Dysgenetic Polycystic Disease of the Salivary Glands: A Case Report of This Rare Entity Occurring for the First Time in the Minor Salivary Glands of the Tongue, and a Review of the Literature.

Dysgenetic polycystic disease, also known just as polycystic disease, is a very rare developmental abnormality affecting the salivary gland duct system. This entity has been reported in only 21 patients previously, although a careful review suggests only 16 patients have histological evidence of the disease. In previously reported cases, this lesion most commonly presents as either an incidental finding or as a swelling affecting the parotid glands bilaterally, or rarely the submandibular glands bilaterally. This case report details the first time dysgenetic polycystic disease is found affecting the minor salivary glands of the tongue in a 55-year-old male. Histochemical and immunohistochemical stains are presented and include positivity for AE1/AE3 and p63, and negativity for progesterone receptor, androgen receptor, mammaglobin, S100 and BRAF V600E. PAS-D and Congo Red highlight special microamyloid spheroliths structures intraluminally.

CD1a is not effective in distinguishing calcifying epithelial odontogenic tumors from amyloid-rich central odontogenic fibroma.

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.

Mucoepidermoid carcinoma showing continuity with the surface mucosa of the oral cavity: a report of 14 cases.

OBJECTIVE: We aimed to characterize the histology and the clinicodemographic features of mucoepidermoid carcinoma (MEC), showing continuity with the oral surface mucosa. STUDY DESIGN: We reviewed 138 cases of intraoral MEC to identify cases that showed continuity with the surface mucosa and compared their clinicodemographic findings with those of MECs not showing continuity. We compared the sex ratio using the 2-sample Z-test and compared the age distribution using the 2-sample Kolmogorov-Smirnov test. RESULTS: Of the 138 cases examined, 14 showed continuity with the surface mucosa. Their histology showed surface mucosa with an apparent transition to an infiltrating tumor with mucous, intermediate, and epidermoid tumor cells growing in solid and cystic patterns. Their clinical appearance ranged from firm submucosal nodules to erythematous to ulcerated lesions. They showed a strong female predilection (6:1) and sharply bimodal age distribution, with sharp peaks in the fourth and seventh decades. CONCLUSIONS: Mucoepidermoid carcinomas that show continuity have a demographic pattern distinct from that of conventional MECs, showing a striking female predilection and bimodal age distribution and suggesting a difference in etiology. Pathologists should remain aware that MEC in the oral cavity can have a histologic appearance of surface origin to reach the correct diagnosis.

Oral epithelium response of electronic cigarette users to electronic cigarette.

BACKGROUND: Electronic cigarettes are increasing in popularity, but there is only little information on their biologic effects on the oral epithelium, the initial site exposed to electronic cigarette smoke. METHODS: We assessed the oral epithelium response to electronic cigarettes by comparing the histology and RNA transcriptome (mRNA and miRNA) of healthy electronic cigarette vapers to nonsmokers. mRNA was assessed based on: (1) genome-wide; (2) genes previously identified as dysregulated in the oral epithelium of electronic cigarette vapers versus nonsmokers; (3) immune and inflammatory-related genes previously identified as dysregulated in the nasal epithelium of electronic cigarette vapers compared to nonsmokers; (4) genes previously identified as dysregulated in the small airway epithelium of nonsmokers following an acute exposure to electronic cigarette; and (5) genes related to the initial steps of COVID-19 infection. In addition, miRNA was assessed genome-wide. Comparisons were performed using analysis of variance, and Benajmini-Hochberg corrected p < 0.05 was considered significant. RESULTS: The histology of the epithelium, lamina propria and basal layer in electronic cigarette vapers appeared normal. Assessment of mRNA and miRNA, based on all gene lists, did not identify any genes significantly modified in the oral epithelium of electronic cigarette vapers in response to electronic cigarette use. CONCLUSION: An average history of 2 years of vaping results in no detectable histologic or transcriptome abnormalities in the buccal mucosa.

Atypical herpetic ulcerations in COVID-19 positive patients: A report of three cases.

Since the global COVID-19 pandemic, numerous reports have been made regarding oral lesions seen in COVID-19 patients. It remains unclear whether or not these are true manifestations of COVID-19. Here we present 3 patients who were hospitalized for COVID-19 and who developed atypical herpetic ulcerations during their treatment with remdesivir (Veklury) and steroids. In healthy patients, recurrent infection by herpes simplex virus (HSV) presents as lesions only on the lips and the attached oral mucosa. Atypical herpetic ulcerations are seen in immunocompromised patients. They present as large, stellate shaped ulcerations with raised borders and may involve movable mucosa. The 3 cases presented in this report resembled the atypical herpetic ulcerations typically seen in patients with immunosuppression. Through our report, we aimed to introduce the possibility of atypical herpetic ulcers in patients being treated for COVID-19, to allow for their timely diagnosis and to raise awareness of the underlying immunocompromised state.

Histologic and immunohistochemical identification of cribriform adenocarcinoma.

OBJECTIVE: To assess whether tumors originally classified as adenocarcinoma not otherwise specified (Adca-NOS), adenoid cystic carcinoma (ACC), and polymorphous low-grade adenocarcinoma (PLGA) could be reclassified as cribriform adenocarcinoma of the tongue and minor salivary gland (CATMSG). STUDY DESIGN: Tumors diagnosed between 1992 and 2014 at Oral Pathology Laboratory, Inc. (New York Presbyterian Hospital) were selected. Each tumor was reviewed by 3 oral pathologists to confirm the diagnosis of CATMSG. After review, 11 of 70 Adca-NOS, 5 of 38 ACCs, and 5 of 23 PLGAs met the histologic criteria for CATMSG. One case diagnosed as CATMSG in 2014 was used as a control, and the following stains were completed: epithelial membrane antigen, Hector battifora mesothelial-1, p16, and CAM 5.2. Eleven Adca-NOS, 2 ACCs (tissue was available for only 2 of the 5 adenoid cystic carcinoma cases), and 5 PLGAs underwent immunohistochemical (IHC) staining. Positive HMBE-1, p16, and CAM 5.2 staining along with negative staining for epithelial membrane antigen were considered supportive of a diagnosis of CATMSG. RESULTS: Based on histologic features and IHC results, we were able to reclassify 10 tumors (8 Adca-NOS and 2 PLGA) as CATMSG. CONCLUSION: CATMSG should be distinguished from other salivary gland tumors with similar histologic features. The diagnosis of CATMSG can be made on hematoxylin and eosin staining alone, but IHC analysis can be useful for confirmation.

Epithelioid Multinodular Osteoblastoma of the Mandible: A Case Report and Review of Literature.

Epithelioid multinodular osteoblastoma is a rare variant of osteoblastoma characterized by numerous nodules of epithelioid osteoblasts surrounding bony trabeculae, as well as clusters of epithelioid osteoblasts without osteoid formation. It commonly occurs in the gnathic bones of the face and spine, and has a male predominance. To date, only 26 cases of epithelioid multinodular osteoblastoma have been reported and described in detail in the literature. Lucas et al. (Hum Pathol 25:117-134, 1994) described 43 cases of a variant of osteoblastoma that he termed epithelioid multifocal osteoblastoma. These both likely represent the same entity. Here, we report another case of this rare variant of osteoblastoma. An 18-year-old male patient presents with a periapical radiolucency in the region of vital tooth #30. The surgeon's differential diagnosis for this radiolucent lesion was ameloblastoma versus cyst. An incisional biopsy of the lesion revealed well-vascularized fibrous connective tissue containing a multinodular tumor composed of collections of epithelioid cells with osteoblastic differentiation surrounding zones of hyalinization and bony trabeculae. Multinucleated giant cells and rare typical mitotic figures were noted. Additionally present within the tumor were clusters of epithelioid osteoblasts without bony trabeculae. Residual immature viable bone trabeculae were noted surrounding the tumor. A diagnosis of epithelioid multinodular osteoblastoma was rendered. In this paper we present a rare case of epithelioid multinodular osteoblastoma of the mandible, provide a general review of the literature, and highlight the unique histological features that help differentiate this tumor from tumors classified as conventional osteoblastoma, aggressive osteoblastoma, pseudoanaplastic osteoblastoma and, most importantly, low-grade or osteoblastoma-like osteosarcoma.

Sebaceous carcinoma of the maxillary gingival: First reported case involving the gingiva.

Sebaceous carcinoma of the oral cavity is extremely rare. To date, only 9 cases of primary sebaceous carcinoma involving the oral cavity have been reported in the English literature, with the majority of cases occurring on the buccal mucosa. This report describes a case of sebaceous carcinoma involving the maxillary gingiva. To our knowledge this is the first reported case of sebaceous carcinoma of the gingiva.

Malignant rhabdoid tumor of the floor of mouth: first reported case in the oral cavity of an adult.

Malignant rhabdoid tumors (MRTs) are exceedingly rare lesions. To our knowledge, only 2 cases have been reported in the oral cavity, with both examples occurring in infants. The current case is the third reported case of MRT of the oral cavity and the first reported case to occur in an adult at this location. The following report describes the clinical, histologic and immunohistochemical features of this tumor.

Two cases of CD30+, anaplastic lymphoma kinase (ALK)-negative anaplastic large cell lymphoma with oral manifestations.

Anaplastic large cell lymphoma (ALCL), a subgroup of T-cell non-Hodgkin's lymphoma, is an uncommon tumor exhibiting CD30 positivity and a characteristic immunophenotypic profile. Histologically, ALCL is characterized by the proliferation of large, anaplastic lymphoid cells with eccentric horseshoe- or kidney-shaped nuclei and one or more prominent nucleoli. Rare cases have been cited in the literature of ALCL presenting primarily in the oral cavity. The purpose of this article was to present 2 instructive cases of CD30+, anaplastic lymphoma kinase-negative ALCL with oral and systemic involvement.

AAOMP case challenge: A nonulcerated, slowly growing mass of the mandible.

A 38-year-old male presented with a slowly growing mass of the right mandible. The lesion displaced the mandibular canine and premolar teeth on the same side.

Oral ectomesenchymal chondromyxoid tumor: case report and literature review.

Ectomesenchymal chondromyxoid tumor (ECMT) of the oral cavity is a rare lesion. We describe the 33rd reported case in the current English-language literature. This patient had originally presented 5 years earlier with a tongue neoplasm with biopsy results consistent with nerve sheath myxoma. A general surgical pathologist gave the patient's current lesion a preliminary diagnosis of low-grade sarcoma. After further evaluation by an oral and maxillofacial pathologist, the diagnosis of ectomesenchymal chondromyxoid tumor was established. It is important for clinicians to have a better understanding of ECMT, so as to avoid future misdiagnosis of this entity.

Oral melanoacanthoma: a report of 10 cases, review of the literature, and immunohistochemical analysis for HMB-45 reactivity.

Oral melanoacanthoma (MA) is rare reactive mucosal lesion that, like cutaneous MA, demonstrates hyperplasia of spinous keratinocytes and melanocytes. Unlike MA of the skin, oral MA is unrelated to seborrheic keratosis. This series adds 10 cases to the limited number of previous reports of oral MA. The clinicopathologic features of the cases in this series are generally consistent with those previously reported in the literature; that is, although documented in various intraoral locations in patients of differing ethnicity, oral melanoacanthoma most often presents as an enlarging flat or slightly raised area of hyperpigmentation on the buccal mucosa of adult black women. The current series provides evidence of occurrence over a wider age range (5-77 years) than previously reported. Additionally, the reactivity of oral melanoacanthoma to HMB-45 was investigated. Strong HMB-45 reactivity was present in all cases, thus demonstrating its limited utility in distinguishing oral MA from malignant melanoma.