Secretory Carcinoma of the Salivary Gland - Systematic Review of the Literature and Report of 2 Cases.

PURPOSE: Secretory carcinoma (SC) of the salivary gland, formerly known as mammary analogue secretory carcinoma, is an uncommon and fairly newly described low grade malignant neoplasm of the salivary gland. Given the small number of cases reported in the literature to date, treatment guidelines are scarce. This study aimed to describe the clinical characteristics of SC, discuss prior management strategies, and provide recommendations for future treatment. METHODS: We performed a systematic review of all the cases of SC reported in the literature since it was first recognized in 2010. Using Pubmed, Crossref, and Google Scholar, we identified all articles reporting cases of SC. RESULTS: We identified 657 cases of SC in 109 articles. In addition, we provided 2 new cases, for a total of 659 cases in 110 articles. To our knowledge, this is the largest review of cases of SC in the literature to date. We summarized the clinical characteristics of SC, as well as the nodal status, clinical management, recurrence rate, and death rate. CONCLUSIONS: SC occurs on average in middle age (with a large age range), presents most often initially as localized disease without metastasis, and has a low but not insignificant recurrence rate. Deaths have been reported. The generalized recommendations for treatment of SC are in line with those of other low-grade salivary gland malignancies.

Vascular Compromise After Soft Tissue Facial Fillers: Case Report and Review of Current Treatment Protocols.

The use of facial fillers for soft tissue augmentation is becoming a mainstream treatment modality for patients. Owing to the relative ease of administration, as well as the lucrative nature of such procedures, the number of providers offering facial fillers has been expanding. Although many adverse effects of facial fillers are minor and localized to the site of injection, 1 potential serious, albeit uncommon, adverse effect of facial filler treatment is avascular necrosis. In this article, we review soft tissue filler complications and describe the case of a 52-year-old female patient in whom vascular compromise developed after facial filler administration. In addition to reviewing complications and best practices for treatment management, we discuss anatomic considerations, present an overview of the most common filler materials, describe histologic changes with dermal fillers, and discuss litigation consequences with the use of these minimally invasive procedures. Although facial filler treatment is regarded as a minimally invasive and extremely safe procedure, it is not without complications. Avascular necrosis after soft tissue augmentation with facial fillers is exceedingly rare, with only a few cases reported in the literature, but practitioners who offer this procedure need to be well versed in treatment protocols.

Which Clinicopathologic Factors Affect the Prognosis of Gingival Squamous Cell Carcinoma: A Population Analysis of 4,345 Cases.

PURPOSE: The purpose of this study was to present the characteristics and survival outcomes of primary gingival squamous cell carcinoma (SCC). MATERIALS AND METHODS: We performed a retrospective cohort study using data from the Surveillance, Epidemiology, and End Results (SEER) tumor registry. Patients who received a diagnosis of primary gingival SCC were included in the analyses. The predictor variables were chosen from baseline demographic and tumor characteristics. The outcome of interest was survival, and Kaplan-Meier analyses were used to estimate rates of overall survival (OS) and disease-specific survival (DSS). Cox proportional hazards regression models were used for multivariate analyses. RESULTS: In total, 4,345 patients (mean age, 70.2 years) with primary gingival SCCs were identified. There was a significantly positive association between T category and both nodal and distant metastases. The 2-, 5-, and 10-year OS rates were 63.1%, 46.5%, and 28.1%, respectively, whereas the DSS rates were 78.2%, 70.7%, and 62.2%, respectively. Multivariate Cox proportional hazards regression showed that the independent predictors of overall and disease-specific death were older age, intermediate and high histologic grade, tumor size, nodal disease, and the absence of surgery. T4 classification was significantly associated with only DSS. Race, gingival location (maxillary vs mandibular), verrucous histology, and the presence of distant metastases did not significantly affect survival when all other predictors were controlled for. CONCLUSIONS: Older age at diagnosis, higher grade, increased tumor size, nodal disease, and the absence of surgery were each individually associated with lower OS and DSS. Because of low nodal disease rates, both T1N0M0 lesions and verrucous subtypes may be candidates for neck observation.

Stafne Bone Defect Involving the Ascending Ramus.

The Stafne bone defect (SBD) classically presents as a well-defined, radiolucent lesion in the molar-mandibular angle region beneath the level of the mandibular canal. Due in large part to this prototypical appearance, most cases of SBDs are radiographically diagnostic without the need for tissue biopsy. A diagnostic challenge may arise, however, when the SBD occurs in other locations in the mandible. The authors report a case of a 57-year-old male who presented with a SBD of the left ascending ramus. The lesion was found incidentally on a panoramic radiograph. Additional imaging studies confirmed a lingual concavity of the mandibular bone consistent with a SBD, and a retrospective study of the patient's records revealed the presence of the lesion 5 years prior without any radiographic alterations. To date, only 10 documented cases of an SBD involving the ascending ramus have been reported. The rarity of such a presentation may obscure the diagnosis and lead to unwarranted surgical procedures. It is important to recognize atypically located SBDs, because unlike other radiolucent lesions, imaging studies are diagnostic. With proper diagnosis, additional surgical procedures and any associated morbidities can be avoided.

A 3-Year-Old With Gingival Hemorrhage and Musculoskeletal Pain.

A 3-year-old had spontaneous gingival hemorrhage and bilateral limb weakness with inability to bear weight. He had no preceding oral trauma or recent infection, took no regular medications, and had no recent use of aspirin or nonsteroidal anti-inflammatory drugs; his diet was limited to primarily chicken nuggets and milk. What is the diagnosis and what would you do next?

Acellular Dermal Matrix as a Barrier for Guided Bone Regeneration of Dehiscence Defects Around Dental Implants: A Clinical and Histological Report.

The presence of hard tissue dehiscence and thin periodontal biotype in the esthetic area is a challenge that can jeopardize function and aesthetic outcome of implants. Here, we present a successful simultaneous soft tissue and bone regeneration of dehiscence defects in the maxillary incisor region. The novelty of this case lies in the simultaneous bone regeneration and soft tissue augmentation and the use of acellular dermal matrix as a barrier for regeneration and biotype conversion around dental implants. Reentry surgery 5 months after soft tissue and bone augmentation showed more than 95% new bone formation at the facial surface of implants and histological evidence of new vital bone formation. Biotype conversion from thin (<0.8 mm) to thick (2 mm) was noted in the area.

Ameloblastic Fibro-Odontoma in an Adolescent: A Case Report and Review of Literature.

Ameloblastic fibro-odontoma (AFO) is a rare benign odontogenic tumor with the histologic features of ameloblastic fibroma (AF) but also contains enamel and dentin. It is most commonly observed in the pediatric population. Distinction between AFO and AF becomes important as ameloblastic fibromas are associated with higher recurrence rates of up to 18%, and 35% of these recurrent lesions can undergo malignant transformation to ameloblastic fibrosarcoma. Hence, for amelobastic fibroma, conservative curettage is recommended for the initial lesion and marginal resection is considered for recurrent cases. In contrast, AFO can be treated with simple curettage and the recurrence rate is approximately seven percent. Malignant transformation of AFO is exceedingly rare. Therefore, the treatment and prognosis differs for these two histologically similar neoplasms. We present a case of a 17-year-old boy who was initially diagnosed with ameloblastic fibroma upon biopsy, with subsequent curettage specimen showing AFO, which carries a better prognosis.

Pyogenic Granuloma in the Tongue in a Five Year Old: A Case Report.

Pyogenic granuloma, also called lobular capillary hemangioma, is a benign vascular lesion of the skin and mucous membranes. While the majority of pyogenic granulomas in the oral cavity involve the gingiva, they also infrequently present at other sites including lips, oral mucosa, palate, and tongue. We report a case of a pyogenic granuloma of the tongue in a five-year old female patient.

Intraosseous mandibular clear cell odontogenic carcinoma with predominant small round blue cells: a potential diagnostic pitfall.

OBJECTIVE: Clear cell odontogenic carcinoma (CCOC) is a rare malignancy of the jaw, presenting significant diagnostic challenges. This report aims to highlight the complexities associated with biopsy-based diagnoses of oral and maxillofacial lesions, as demonstrated in a case of intraosseous mandibular CCOC initially suggestive of Ewing's sarcoma due to its presentation with small round blue cells. RESULTS: The patient, a 37-year-old male, presented with a mandibular lesion that on incisional biopsy was suggestive of Ewing's sarcoma. Subsequent, comprehensive histologic evaluation after definitive resection via mandibulectomy revealed a CCOC, characterized by a biphasic pattern of clear and basaloid cells. Histological examination confirmed the presence of glycogen-rich clear cells, supported by periodic acid-Schiff (PAS) staining and confirmed by PAS diastase staining, which demonstrated glycogen digestion. Immunohistochemistry was positive for AE1/AE3, p40, and p63, while negative for c-kit and CD34, confirming CCOC and excluding other malignancies such as Ewing's sarcoma, which would have been treated with neoadjuvant chemotherapy instead of primary surgical treatment as in CCOC. CONCLUSION: This case highlights the essential need for thorough histopathological evaluation and the value of a second opinion via additional histologic consultation, particularly due to the diagnostic challenges of heterogeneous lesions in the oral and maxillofacial region.

Ulcerative and Inflammatory Lesions of the Oral Mucosa.

Ulcerated and inflammatory lesions of the oral mucosa are not rare. A detailed patient medical and social history including habits and abuses, as well as the duration, location, focality and presence, or lack of local and/or systemic symptoms is critical in establishing a proper diagnosis. This article discusses the clinical presentation, management, and histopathologic characteristics of a variety of ulcerative and inflammatory lesions seen in the oral cavity.

Oral manifestations of COVID-2019-related multisystem inflammatory syndrome in children: a review of 47 pediatric patients.

BACKGROUND: Although much is still unknown about the full effects of COVID-19, literature from the early stages of the COVID-19 pandemic (spring and summer 2020) supports a postviral immunologic reaction resulting in a multisystem inflammatory syndrome in children (MIS-C). The purpose of this study was to report the rates of documented oral and oropharyngeal manifestations among these patients and to determine the association of these findings with other MIS-C symptoms. METHODS: The authors conducted a retrospective review of pediatric patients with COVID-19 who were admitted to the Morgan Stanley Children's Hospital of NewYork-Presbyterian. Patients fulfilling the Centers for Disease Control and Prevention criteria for MIS-C were included in this study. The documented signs, symptoms, and laboratory values were collected and compared with the presence of oral or oropharyngeal findings. RESULTS: The mean (standard deviation) age of MIS-C patients was 9.0 (5.0) years (range, 1.3-20.0 years), and there was no obvious sex difference (51.1% male, 48.9% female). With respect to oral findings, 23 patients (48.9%) had red or swollen lips, whereas only 5 (10.6%) had a strawberry tongue. Oral or oropharyngeal findings were associated significantly with the presence of systemic rash (P = .04) and conjunctivitis (P = .02). CONCLUSIONS: The presence of oral or oropharyngeal changes may be an early indicator of MIS-C and should be considered suggestive of MIS-C in the setting of COVID-19 infection. PRACTICAL IMPLICATIONS: Dental care providers may play an integral role both in the early detection of oral manifestations of MIS-C and in the identification of oral lesions in hospitalized patients with confirmed MIS-C.

MicroRNA-Based Cancer Mortality Risk Scoring System and hTERT Expression in Early-Stage Oral Squamous Cell Carcinoma.

We have previously constructed a novel microRNA (miRNA)-based prognostic model and cancer-specific mortality risk score formula to predict survival outcome in oral squamous cell carcinoma (OSCC) patients who are already categorized into "early-stage" by the TNM staging system. A total of 836 early-stage OSCC patients were assigned the mortality risk scores. We evaluated the efficacy of various treatment regimens in terms of survival benefit compared to surgery only in patients stratified into high (risk score ≥0) versus low (risk score <0) mortality risk categories. For the high-risk group, surgery with neck dissection significantly improved the 5-year survival to 75% from 46% with surgery only (p < 0.001); a Cox proportional hazard model on time-to-death demonstrated a hazard ratio of 0.37 for surgery with neck dissection (95% CI: 0.2-0.6; p=0.0005). For the low-risk group, surgery only was the treatment of choice associated with 5-year survival benefit. Regardless of treatment selected, those with risk score ≥2 may benefit from additional therapy to prevent cancer relapse. We also identified hTERT (human telomerase reverse transcriptase) as a gene target common to the prognostic miRNAs. There was 22-fold increase in the hTERT expression level in patients with risk score ≥2 compared to healthy controls (p < 0.0005). Overexpression of hTERT was also observed in the patient-derived OSCC organoid compared to that of normal organoid. The DNA cancer vaccine that targets hTERT-expressing cells currently undergoing rigorous clinical evaluation for other tumors can be repurposed to prevent cancer recurrence in these high-risk early-stage oral cancer patients.

Oral Pseudomyogenic Hemangioendothelioma: Case Report and Review of the Literature.

Pseduomyogenic hemangioendothelioma (PMH) is a vascular neoplasm of intermediate biological potential first described by Hornick and Fletcher (Am J Surg Pathol 35:190-201, 2011). Despite its initial categorization as a malignant entity, PMH often demonstrates an indolent behavior profile, and thus was classified as a rarely metastasizing endothelial neoplasm in the 2013 WHO Classification of Tumors of Soft Tissue and Bone. It is a tumor primarily of skin and soft tissue, with most reported cases involving the trunk or extremities. To date, only one case of PMH involving the oral cavity has been reported. Herein, we present a case of PMH involving the mandibular gingiva and vestibule of a 33-year-old female and discuss the salient features of this entity.

MicroRNA-based risk scoring system to identify early-stage oral squamous cell carcinoma patients at high-risk for cancer-specific mortality.

BACKGROUND: For early-stage oral squamous cell carcinoma (OSCC), there is no existing risk-stratification modality beyond conventional TNM staging system to identify patients at high risk for cancer-specific mortality. METHODS: A total of 568 early-stage OSCC patients who had surgery only and also with available 5-year clinical outcomes data were identified. Signature microRNAs (miRNAs) were discovered using deep sequencing analysis and validated by qRT-PCR. The final 5-plex prognostic marker panel was utilized to generate a cancer-specific mortality risk score using the multivariate Cox regression analyses. The prognostic markers were validated in the internal and external validation cohorts. RESULTS: The risk score from the 5-plex marker panel consisting of miRNAs-127-3p, 4736, 655-3p, TNM stage and histologic grading stratified patients into four risk categories. Compared to the low-risk group, the high-risk group had 23-fold increased mortality risk (hazard ratio 23, 95% confidence interval 13-42), with a median time-to-recurrence of 6 months and time-to-death of 11 months (vs >60 months for each among low-risk patient; p < .001). CONCLUSION: The miRNA-based 5-plex marker panel driven mortality risk score formula provides clinically practical and reliable measures to assess the prognosis of patients assigned to an early-stage OSCC.

Salivary gland anlage tumor: molecular profiling sheds light on a morphologic question.

OBJECTIVE: Salivary gland anlage tumor (SGAT), previously described as a squamous proliferative lesion or "congenital pleomorphic adenoma," is a rare, benign entity that presents within the first months of life. It occurs almost exclusively in the nasopharynx or posterior nasal cavity and demonstrates a biphasic composition of epithelial and mesenchymal elements. Although the clinical and histologic features of SGAT are well described, its etiology remains poorly understood. SGAT is currently considered a hamartoma rather than a neoplasm, partly because of its benign behavior and lack of reported recurrence after treatment. However, investigators have not yet evaluated this concept by using genomic methods. STUDY DESIGN: Here, we present 3 SGAT cases where we performed whole-exome sequencing. RESULTS: Examination of sequence data, with specific attention to variants affecting 964 cancer-related genes, showed no plausible driver-type alterations. CONCLUSIONS: The lack of apparent driver mutations supports the classification of this entity as a hamartomatous (nonneoplastic) process.

Characteristics and Prognosis of Primary Head and Neck Angiosarcomas: A Surveillance, Epidemiology, and End Results Program (SEER) Analysis of 1250 Cases.

Head and neck angiosarcomas (HN-AS) are rare malignancies with a poor prognosis relative to other soft tissue sarcomas. To date, the HN-AS literature has been limited to short reports and single-institution experiences. This study evaluated patients registered with the Surveillance, Epidemiology, and End Results (SEER) program who had been diagnosed with a primary HN-AS. Predictors were drawn from demographic and baseline tumor characteristics. Outcomes were survival months and cause of death. Kaplan-Meier analyses were used to estimate overall (OS) and disease-specific survival (DSS) rates. Cox proportional hazards regression models were used for multivariate analyses. A total of 1250 patients (mean age 73.3 years) were identified, and nearly all lesions (93.5%) were cutaneous. Two- and 5-year OS rates were 47.3% (95% CI 44.3-50.3) and 26.5% (95% CI 23.7-29.3), while 2- and 5-year DSS rates were 66.6% (95% CI 63.6-69.6) and 48.3% (95% CI 44.5-52.1). In the univariate analyses, age, race, tumor grade, tumor size, AJCC stage, SEER historic stage, and surgery were significant predictors of both OS and DSS. Multivariate regression revealed that independent predictors of poor OS and DSS were older age [OS: HR 1.04 (95% CI 1.02-1.05), p < 0.01; DSS: HR 1.03 (95% CI 1.01-1.05), p < 0.01], increased tumor size [OS: HR 1.01 (95% CI 1.01-1.01), p < 0.01; DSS: HR 1.01 (95% CI 1.01-1.02), p < 0.01], and distant disease [OS: HR 2.97 (95% CI 1.65-5.34), p < 0.01; DSS: HR 4.99 (95% CI 2.50-9.98), p < 0.01]. Age, tumor size, and disease extent were determinants of HN-AS survival. When all other factors were controlled, lower histologic grade and surgery did not improve the risk of death.

Lingual cyst with respiratory epithelium: report of 2 cases and review of the literature.

The lingual cyst with respiratory epithelium, a congenital cyst of the tongue or floor of the mouth, is lined predominately by respiratory-type epithelium. The terminology for this lesion was first proposed in 1999 by Manor et al., who stated that a descriptive term is best for this cyst of debatable pathogenesis. Although it is a cyst of foregut origin, the lingual cyst with respiratory epithelium is distinguished from the traditional enteric (foregut) duplication cyst in that the latter contains gastric and/or intestinal-type mucosa and has smooth muscle within the cyst wall. This article presents 2 new cases of this entity, as well as reviews the 19 cases that have been reported in the literature and were found to fulfill the histologic criteria of an lingual cyst with respiratory epithelium.