six publications were identified, totaling 136 distinct cases reported. Female sex was the most affected (63.1%) with 63.3% of cases occurring in patients in the fifth, sixth, or seventh decades of life. The mandible was more affected than the maxilla (74.2%). Association of CCOC with impacted teeth was found in 2.4% of cases, thus rendering it a rare occurrence. The present case report corroborates the results of the survey regarding sex and anatomical location of the tumor; however, it contradicts the findings regarding age predilection. The case described is the fourth known occurrence of tooth impaction associated with the tumor and the first in a female. In conclusion, CCOC should be considered, as well as other malignancies, as a possible diagnosis of maxillary or mandibular intraosseous lesions even in unusual circumstances such as in association with impacted teeth and in young patients., (© 2021. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

BACKGROUND Dentinogenic ghost cell tumor (DGCT) is a rare, locally invasive odontogenic neoplasm, considered as a solid variant of the calcifying odontogenic cyst (COC). DGCT accounts for only 2% to 14% of all COCs and less than 0.5% of all odontogenic tumors. It is characterized by an ameloblastomatous odontogenic epithelium and the presence of ghost cells and dentinoid material. CASE REPORT A 42-year-old male patient presented to the clinic with recurrent pain and swelling in the left maxilla. The patient had a similar presentation 3 years before, which had been managed by excisional biopsy and was misdiagnosed as unicystic ameloblastoma. Examination revealed a mild swelling in the left cheek with boney expansion on the posterior area of the left maxilla, with mobility in adjacent teeth. Radiographic evaluation revealed a large, well-demarcated radiolucent lesion with a sclerotic border involving the left maxilla and associated with root resorption in the adjacent teeth. An incisional biopsy was performed, and a diagnosis of DGCT was made. Segmental maxillectomy with safe margins was conducted, considering the aggressiveness of this pathological entity. The defect was reconstructed using the buccal fat pad. Histopathology examination confirmed the diagnosis of DGCT, which was characterized by ameloblastomatous odontogenic epithelium, dentinoid material, and ghost cells. One month follow-up revealed good healing of the surgical site. The patient was scheduled for regular follow-up. CONCLUSIONS This case reports the aggressiveness and high recurrence rates of DGCT. It is recommended to treat DGCT aggressively with safe margins and a long-term follow-up.

Background: Odontogenic tumors (OTs) comprise a group of heterogeneous lesions ranging from hamartomatous or non-neoplastic tissue proliferation to benign or malignant neoplasms with metastatic potential. OTs are derived from epithelial, ectomesenchymal, and/or mesenchymal elements of tooth-forming ("odontogenic") tissues, which show variable clinical and histopathological features., Objective: Herein, the authors summarize the World Health Organization (WHO) 2022 classification of OTs and further highlight diagnostic tips and differential clues for the most common OTs., Conclusion: OTs may not be commonly encountered in the daily practice of many pathologists. This makes their diagnosis challenging as there is little practice in understanding the features required for their classification. However, diagnosing the vast majority of these lesions is not difficult provided the following aspects are considered: 1) the general knowledge of tooth development; 2) a few key histological observations; 3) very basic knowledge of the clinical and especially the radiographic features with which they are associated., (© 2022. The Author(s), under exclusive licence to Springer Medizin Verlag GmbH, ein Teil von Springer Nature.)

Background: Odontogenic tumors (OTs) are rare, with an estimated incidence rate of less than 0.5 cases 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 a high proportion of ameloblastoma and ameloblastoma-related tumors and adenomatoid odontogenic tumors, respectively., Objective: To discuss how molecular profiling aids in diagnostic classification of odontogenic tumors., Conclusion: Molecular profiling of odontogenic tumors helps to identify patients for neoadjuvant therapies and reduces postoperative morbidity., (© 2022. The Author(s).)

Adenoid ameloblastoma is a hybrid odontogenic tumour showing histopathological features of both ameloblastoma and adenomatoid odontogenic tumour (AOT), with approximately 40 cases reported in the literature. The aims of the report are to illustrate the diagnostic challenges of adenoid ameloblastoma using three new cases and to analyze evidence in literature to consider adenoid ameloblastoma as a new sub type of ameloblastoma. A literature review was performed with the key words-adenoid ameloblastoma, hybrid/composite odontogenic tumours, hybrid ameloblastoma and adenomatoid odontogenic tumour, ameloblastoma with inductive changes, dentinoid and dentinoma to select the cases compatible with the diagnosis of adenoid ameloblastoma. Out of the 40 cases reported in literature, 31 cases with sufficient information and 3 new cases were analyzed. Out of the 34 adenoid ameloblastomas majority of tumours (76.5%) occurred in adults with age ranging from 25 to 55 years. Slight female predilection with a male:female ratio of 0.9:1 was observed. Approximately, 64.7% occurred in the mandible. Radiologically, 82.4% of adenoid ameloblastomas presented as radiolucent lesions while 47.1% occurred with ill-defined margins and cortical perforation at diagnosis. Histopathologically, 70.8% of tumours presented as plexiform ameloblastomas, while duct like structures/glandular structures were the commonest feature supportive of adenomatoid odontogenic tumour observed in overwhelming majority of 95.9% of adenoid ameloblastomas. 91.6% of tumours showed inductive change in the form of dentinoid. Further, 45.4% of the tumours developed at least one recurrence following surgical excision. The report presents literature review based evidence to show the existence of adenoid ameloblastoma, which is demographically similar to conventional ameloblastoma but with histopathological differences and presenting with higher rate/multiple recurrences, indicating its biological aggressiveness. Thus, we would like to propose the inclusion of adenoid ameloblastoma as a sub type of ameloblastoma in the next revision of the WHO odontogenic tumour classification., (© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.)

Objectives: Nevoid Basal Cell Carcinoma Syndrome (NBCS) is a rare genetic condition affecting multiple organs including the maxillofacial and dental region. The surgical removal of the odontogenic keratocystic tumors (OKT), the high rate of recurrence leads to a iatrogenic tooth loss requiring dental care. The aim of this study is therefore to describe the dental and orthodontic management, and to assess the impact of surgery on facial growth and oral development., Methods: A retrospective study including 14 patients with GGS, followed at the Necker Enfants Malades Hospital. The study was carried out on the medical files and photographic records RESULTS: Patients developed on average 5.5 OKT (range: 1 to 11) and 1.7 recurrences (range:0 to 9) during the follow-up. The mean age at diagnosis of first OKT was 11.23 years (range: 6.75 to 16). KOTs were more frequently localized at the mandibular (30.9%) and maxillary molar level (25.1%). Forty-seven impacted teeth were extracted during the OKT removal. Eight patients out of 12 presented a class III skeletal relationship. The remaining ones had a skeletal class II associated with a hyperdivergent typology. Almost all patients had dental impactions with ectopic positions of the succedaneums tooth. At the inter-arch level, all patients needed orthodontic care, 3 patients did not begin their orthodontics. Orthodontic treatments began with an orthopedic phase followed by braces for the majority in 8 patients. Two patients had to undergo orthognathic surgery. Impacted teeth were treated by traction or extraction with further rehabilitation., Conclusion: The objective is not to simply compensate the iatrogenic hypodontia generated by the surgical procedure but to take into consideration the maxillofacial phenotype, skeletal characteristics and numerous intra- and inter-arch dental anomalies for a healthy oral management., Competing Interests: Declaration of Competing Interest No conflict of interest., (Copyright © 2021. Published by Elsevier Masson SAS.)

Peripheral ossifying fibromas (POFs) and peripheral odontogenic fibromas (POdFs) appear clinically similar but of different histogenesis. The novel marker SATB2 is involved in regulation of osteoblastic differentiation and phenotype. However, SATB2 expression has not been previously explored in POFs and POdFs. Given the potential for mineralized tissue formation in POFs and POdFs, and to more clarify the phenotype of the lesional cells, this study was aimed to immunohistochemically investigate SATB2 expression in POFs and POdFs. Fourteen cases of POF and POdF (7 cases each) were selected, stained for SATB2 immunohistochemically, and scored according to the percentage of positive lesional cells (0, no staining; 1 +, < 5%; 2 +, 5-25%; 3 +, 26-50%; 4 +, 51-75%; and 5 +, 76-100%), and the intensity of staining was graded as weak, moderate, or strong. The control group included the inflammatory fibrous hyperplasia-like area present in two cases, 1 case fibroma, and 1 case giant cell fibroma. Moderate to strong, and diffuse SATB2 nuclear immunoreactivity was detected in the lesional cells of all cases of POFs and POdFs with variable scores; 3-5 + for the POFs and 3-4 + for the POdFs (P = 0.101). The distribution of staining was more prominent in those lesional cells associated with the osteoid/calcification in the cases of POFs. No staining was noted in the control group. The lesional cells in both POFs and POdFs express SATB2 and may exhibit the osteoblastic-like phenotype. SATB2 staining may be useful for diagnosis of subsets of POFs with minimal or absent calcification and some POdFs with unidentifiable odontogenic epithelium., (© 2021. This is a U.S. government work and not under copyright protection in the U.S.; foreign copyright protection may apply.)

Background: Although pathology in the maxillary and mandibular bones is rare in young patients, the differential diagnosis is broad. The World Health Organization (WHO) updated its classification of maxillofacial bone pathology in 2017. Using these updated guidelines, a systematic review of common maxillofacial bone lesions in the pediatric population was performed., Methods: A PubMed search was conducted capturing English language articles from inception to July 2020. Thirty-one articles were identified that described the frequency of maxillofacial bone pathology. Data were extracted and organized using the WHO 2017 classification of odontogenic and maxillofacial bone tumors. Prevalence data were analyzed among diagnostic categories and geographical regions. The SAS version 9.4 was used to complete statistical analyses., Results: The articles included patients from birth to a maximum age of 14 to 19 years. The most common odontogenic cysts included radicular cyst (42.7%) and dentigerous cyst (39.0%) followed by odontogenic keratocyst (15.0%). Among odontogenic bone tumors, odontoma (49.3%) was most common followed by ameloblastoma (29.1%). The most common nonodontogenic bone tumor was fibrous dysplasia (42.4%), and the most common malignant bone tumor was osteosarcoma (75.0%). Significant variations were found by geographic region, with dentigerous cyst more common than radicular cyst, and ameloblastoma more common than odontoma in African and Asian countries (P < 0.0001)., Conclusions: This systematic review uses the WHO 2017 guidelines to classify common odontogenic and nonodontogenic maxillofacial bone lesions around the world. Pathogenesis, presentation, and available treatment options for the most common maxillofacial bone lesions are reviewed., Competing Interests: The authors report no conflicts of interest., (Copyright © 2021 by Mutaz B. Habal, MD.)

Background: Ectopic odontogenic tumours are rare and difficult to diagnose. Consequently, they are occasionally misdiagnosed as other tumours and overtreated. Dentinogenic ghost cell tumours (DGCTs) are odontogenic neoplasms characterised by a CTNNB1 mutation, ghost cell appearance, and dentinoid-like calcification. Herein, we present a case of ectopic DGCT on the floor of a patient's mouth, providing reliable clinicopathological and genetic evidence of its odontogenicity for the first time., Case Presentation: A 72-year-old man presented with painless sublingual swelling. Imaging revealed a multi-lobulated, solid-cystic mass on the floor of his mouth. Cytological evaluation showed folded epithelial clusters composed of basaloid cells, keratinised material, and calcification. Histological analysis revealed a multi-cystic, cribriform to solid nest, with an odontogenic satellate reticulum-like epithelium, including ghost cells and dentinoid matrix deposition. Immunohistochemical analysis found that CK19, CK5/6, bcl-2, and p63 were diffuse positive, β-catenin was focal positive in the nuclei, and the cells in the dentinoid matrix were positive for DMP1. The CTNTTB1 mutation was detected, leading to the final diagnosis of ectopic DGCT. There was no recurrence during the 6-month follow-up., Conclusions: Overall, we have presented a comprehensive clinical overview of DGCT and identified its pathological and genetic features. This report will aid in the recognition of this rare disease in the future and help to avoid misdiagnosis and overtreatment., (© 2022. The Author(s).)

Background: Granular cell odontogenic tumor (GCOT) is a rare neoplasm with about 45 cases reported in the literature. It usually occurs in the posterior mandible of middle-aged women., Case Presentation: We report a case of asymptomatic GCOT in the posterior mandible of a 28 years old female and provide a literature review of GCOT cases. Some unusual features such as root resorption, displacement of inferior tooth canal, and multilocular appearance were considerable in this case., Conclusions: Complete surgical excision of the lesion was beneficial for the patient., (© 2022. The Author(s).)

(1) Background: Many radiolucent jaw lesions exist, and they often show a radiographic resemblance, rendering diagnosis a challenging act. Closely related lesions should be frequently mentioned together in the academic literature, which might be helpful for junior practitioners in determining their differential diagnosis. The usefulness of bibliometric analysis in this respect has yet to be demonstrated. (2) Methods: This study evaluated academic publications on radiolucent jaw lesions, as indexed by the Web of Science Core Collection database. The mentions of radiolucent jaw lesions were extracted from the complete bibliographic records of the publications, and co-word analyses were conducted with the aid of VOSviewer. (3) Results: Based on 1897 papers, visualization maps were synthesized to evaluate co-occurrences of the radiolucent jaw lesions. Ameloblastoma was frequently mentioned together with odontogenic keratocyst, dentigerous cyst, and radicular cyst. Osseous dysplasia was co-mentioned with osteomyelitis, ossifying fibroma, odontoma, fibrous dysplasia, and apical periodontitis. (4) Conclusions: The co-word analysis, a form of bibliometric analysis, could demonstrate a relatedness of radiolucent jaw lesions that could be considered at differential diagnosis.

Introduction: The aim of the present article was to review and depict the main radiological features of odontogenic keratocysts (OKCs), thus helping the differential diagnoses from other odontogenic cysts and neoplasms., Evidence Acquisition: A review of articles published between January 2000 and October 2020 using Medline and the MeSH Term "odontogenic keratocyst" in combination with the following terms "imaging," "radiology," "panoramic radiograph," and "computed tomography," was performed., Evidence Synthesis: Radiographically, OKCs are well-defined unilocular or multilocular radiolucencies bounded by corticated margins. Most lesions are unilocular; instead, multilocular OKCs represent about the 30% of cases, mainly involving the posterior mandible. When, particularly in large lesions, OKCs display a multilocular presentation with adjacent satellite cysts (daughter cysts) a "soap-bubble appearance" can be recognized., Discussion: Panoramic radiograph and CT still play an important role in the diagnosis and treatment planning of OKCs. Unfortunately, it may not be easy to differentiate OKCs from other odontogenic lesions, especially when they are small and unilocular., Conclusions: Histopathological findings are still necessary to obtain a definitive diagnosis.

Objectives: Calcifying odontogenic cysts (COC) and adenomatoid odontogenic tumors (AOT) have similar radiographic findings. We examined the radiographic and computed tomography (CT) images of patients histologically diagnosed with COC or AOT and identified their characteristic findings., Methods: The subjects included 12 patients histologically diagnosed with COC or AOT (one female and five males per group), who underwent CT at our hospital between Nov 1998 and Jun 2019. The location of the lesion, impacted tooth, bone expansion, root resorption, tooth migration, calcified body, and presence or absence of a high-intensity zone in the marginal area of the lesion were examined., Results: In patients with COC, five patients with COC exhibited bone expansion toward the buccal side. The lesion encompassing the crown was attached to the cement-enamel junction and contained a radiopaque lesion with a calcified body. In 6 patients with COC, irregularly shaped calcified bodies were observed with small tooth-like structures. In patients with AOT, all six patients with AOT exhibited bone expansion toward the buccal and lingual sides. The lesion encompasses a part of the tooth root or the entire tooth. Punctate calcification was observed within the lesion and the marginal area in three patients, and a high-intensity zone was observed in the marginal area of the lesion in two patients., Conclusion: We report imaging findings that may be characteristic of COC and AOT, suggesting that CT findings may be useful for differentiating between COC and AOT., (© 2021. The Author(s), under exclusive licence to Japanese Society for Oral and Maxillofacial Radiology and Springer Nature Singapore Pte Ltd.)