Keratoameloblastoma of the lower jaw: A rare challenging entity.

Ameloblastoma is the second most common benign odontogenic tumor with various histopathologic features. Except for the unicystic type of ameloblastoma, the different microscopic patterns of this tumor show no significant correlation with long-term clinical behavior. During recent decades, additional challenging subtypes of ameloblastoma, including "Keratoameloblastoma" (KA), have been introduced in the literature. Here, we present a case of KA and discuss the important diagnostic microscopic features.

Cyst Formation After Subepithelial Connective Tissue Grafting: Management and Review.

Lesion formation after soft tissue gingival grafting is a rare but challenging clinical scenario to manage. This report presents a unique case of cyst formation after connective tissue grafting. All previously reported cases are confined to the mandibular labial lateral-canine space, whereas the present case is the first found in the maxilla. These cysts manifest clinically 9 months to 1.5 years after grafting and may communicate with the surface, as evidenced by sinus tract or cystic discharge. Because of the unique nature of these lesions with respect to clinical history, appearance, symptoms, and location, the differential diagnosis should be limited. The treatment recommendation is complete surgical excision, which should eliminate the risk of recurrence. In this report, a novel case is presented, and the literature is reviewed to discuss etiology and provide treatment recommendations. Int J Periodontics Restorative Dent 2023;43:257-263. doi: 10.11607/prd.5300.

Mucosal Melanoma In Situ of the Oral Cavity: A Case Report and Systematic Review of the Literature.

Objective: Malignant mucosal melanomas of the head and neck comprise a very small portion of all melanomas, particularly in the oral cavity. These lesions are associated with high rates of local recurrence, distant metastasis, and a very poor 5-year survival rate; however, the clinical outcomes of mucosal melanoma in situ of the oral cavity are unclear. Therefore, we present a case report of mucosal melanoma in situ and a systematic review of the literature to shed light on this rare but important disease. Methods: PubMed, Scopus, and CINAHL were searched per the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Studies were only considered for inclusion if they described oral cavity melanoma in situ and documented specific data pertaining to treatment including modality, lesion size, or outcomes. Results: A total of 28 reported cases from the literature fulfilled the inclusion criteria, as well as one case from our own institution. Men comprised the majority (64.3%) of the cases, and the average age at presentation was 57.4 years. The hard palate was the most common location, and most cases were treated with surgical excision. Eight had no evidence of disease after a minimum of six months of follow-up, one reported spread to the cervical lymph nodes, and only one reported progression with distant metastasis. Conclusion: Oral mucosal melanoma in situ is a rare entity and most commonly treated with surgical excision. High rates of recurrence necessitate long term follow-up. Further studies may be useful to determine whether adjuvant therapy may play a role in reducing recurrence.

Common Oral Lesions.

Familiarity with common oral conditions allows clinicians to observe and treat patients in the primary care setting or refer to a dentist, oral surgeon, otolaryngologist, or other specialist. Recurrent aphthous stomatitis (canker sores) is the most common ulcerative condition of the oral cavity. Recurrent herpes simplex labialis and stomatitis also commonly cause oral ulcers. Corticosteroids, immunocompromise, antibiotics, and dentures can predispose patients to oral candidiasis. Benign migratory glossitis (geographic tongue) occurs in up to 3% of the population but generally lacks symptoms, although some people experience food sensitivity or a burning sensation. Hairy tongue is associated with a low fiber diet, tobacco and alcohol use, and poor oral hygiene in older male patients. Generally, hairy tongue is asymptomatic except for an unattractive appearance or halitosis. Tobacco and alcohol use can cause mucosal changes resulting in leukoplakia and erythroplakia. These can represent precancerous changes and increase the risk of squamous cell carcinoma. Mandibular and maxillary tori are common bony cortical outgrowths that require no treatment in the absence of repeat trauma from chewing or interference with dentures. Oral lichen planus occurs in up to 2% of individuals and can present as lacy reticulations or oral erosions and ulcerations. Traumatic buccal mucosal fibromas and labial mucoceles from biting can be excised.

Calcified facial and maxillary arteries: Incidental radiographic findings indicative of Mönckeberg arteriosclerosis.

BACKGROUND AND OVERVIEW: Mönckeberg arteriosclerosis is a disease of unknown etiology characterized by dystrophic calcifications within the tunica media of small- and medium-sized arteries, leading to reduced arterial compliance. The authors report a case discovered incidentally on dental radiographs. CASE DESCRIPTION: A 78-year-old man with a complex medical history was seen for routine oral health care. Panoramic and bite-wing radiographs revealed a tortuous, linear calcification in the area of the left mandibular first molar anterior to the angle of the mandible and suggestive of a calcified facial artery. CONCLUSIONS AND PRACTICAL IMPLICATIONS: Medical radiologists have used the presence of arterial calcifications to determine the severity and prognosis of such diseases as primary hyperparathyroidism, secondary hyperparathyroidism, coronary artery disease, and diabetes. The presence of Mönckeberg arteriosclerosis on dental radiographs can help oral health care professionals identify patients with undiagnosed systemic disease.

Plexiform Schwannoma of the Oral Cavity: Report of Eight Cases and a Review of the Literature.

Plexiform schwannoma represents an unusual schwannoma variant, characterized by multinodular growth grossly and/or microscopically. A review of the English-language literature reveals only 28 previously reported cases involving the oral cavity, and herein we present 8 additional cases. Among these 36 patients, the average age at diagnosis was 28 years (range 5 to 62 years), with a female-to-male ratio of 1.4:1. The most frequently involved sites were the tongue (n = 13) and lip (n = 11). Lesion duration prior to presentation averaged 5.3 years (range, 6 weeks to 26 years). The average lesion size was 2.1 cm (range, 0.3 to 16 cm). The typical clinical presentation was a painless mass, although infrequent findings included pain/discomfort, paresthesia, difficulty chewing, and limited buccal mobility. All cases clinically appeared as a solitary mass or localized cluster of tumor nodules, with the exception of one patient who had neurofibromatosis 2 (NF2) and exhibited two distinct nodules on the tongue and buccal mucosa. In addition, extraoral neural neoplasms were evident in four patients, including three with NF2. Typical microscopic findings included multiple well-circumscribed tumor nodules, each surrounded by a perineurium-derived capsule with immunoreactivity for epithelial membrane antigen. The nodules contained characteristically bland and diffusely S-100-positive spindle cells arranged in Antoni A and B patterns; however, modest nuclear pleomorphism was evident in three cases. Most patients (n = 23) were treated by excision or enucleation and curettage, and three patients experienced recurrence. Unlike plexiform neurofibromas, plexiform schwannomas exhibit only a weak association with neurofibromatosis and have no known malignant potential.

Grading systems of oral cavity pre-malignancy: a systematic review and meta-analysis.

PURPOSE: Oral potentially malignant disorders (OPMDs) may have varying degrees of oral epithelial dysplasia (OED). Traditional grading schemes separate OED into three-tiers (mild, moderate, and severe). Alternatively, a binary grading system has been previously proposed that stratifies OED into low-risk and high-risk categories based on a quantitative threshold of dysplastic pathologic characteristics. This systematic review evaluates the predictive value of a binary OED grading system and examines agreement between pathologists. METHODS: This meta-analysis queried 4 databases (PubMed, Ovid-MEDLINE, Cochrane, and SCOPUS) and includes 4 studies evaluating binary OED grading systems. Meta-analysis of proportions and correlations was performed to pool malignant transformation rates (MTR), risk of malignant transformation between OED categories, and measures of interobserver agreement. RESULTS: Pooled analysis of 629 lesions from 4 different studies found a six-time increased odds of malignant transformation in high-risk lesions over low-risk lesions [odds ratio (OR) 6.14, 95% 1.18-15.38]. Reported ORs ranged from 2.8 to 22.4. The overall MTR was 26.8%, with the high-risk and low-risk lesions having MTRs of 57.9% (95% CI 0.386-0.723) and 12.7% (95% CI - 0.210 to 0.438), respectively. Pooled unweighted interobserver kappa values for the binary grading system and three-tiered system were 0.693 (95% CI 0.640-0.740) and 0.388 (95% CI 0.195-0.552), respectively. CONCLUSION: Binary grading of OED into low-risk and high-risk categories may effectively determine malignant potential, with improved interobserver agreement over three-tiered grading. Improved grading schemes of OED may help guide management (watchful waiting vs. excision) of these OPMDs.

Oral Leiomyomatous Hamartoma of the Anterior Maxillary Gingiva.

INTRODUCTION: Oral leiomyomatous hamartoma (OLH) is a rare developmental tumor-like anomaly of the oral cavity. CASE PRESENTATION: This report documents a 4-year-old female who was diagnosed with an OLH of the palatal gingiva between the maxillary central incisors. This lesion was treated successfully by local surgical excision. CONCLUSION: Because many of the reported cases of this lesion have developed on the anterior maxillary gingiva, it is important for the periodontist to be aware of this entity.

Oral cavity and oropharyngeal squamous cell carcinoma--an update.

Oral cavity squamous cell carcinoma (OC-SCC) is the most common malignancy of the head and neck (excluding nonmelanoma skin cancer). Recent trends have shown a dramatic rise in the incidence of oropharyngeal squamous cell carcinoma (OP-SCC), with a marked increase in lesions related to human papillomavirus infection. This update presents the latest evidence regarding OC-SCC and OP-SCC. In particular, the authors compare and contrast tumors at these two sites with respect to epidemiology, etiopathogenesis, clinicopathologic presentation, clinical assessment, imaging, management, and prognosis. It is important for clinicians to be aware of differences between OC-SCC and OP-SCC so that appropriate patient education and multidisciplinary care can be provided to optimize outcomes.

Melanotic Neuroectodermal Tumor of Infancy: A Systematic Review.

Melanotic neuroectodermal tumor of infancy (MNTI) is a rare tumor, usually diagnosed within the first year of age, with a predilection for the maxilla. Although the tumor is usually benign, its rapidly growing nature and ability to cause major deformities in surrounding structures necessitate early diagnosis and intervention. It is important that medical and dental specialists are prepared to make the diagnosis and proceed with appropriate intervention. The authors performed a systematic review of the 472 reported cases from 1918 through 2013 and provided a comprehensive update on this rare entity that can have devastating effects on young patients. This investigation uncovered age at diagnosis as an important prognostic indicator, because younger age correlated with a higher recurrence rate. The authors also present a case report of a 5-month-old girl diagnosed with MNTI and review her clinical presentation and imaging and histopathologic findings.

Surface Papillary Epithelial Hyperplasia (Rough Mucosa) is a Helpful Clue for Identification of Polymorphous Low-Grade Adenocarcinoma.

The purpose of this study is to evaluate surface papillary epithelial hyperplasia, a microscopic finding that corresponds to the clinical finding of rough or stippled mucosa, as a predictor of polymorphous low-grade adenocarcinoma (PLGA). We conducted a retrospective review of minor salivary gland neoplasms submitted to our biopsy service from 1991 to 2013. Our review was limited to lesions involving the oral cavity/soft palate with the following diagnoses: PLGA, pleomorphic adenoma (PA), mucoepidermoid carcinoma (MEC), and adenoid cystic carcinoma (ACC). A total of 202 minor salivary gland neoplasms were included in the study. Among cases in which surface epithelium was present for evaluation (n = 112), surface papillary epithelial hyperplasia was evident in 30 % of PLGA and 1 % of non-PLGA (i.e., MEC, ACC, PA). The greater frequency of surface papillary epithelial hyperplasia in the PLGA versus non-PLGA cases and in the benign versus malignant cases was significant (p = .0001 and p = .041, respectively). The sensitivity and specificity of papillary epithelial hyperplasia for PLGA were 30 % (95 % confidence interval (CI) 11.97-54.27 %) and 99 % (95 % CI 94-99.82 %), respectively. The clinical presentation of PLGA appeared relatively nonspecific, with all analyzed tumor types exhibiting a predilection for females, middle-aged to older adults, palatal location, pink/tan/normal color, and firm consistency. In conclusion, papillary epithelial hyperplasia was evident in only a minority of PLGA. However, when present within the context of a palatal salivary gland neoplasm, it appears to indicate a high probability of PLGA. Accordingly, rough mucosa may be a useful clinical pearl for identification of PLGA.

Odontogenic carcinoma with dentinoid: a new odontogenic carcinoma.

Dentinoid is an integral part of some odontogenic tumors. This article describes the clinico-pathological features of three cases of odontogenic carcinomas with dentinoid (OCD). A comparison of these with previously reported cases of dentinoid-producing epithelial odontogenic tumors allowed us to identify another six cases that may be considered as examples of OCD. Six cases occurred in the mandible and three in the maxilla, all developing behind the canines. There was no sex predilection (five men and four women; age range 14-61 years, mean 38.1). Pain or discomfort was mentioned in five cases, four of which showed tooth resorption. All cases appeared initially as well-defined radiolucencies, five of which showed variable amounts of calcified material. Recurrences were recorded in three instances, but no evidence of metastasis has been found. Seven cases were composed predominantly or entirely of clear cells, usually with minimal cellular atypia and variable mitotic activity; however, in all cases there was evidence of tumor infiltration into adjacent tissues, including the presence of perineural invasion in two tumors. Those cases in which no reference was made to the presence of clear cells exhibited evident mitotic activity and cellular pleomorphism. The epithelium in OCD does not produce buds or enamel organ-like structures such as those found in ameloblastic fibro-dentinoma and this tumor does not contain a mesenchyme-like connective tissue resembling dental papilla as observed in several mixed odontogenic tumors. Based on the existing data and the present series of cases, OCD appears to represent a distinct entity.

Evaluation of SOX2 as a potential marker for ameloblastic carcinoma.

OBJECTIVE: Ameloblastic carcinoma often poses diagnostic challenges in its separation from benign ameloblastoma with atypical cytologic features or an unusual clinical course. This study aimed to determine whether SOX2 (sex determining region-Y-related high mobility group box 2), a protein expressed in the epithelial basal proliferative zone in dentigerous cysts, is a marker for ameloblastic carcinoma as well as for high-grade transformation in ameloblastic neoplasms. STUDY DESIGN: Immunoperoxidase stains were performed according to a standard protocol. Immunostains were interpreted independently by 3 pathologists, and scores were recorded based on the percentage of staining and intensity of staining in the cells of interest. RESULTS: The diffuse strong nuclear staining pattern has 86.4% specificity (19 of 22) to indicate the presence of high-grade features and has 76.9% sensitivity (10 of 13) in comparison with benign counterparts (P = .0021). Although previously shown as a marker for ameloblastic neoplasms, calretinin is weakly positive in a few cells in 50% (5 of 10) of ameloblastic carcinoma and 43% (3 of 7) of benign ameloblastic neoplasms, with little value in highlighting the high-grade change (P = .36). CONCLUSIONS: The diffuse nuclear staining pattern of SOX2 is suggestive of a high-grade process in ameloblastic neoplasms. Numerous aggregates of cells harboring dense nuclear stain should raise concern for a malignancy.

Sclerosing rhabdomyosarcoma: report of a case arising in the head and neck of an adult and review of the literature.

Sclerosing rhabdomyosarcoma is a unique rhabdomyosarcoma variant, characterized by a prominent hyalinizing matrix. A notable pitfall is the potential for the unusual matrix and often pseudovascular growth pattern of this lesion to lead to confusion with other sarcoma types, including osteosarcoma, chondrosarcoma, and angiosarcoma. Here we report a case of sclerosing rhabdomyosarcoma arising in a 40-year old male. The tumor was centered in the pterygomaxillary fossa with extensive infiltration into adjacent structures. Fine needle aspiration yielded a preliminary diagnosis of high-grade pleomorphic undifferentiated sarcoma, for which he received neoadjuvant chemotherapy and surgical resection. Microscopic examination showed a malignant spindled to round cell neoplasm with prominent osteoid-like, hyaline stroma. Focal rhabdomyoblastic differentiation and diffuse immunoreactivity for desmin and myogenin aided in diagnosis. Nineteen months status post primary resection, the patient expired with multiple lung and bony metastases. Among 39 cases reported thus far (including the present case), there is a broad age range (0.3-79 years), with an average age at presentation of 27 years. The most commonly involved sites are the extremities (n = 19) and head and neck (n = 15). Most cases have been treated by resection, often combined with radiation and/or chemotherapy. Out of 31 cases with follow-up information provided, 6 patients developed local recurrence, 7 patients developed regional or distant metastasis, and 5 patients died of disease. Herein we discuss the ongoing controversy regarding how sclerosing rhabdomyosarcoma might best fit into existing rhabdomyosarcoma classification schemes, based upon current clinicopathologic and molecular genetic evidence.

Intravascular fasciitis: report of an intraoral case and review of the literature.

Intravascular fasciitis (IF) is an unusual variant of nodular fasciitis. It is characterized by intraluminal, intramural, and extramural involvement of small to large arteries or veins. Only three cases involving the oral cavity have been reported previously in the literature. Here we present an additional case of oral IF arising in the submucosa of the upper lip of a 20-year old female. Microscopic examination showed a well-circumscribed, nodular proliferation of spindle cells arranged in intersecting fascicles. Occasional multinucleated giants cells also were noted. The tumor was present within the lumen of an intermediate-sized artery and extended into adjacent smaller vessels, thereby creating a multinodular appearance. Extramural extension into the surrounding connective tissue also was observed. Among the 31 cases of IF reported thus far (including the present case), the majority (n = 23) arose in individuals in the 1st through 3rd decades, with a 1.4:1 male:female ratio. The most common sites of involvement were the head and neck (n = 11) and upper extremity (n = 11), followed by the lower extremity (n = 6) and trunk (n = 3). Conservative excision is standard treatment, although local recurrence has been reported in three cases. It is important for the pathologist to be aware of this lesion in order to avoid misdiagnosis as a sarcoma with angioinvasion.

Implication of RNA-binding protein La in proliferation, migration and invasion of lymph node-metastasized hypopharyngeal SCC cells.

The 5-year survival rate for oral cavity cancer is poorer than for breast, colon or prostate cancer, and has improved only slightly in the last three decades. Hence, new therapeutic strategies are urgently needed. Here we demonstrate by tissue micro array analysis for the first time that RNA-binding protein La is significantly overexpressed in oral squamous cell carcinoma (SCC). Within this study we therefore addressed the question whether siRNA-mediated depletion of the La protein may interfere with known tumor-promoting characteristics of head and neck SCC cells. Our studies demonstrate that the La protein promotes cell proliferation, migration and invasion of lymph node-metastasized hypopharyngeal SCC cells. We also reveal that La is required for the expression of ß-catenin as well as matrix metalloproteinase type 2 (MMP-2) within these cells. Taken together these data suggest a so far unknown function of the RNA-binding protein La in promoting tumor progression of head and neck SCC.

Multifocal lateral periodontal cysts: a report of 4 cases and review of the literature.

Lateral periodontal cyst (LPC) is a developmental jaw cyst of odontogenic origin. It has characteristic histopathologic features that are identical to those seen in the peripherally occurring gingival cyst of adults (GCA). The polycystic variant of LPC is termed the botryoid odontogenic cyst (BOC). The histogenetic origin of LPC is probably the rests of dental lamina in the alveolar bone. In the case of BOC, it might be that several adjacent epithelial rests simultaneously undergo cystic change and eventually form a polycystic lesion. Few previous examples of multifocal occurrence of LPC can be found in the literature. We report an additional 4 patients with this rare presentation of multiple, separate LPCs, and review the literature on this topic.

Oral mucoceles: a clinicopathologic review of 1,824 cases, including unusual variants.

PURPOSE: To review the clinicopathologic features of oral mucoceles, with special consideration given to unusual variants and exclusion of salivary duct cysts. MATERIALS AND METHODS: This was a retrospective consecutive case review of all oral mucoceles diagnosed by the Medical University of South Carolina, Oral Pathology Biopsy Laboratory, from 1997 to 2006. The following data were recorded: patient demographics, clinical features (anatomic location, color, size, and consistency), clinical impression, history of trauma, history of periodic rupture, and occurrence of unusual mucocele variants. RESULTS: During the study period, 1,824 oral mucoceles were diagnosed. Of these cases, 1,715 represented histopathologically confirmed cases that were not recurrences. There was no significant gender predilection, and the average age was 24.9 years. The most common locations were the lower labial mucosa (81.9%), floor of mouth (5.8%), ventral tongue (5.0%), and buccal mucosa (4.8%); infrequent sites included the palate (1.3%) and retromolar area (0.5%). The lesions most often were described as blue/purple/gray or normal in color. The mean maximum diameter was 0.8 cm (range, 0.1 to 4.0 cm). In 456 cases, a history of trauma was reported, and in 366 cases a history of periodic rupture was reported. Unusual variants included superficial mucoceles (n = 3), mucoceles with myxoglobulosis (n = 6), and mucoceles with papillary synovial metaplasialike change (n = 2). CONCLUSIONS: Our results confirm the findings of previous investigators regarding the major clinicopathologic features of oral mucoceles. Special variants of oral mucoceles occur infrequently, although it is important to recognize these variants to avoid misdiagnosis.