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The article focuses on advancing in the specialty of oral and maxillofacial pathology. The specialty of oral and maxillofacial pathology encompasses a wide range of activities ranging from laboratory services, clinical patient care, basic science research, clinical and translational research, and teaching. To advance specialty, there is need to strive to demonstrate value to patients, colleagues, and universities.

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. [ABSTRACT FROM AUTHOR]

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. [ABSTRACT FROM AUTHOR]

Purpose: To study the differential expression of Ceramide Synthase isoforms in oral mucosal dysplasia and its malignant transformation. Methods: We identified 12 cases of oral mucosal dysplasia with subsequent malignant transformation from the College of Dental Medicine, Division of Oral Pathology Database at the Medical University of South Carolina. These were formalin fixed paraffin embedded (FFPE) biopsy specimens of dysplasia from one oral subsite at one time point and a matching specimen from the same subsite and same patient at a later time-point with frank invasive squamous cell carcinoma. We also identified 42 cases of alveolar ridge keratosis which were age and gender matched to our set of cases with malignant transformation. Two sections, 5 microns each from the FFPE blocks to extract RNA using the High Pure RNA FFPE micro-kit from Roche Applied Sciences (Catalog No. 04823125001), followed by reverse transcription and q-PCR using Ceramide Synthase (CerS) isoform 1, 4 and 6 probes. Results: CerS 1 levels are higher in malignancy compared to the matched dysplasia samples whereas CerS 4 levels are lower in malignancy compared to the matched dysplasia samples. The levels of CerS6 are not significantly changed across the two sample sets. Conclusion: Ceramide synthase isoforms play a role in malignant transformation of oral mucosal dysplasia however the mechanism needs to be further studied and understood. This could be a novel marker for early detection of potentially malignant lesions in the oral mucosa and may serve as a target for chemoprevention strategies in oral squamous cell carcinoma. [Copyright &y& Elsevier]

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.

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.

This report describes the diagnostic process of a rare disorder in a Brazilian female child. The patient presented initially as a 7-year-old with multiple whitish submucosal nodules of a fibrous consistency in the lower lip, but with an inconclusive pathology report. When she turned 9 years of age, she presented with exacerbation of the original clinical findings, which then involved the upper lip, buccal mucosa, tongue and lingual frenulum. In addition, dermatological lesions were noted on the child's limbs and face, as well as a hoarse voice. Histopathological examination of the buccal mucosa revealed dense connective tissue with hyaline foci, which were positive with periodic acid-Schiff (PAS) staining and resistant to diastase digestion. Clinical and histopathological findings led to the diagnosis of a rare genetic disease with fewer than 300 reported cases - lipoid proteinosis. Magnetic resonance imaging revealed calcium deposits in her amygdaloid region of the brain, and nasopharyngolaryngoscopy revealed lesions in her vocal cords. The patient currently is stable and under multidisciplinary follow-up, but no treatment has been recommended to date.

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.

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.

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., (Copyright © 2014 Elsevier Inc. All rights reserved.)

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 (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.

This article presents various odontogenic cysts and tumors, including periapical cysts, dentigerous cysts, odontogenic keratocysts, orthokeratinized odontogenic cysts, lateral periodontal cysts, glandular odontogenic cysts, ameloblastomas, clear cell odontogenic carcinomas, adenomatoid odontogenic tumors, calcifying epithelial odontogenic tumors, squamous odontogenic tumors, ameloblastic fibromas, ameloblastic fibro-odontomas, odontomas, calcifying cystic odontogenic tumors, and odontogenic myxomas. The authors provide an overview of these cysts and tumors, with microsopic features, gross features, differential diagnosis, prognosis, and potential diagnostic pitfalls., (Copyright © 2011 Elsevier Inc. All rights reserved.)

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., (Copyright © 2011 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

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., (Copyright © 2011 Mosby, Inc. All rights reserved.)

Background: Teaching cancer prevention and detection is important in health professional education. It is desirable to select a comprehensive framework for teaching oral cancer (OC) prevention and detection skills., Methods: The PRECEDE-PROCEED model was used to design a randomized pretest and posttest study of the OC prevention and detection skills of dental students (n = 104). OC knowledge, opinions, and competencies were evaluated., Results: Second year students in the intervention group were more competent than those in the control group., Conclusions: The novel use of PRECEDE-PROCEED sets a precedent for designing a standardized OC curriculum for a wide range of health professional disciplines.

Background: Alveolar ridge keratosis (ARK) is not widely recognized as a distinct clinicopathologic entity, and it often is included in studies of oral leukoplakia (OL), thereby implying premalignant potential. The authors' objectives were to characterize the clinicopathologic features of ARK and determine whether removing ARK from the OL category would significantly affect the prevalence of dysplasia or carcinoma in OL., Methods: The authors conducted a retrospective consecutive case review of 477 ARK cases and 1,676 OL cases submitted to their biopsy service from 1995 through 2004. The authors defined ARK as a white plaque without erythema or ulceration and limited to the retromolar pad or edentulous ridge., Results: Microscopically, most ARK cases (97.9 percent) exhibited hyperkeratosis without dysplasia. The few dysplastic cases (2.1 percent) were associated with one or more of the following: verrucous appearance, tobacco or alcohol use, multiple OL lesions and previous oral squamous cell carcinoma. Excluding ARK from OL increased the percentage of OL cases exhibiting dysplasia or carcinoma from 20.2 percent to 24.8 percent. Including versus excluding ARK resulted in good but less than excellent agreement in the OL case definition (kappa = 0.6128)., Conclusions: ARK in patients without high-risk habits or other clinical warning signs appears to be a distinctly different lesion from OL, with a much smaller proportion of dysplasia or carcinoma evident among ARK versus OL cases. However, prospective studies are needed to confirm this hypothesis., Clinical Implications: Although most cases clinically consistent with ARK are benign hyperkeratoses, dysplasia or carcinoma can be excluded only by means of biopsy and histopathologic examination.

Patients with premalignant oral lesions have varying levels of risk of developing oral squamous cell carcinoma (OSCC), whose aggressiveness requires increased motility. Not known is if and how premalignant oral lesion cells acquire the increased motility characteristic of OSCC. This was addressed by immunohistochemical analysis of banked premalignant lesion tissues and by functional analyses using cultures established from premalignant oral lesions and OSCC. These studies showed premalignant oral lesion cells and OSCC to be more motile than normal keratinocytes. Concomitantly, levels of ceramide were reduced. The activity of the protein phosphatase PP-2A, which restricts motility and which can be activated by ceramide, was also diminished. This was due to IL-10 released from premalignant lesion cells. Treatment with a membrane-permeable ceramide restored PP-2A activity and blocked migration. These studies show an autocrine motility-stimulatory pathway that is mediated in premalignant lesion cells by IL-10 through its reduction of ceramide levels and inhibition of PP-2A activity.