The Rare Radiographic Sunburst Appearance of Odontogenic Myxomas: A Case Report and Review of the Literature.

Odontogenic myxomas often have a distinctive radiographic presentation described as a "soap bubble", "tennis racket", or "honeycomb" pattern. Less frequently, examples of odontogenic myxomas with a "sunray" or "sunburst" pattern have been reported. Because malignant entities such as osteosarcomas more classically present with a sunray/sunburst appearance, odontogenic myxomas are rarely considered in the radiographic differential diagnosis of a sunburst lesion. The objective of this paper is to report a case of an odontogenic myxoma presenting with a sunburst appearance and to review similar reported cases in the literature. To the best of the authors' knowledge, this additional case of an odontogenic myxoma presenting with a sunburst appearance brings the total number of sunray/sunburst cases reported in the English language literature to 21.

Defining the surgical margins of adenoid cystic carcinoma and their impact on outcome: An international collaborative study.

BACKGROUND: The mainstay of treatment in adenoid cystic carcinoma (ACC) of the head and neck is surgical resection with negative margins. The purpose of this study was to define the margin status that associates with survival outcomes of ACC of the head and neck. METHODS: We conducted univariate and multivariate analyses of international data. RESULTS: Data of 507 patients with ACC of the head and neck were analyzed; negative margins defined as ≥5 mm were detected in 253 patients (50%). On multivariate analysis, the hazard ratios (HRs) of positive margin status were 2.68 (95% confidence interval [CI], 1.2-6.2; p = .04) and 2.63 (95% CI, 1.1-6.3; p = .03) for overall survival (OS) and disease-specific survival (DSS), respectively. Close margins had no significant impact on outcome, with HRs of 1.1 (95% CI, 0.4-3.0; p = .12) and 1.07 (95% CI, 0.3-3.4; p = .23) for OS and DSS, respectively, relative with negative margins. CONCLUSION: In head and neck ACC, positive margins are associated with the worst outcome. Negative or close margins are associated with improved outcome, regardless of the distance from the tumor. © 2017 Wiley Periodicals, Inc. Head Neck 39: 1008-1014, 2017.

Ghost Cell Tumors.

Ghost cell tumors are a family of lesions that range in presentation from cyst to solid neoplasm and in behavior from benign to locally aggressive or metastatic. All are characterized by the presence of ameloblastic epithelium, ghost cells, and calcifications. This report presents the cases of a 14-year-old girl with a calcifying cystic odontogenic tumor (CCOT) and a 65-year-old woman with a peripheral dentinogenic ghost cell tumor (DGCT) with dysplastic changes, a rare locally invasive tumor of odontogenic epithelium. The first patient presented with a 1-year history of slowly progressing pain and swelling at the left body of the mandible. Initial panoramic radiograph displayed a mixed radiolucent and radiopaque lesion. An incisional biopsy yielded a diagnosis of CCOT. Decompression of the mass was completed; after 3 months, it was enucleated and immediately grafted with bone harvested from the anterior iliac crest. The second patient presented with a 3-month history of slowly progressing pain and swelling at the left body of the mandible. Initial panoramic radiograph depicted a mixed radiolucent and radiopaque lesion with saucerization of the buccal mandibular cortex. An incisional biopsy examination suggested a diagnosis of DGCT because of the presence of ghost cells, dentinoid, and islands of ameloblastic epithelium. Excision of the mass with peripheral ostectomy was completed. At 6 and 12 months of follow-up, no evidence of recurrence was noted.

Incidence of cervical lymph node metastasis and its association with outcomes in patients with adenoid cystic carcinoma. An international collaborative study.

BACKGROUND: The patterns of regional metastasis in adenoid cystic carcinoma (ACC) of the head and neck and its association with outcome is not established. METHODS: We conducted a retrospective multicentered multivariate analysis of 270 patients who underwent neck dissection. RESULTS: The incidence rate of neck metastases was 29%. The rate observed in the oral cavity is 37%, and in the major salivary glands is 19% (p = .001). The rate of occult nodal metastases was 17%. Overall 5-year survival rates were 44% in patients undergoing therapeutic neck dissections, and 65% and 73% among those undergoing elective neck dissections, with and without nodal metastases, respectively (p = .017). Multivariate analysis revealed that the primary site, nodal classification, and margin status were independent predictors of survival. CONCLUSION: Our findings support the consideration of elective neck treatment in patients with ACC of the oral cavity.

Elective neck dissection in patients with head and neck adenoid cystic carcinoma: an international collaborative study.

BACKGROUND: Adenoid cystic carcinoma (ACC) accounts for 3-5 % of all head and neck malignancies. Investigations of outcomes from elective neck dissection (END) for patients with ACC are sparse. This study aimed to assess the impact of END on the survival of patients with ACC. METHODS: This retrospective multicentered study investigated 270 patients who underwent neck dissection. A multivariate analysis assessed associations of clinical and histopathologic characteristics with survival outcomes. RESULTS: The primary tumor sites included the oral cavity in 250 patients (55 %), the major salivary glands in 133 patients (29 %), the sinonasal mucosa in 68 patients (15 %), and the larynx in six patients (1 %). The overall rate of occult nodal metastases among the patients who underwent END was 17 % (38/226). The highest incidence of occult nodal metastases was with the oral cavity (66 %). The 5-year overall survival (72 and 79 % for patients with or without END, respectively) and disease-specific survival (74 and 81 % for patients with or without END, respectively) were similar in the two groups. The subgroup analysis of patients according to the primary site showed no significant impact of END on outcome. In the multivariate analysis, primary site, T classification, and N classification were the only variables associated with outcome. CONCLUSIONS: The incidence of occult neck metastases among patients with ACC is 17 %. The highest incidence of occult metastases is with the oral cavity. Statistical analysis showed no survival advantage for patients who underwent END compared with those who did not.

International collaborative validation of intraneural invasion as a prognostic marker in adenoid cystic carcinoma of the head and neck.

BACKGROUND: The purpose of this study was to characterize the incidence, pattern of spread, and prognostic correlation of nerve invasion in patients with adenoid cystic carcinoma (ACC). METHODS: Using 3 different pathological categories of perineural invasion, intraneural invasion, and perineural inflammation, we investigated the prognostic value of nerve invasion in a total of 495 ACCs from 9 international patient cohorts with median follow-up 90 months (range, 12-288 months). RESULTS: Of 239 patients (48%) with nerve invasion, 174 (73%) had perineural invasion, 65 (27%) intraneural invasion, and 37 (15%) perineural inflammation. Multivariate Cox regression analysis identified tumor site (p = .008; hazard ratio [HR] = 1.8; 95% confidence interval [CI] = 0.07-3.7) and intraneural invasion (p < .001; HR = 5.9; 95% CI = 0.8-12.3) as independent prognostic markers for both overall survival (OS) and disease-specific survival (DSS), but not of distant metastases. CONCLUSION: Although perineural invasion has no impact on survival, intraneural invasion is an independent predictor of poor prognosis. Recognition of intraneural invasion may help optimize treatment of patients with head and neck ACC.

Histologic findings within peri-implant soft tissue in failed implants secondary to excess cement: report of two cases and review of literature.

A link has been established between peri-implant disease and excess cement extrusion in cement-retained implant restorations. The histologic findings of two patients with failed implants secondary to residual excess cement are reported here. If excess cement is detected early and adequately removed, resolution can occur in the majority of situations. Simple recommendations are proposed, with the intention of preventing further implant failures from residual excess cement.

Analysis of failure in patients with adenoid cystic carcinoma of the head and neck. An international collaborative study.

BACKGROUND: Adenoid cystic carcinoma (ACC) is a locally aggressive tumor with a high prevalence of distant metastases. The purpose of this study was to identify independent predictors of outcome and to characterize the patterns of failure. METHODS: An international retrospective review was conducted of 489 patients with ACC treated between 1985 and 2011 in 9 cancer centers worldwide. RESULTS: Five-year overall-survival (OS), disease-specific survival (DSS), and disease-free survival (DFS) were 76%, 80%, and 68%, respectively. Independent predictors of OS and DSS were: age, site, N classification, and presence of distant metastases. N classification, age, and bone invasion were associated with DFS on multivariate analysis. Age, tumor site, orbital invasion, and N classification were independent predictors of distant metastases. CONCLUSION: The clinical course of ACC is slow but persistent. Paranasal sinus origin is associated with the lowest distant metastases rate but with the poorest outcome. These prognostic estimates should be considered when tailoring treatment for patients with ACC.

Adenoid cystic carcinoma of the nasal cavity and paranasal sinuses: a meta-analysis.

Objectives To identify independent predictors of outcome in patients with adenoid cystic carcinoma (ACC) of the paranasal sinuses and skull base. Design Meta-analysis of the literature and data from the International ACC Study Group. Setting University-affiliated medical center. Participants The study group consisted of 520 patients, 99 of them from the international cohort. The median follow-up period was 60 months (range, 32 to 100 months). Main Outcome Measures Overall survival (OS) and disease-specific survival (DSS). Results The 5-year OS and DSS of the entire cohort were 62% and 67%, respectively. The local recurrence rate was 36.6%, and the regional recurrence rate was 7%. Distant metastasis, most commonly present in the lung, was recorded in 106 patients (29.1%). In the international cohort, positive margins and ACC of the sphenoid or ethmoidal sinuses were significant predictors of outcome (p < 0.001). Perineural invasion and adjuvant treatment (radiotherapy or chemoradiation) were not associated with prognosis. Conclusion Tumor margin status and tumor site are associated with prognosis in ACC of the paranasal sinuses, whereas perineural invasion is not. Adjuvant treatment apparently has no impact on outcome.

Prognostic value of quantitative p63 immunostaining in adenoid cystic carcinoma of salivary gland assessed by computerized image analysis.

BACKGROUND: In a long-term retrospective immunohistochemical study of adenoid cystic carcinoma (ACC) of salivary gland, we investigated the relation of p63 immunodetection to prognosis. Although it is generally agreed that the solid pattern is the most aggressive pattern of growth, ACCs with predominantly cribriform or tubular patterns have an unpredictable clinical course, with a relatively favorable 5-year survival but a low 20-year survival. METHODS: Formalin-fixed paraffin sections from 35 cases of ACC showing a predominantly better differentiated histopathology, ie, cribriform or tubular patterns of growth, were immunostained for p63. Automated image analysis was used to quantify p63 positivity, using a modification of a previously developed algorithm. RESULTS: Patients alive for more than 10 years had a lower extent of p63 expression than those who died of disease. Kaplan-Meier analysis revealed that separation of patients with morbidity and mortality from those alive with no evidence of disease, could be achieved at a cutoff of 35% p63 positivity (P = .0031, log-rank test). Multivariate analysis using the Cox proportional hazard model revealed p63 and tumor stage to be independent predictors of survival (P = .012 and P = .0003, respectively). CONCLUSIONS: To our knowledge, the present study is the first to report prognostic significance of p63 in salivary gland ACC and the first report of a robust and well-studied immunohistochemical stain performable on routinely fixed and processed tissue with prognostic utility.

Sialolipoma: report of two cases and review of the literature.

Sialolipoma is a new variant of salivary gland lipoma first described in 2001. We present 2 cases of sialolipoma involving the soft palate and buccal mucosa of 2 female patients. A review of the English language literature revealed 16 cases of sialolipoma reported thus far. We have reviewed the clinicopathologic features of the literature cases and are adding the present 2 cases for a total of 18 cases. To the best of our knowledge, this is the first report of sialolipoma involving the buccal mucosa and the second case in the soft palate. The purpose of this publication is to increase awareness of this recently described variant of lipoma and to address the diagnostic challenges posed both clinically and histopathologically.