Adenoid cystic carcinoma of the sublingual gland developing lung metastasis 20 years after primary treatment: A case report and literature review.

RATIONALE: Adenoid cystic carcinoma (ACC) is a rare malignant tumor that primarily occurs in the salivary glands. Distant metastases can develop despite favorable local control. Moreover, distant metastasis of ACC can occur after a long time interval without local recurrence. We report the first case of ACC of the sublingual gland that developed lung metastasis 20 years after primary treatment. PATIENT CONCERNS: A 52-year-old man was referred to our department with a 1-year history of painful swelling on the right oral floor. DIAGNOSIS: An incisional biopsy was performed, and histopathological examination revealed malignancy. INTERVENTIONS: Surgical excision of the right oral floor and right supra-omohyoid neck dissection with postoperative chemoradiation therapy were performed, and ACC of the sublingual gland was diagnosed. Left pulmonary metastasis was detected 20 years after the primary treatment. Metastasectomy was performed; however, subsequently, skin and bone metastases developed. OUTCOMES: After receiving palliative care, the patient died of multiple organ failure. LESSONS: As late distant metastasis of salivary ACC can develop, patients who undergo primary treatment need a long-term, strict follow-up plan even if locoregional control is favorable.

The number and ratio of positive lymph nodes are independent prognostic factors for patients with major salivary gland cancer: Results from the surveillance, epidemiology, and End Results dataset.

INTRODUCTION: To investigate whether the positive lymph node number (PLNN) and positive lymph node ratio (PLNR) could predict the prognosis of patients with major salivary gland cancer (MSGC) and to identify the optimal cutoff points for these variables that stratify patients according to their risk of survival. METHODS: We used the Surveillance, Epidemiology, and End Results (SEER) database to identify all patients with MSGC between 1988 and 2014. A logistic regression analysis was carried out to evaluate the risk factors for lymph node metastasis (LNM) in MSGC. The X-tile program was used to identify the cutoff values for the PLNN and PLNR in MSGC patients with LNM. Cox proportional hazards regression models were performed to identify the predictors of cancer-specific survival (CSS). RESULTS: In the SEER database, 8668 eligible patients were identified and 3046 of them had LNM. The logistic regression analysis indicated that older age, male sex, larger tumor size, higher grade, tumor extension and high-risk pathology were associated with LNM. The X-tile program showed that a PLNN>4 and a PLNR>0.15 were prognostic indicators of CSS. A multivariable analysis indicated that, after the factors that might potentially affect the prognosis were adjusted for, the PLNN and PLNR were still associated with CSS. CONCLUSIONS: Our Results demonstrated that the PLNN and PLNR were independent prognostic indicators for MSGC patients with lymph node metastasis.

Prognostic Factors in Malignant Sublingual Salivary Gland Tumors.

PURPOSE: The present study identified the prognostic factors and outcomes for malignant sublingual salivary gland tumors, which are rare. MATERIALS AND METHODS: A retrospective cohort study of patients treated for malignant sublingual salivary gland tumors from 1997 to 2011 was performed. The predictor variables, including age, gender, tumor stage, nodal stage, perineural invasion, margin status, and lymphovascular invasion, were analyzed. The Cox regression model was used to determine the prognostic factors for locoregional recurrence, distant metastasis, and survival. RESULTS: Of the 38 patients, 16 (42.1%) were men. Their mean age was 53 years (range 36 to 75). A total of 11 patients (28.9%) had T3-T4 tumors, and 6 (15.8%) had positive neck lymph nodes. The recurrence rate at 5 years was 18.4%. The distant metastasis rate at 5 years was 23.7%. Multivariable analysis confirmed the independent prognostic importance of patient age, N stage, and limited tongue mobility in locoregional recurrence and mortality at 5 years. CONCLUSIONS: Our results suggest that patient age, N stage, and limited tongue mobility are useful as independent predictors of locoregional recurrence and mortality in patients with malignant sublingual salivary gland tumors.

Predictors of outcomes in large cell undifferentiated carcinoma of the major salivary glands.

OBJECTIVE: Major salivary gland large-cell undifferentiated carcinoma (LCUC) is rare and has a poor prognosis. Characterization of patient demographics, tumor characteristics, and predictors of outcome have been limited by low case numbers, as well as grouped analysis with other salivary malignancies. The objective of this study was to address these issues using large-scale national data. STUDY DESIGN: Retrospective case series. METHODS: Data from the National Cancer Database, including cases diagnosed from 1998 to 2012, was analyzed, identifying 247 records of LCUC. Tumor, demographic, and survival information was extracted and analyzed retrospectively. RESULTS: Large-cell undifferentiated carcinoma comprised < 1% of all major salivary gland cancers. Seventy percent of patients presented with advanced-stage disease. The incidence of occult nodal disease was 39%. Surgery followed by radiation was the most common treatment. Five-year overall survival was 36%. Comorbidity, distant metastasis, and positive surgical margins were found to be predictors of overall survival. CONCLUSION: To our knowledge, this represents the largest reported case series of LCUC. The survival analysis demonstrates poorer survival in patients with positive surgical margins; therefore, efforts to complete resection are reasonable. Reported high rates of occult nodal disease also strongly support elective treatment of the neck. LEVEL OF EVIDENCE: 4. Laryngoscope, 2016 127:372-376, 2017.

Salivary gland cancer in patients younger than 30 years.

Previous research has shown that salivary gland tumors are rare in the young population. A clinical diagnosis has to be made very carefully because the proportion of malignancies is higher in children than in adults. We present a review of cases of malignant salivary gland carcinoma (SGC) in patients younger than 30 years of age. Data were extracted from the Surveillance, Epidemiology, and End Results (SEER) 17 Registry. A total of 763 patients younger than 30 years with carcinoma of a major salivary gland from 1973 to 2004 were identified within the SEER database. The most common salivary gland cancer was mucoepidermoid parotid gland carcinoma. The incidence of all major salivary gland carcinomas increased with increasing patient age. The 5-year relative survival rate for salivary gland carcinomas in our population was calculated according to the Kaplan-Meier analysis in each age group. Relative 5-year survival was 100% in the 1 patient younger than 1 year, 50.0% in the 1- to 4-year-old group; 87.2% among the 5- to 9-year-olds; 97.0% among the 10- to 14-year-olds; 95.0% among the 15- to 19-year-olds; 95.1% among the 20- to 24-year-olds; and 93.6% in the 25- to 29-year-old group. We found that SGC affects patients of all ages, even children in the first year of life. It is essential for physicians to detect salivary gland neoplasms promptly and to evaluate them thoroughly when they are found in children.

Sublingual gland tumors: clinical, pathologic, and therapeutic analysis of 13 patients treated in a single institution.

BACKGROUND: Sublingual gland tumors are rare, although frequently malignant. This study describes the clinicopathologic features and treatment results and reviews the literature. METHODS: Thirteen cases treated between 1996 and 2007 were reviewed with interest on clinical, pathologic, and therapeutic information. Survival data were calculated by the Kaplan-Meier method. RESULTS: Malignancies represented 92.3% of cases. Adenoid cystic carcinoma was the most common malignant type (66.7%). Most patients (83.3%) presented in advanced pathologic TNM stages (III or IV). All cases underwent surgical treatment. Neck dissection was performed in 69.2% with no metastases detected. Ten patients (83.3%) had adjuvant radiotherapy. Distant metastases occurred in 3 patients (25%). The 5-year overall and disease-free survival rates were 78.7% and 87.5%, respectively. CONCLUSIONS: Tumors of the sublingual gland are rare and are usually malignant. Radical surgery and adjuvant radiotherapy seems to offer adequate local and regional control. Unlike distant failure, local recurrence and regional metastases are not common.

A comprehensive analysis of parotid and salivary gland cancer: worse outcomes for male gender.

BACKGROUND: To determine the effects of patient demographics, socioeconomic status (SES) and clinical variables on outcomes for patients with salivary and parotid gland tumors. METHODS: Florida cancer registry and inpatient hospital data were queried for cancer of the salivary glands diagnosed between 1998-2002. RESULTS: A total of 1573 patients were identified. Women were diagnosed at a younger age (median age (years): women 60.8 versus men 64.3, P=0.003). Men were more often diagnosed with high grade tumors (65.1% versus 41.9% for women, P<0.001) and advanced disease stage (>stage III: 60.2 versus 49.4%, P<0.001), but underwent surgical extirpation and received radiation at equal rates compared with women. Overall 5-year survival rates was superior in women (67.4% versus 55.6%, P=0.001). By multivariate analysis, adjusted for patient comorbidities, age over 65 (HR 3.43 P=0.008), advanced disease stage (HR 8.05 P<0.001), and high tumor grade (HR 2.33, P<0.001) were independent predictors of worse prognosis. Improved outcomes were observed for female gender (HR 0.68, P=0.011). Tumors located in the parotid gland (HR 0.631 P=0.003) and receiving both surgical extirpation and radiation were predictors of improved survival. CONCLUSION: Salivary gland tumors carry a worse prognosis than tumors of the parotid. Male patients have worse outcomes.

Malignant sublingual gland tumors: a retrospective clinicopathologic study of 28 cases.

OBJECTIVES: To evaluate the clinicopathologic features and therapeutic efficacy of malignant sublingual gland tumors. PATIENTS AND METHODS: From 1955 to 2005, the clinicopathologic data of 28 patients treated in our hospital were obtained from their medical records and histopathologic slides. RESULTS: There were 16 males and 12 females, with a mean age of 50.3 years. Eighteen cases (64.3%) were adenoid cystic carcinoma; 16 (57.1%) cases were clinically staged as III-IV. Adenoid cystic carcinoma was mainly of the histologic type, and the other histologic classifications included mucoepidermoid carcinoma, myoepithelial carcinoma, polymorphous low-grade adenocarcinoma, adenocarcinoma and malignant pleomorphic adenoma. Pulmonary metastasis and tumor recurrence were the main death reasons. Eleven patients remain alive and well 34-312 months (median 108) after treatment. CONCLUSIONS: Malignant sublingual gland tumors are extremely rare and most are adenoid cystic carcinoma. Surgery is the main treatment option. For adenoid cystic carcinoma, hematogenous spread is common, and pulmonary metastasis is a common pathway of the distant metastasis. For some patients having lung metastasis, regional control is also important as there are some examples of patients surviving many years with asymptomatic pulmonary metastases. Postoperative radiation therapy may be adjuvant for selected patients with high-stage and high-grade tumors, or when there is concern about the inadequacy of the resection. The effect of chemotherapy remains elusive.

Sublingual gland resection in squamous cell carcinoma of the floor of mouth: is it necessary?

OBJECTIVES: Little evidence exists to guide surgeons in the management of the sublingual glands (SLG) not macroscopically involved by squamous cell carcinoma of the floor of mouth and oral tongue. This study aims to determine the frequency with which the SLG is invaded, to identify variables predicting for SLG invasion and the morbidity associated with it's resection in entirety. STUDY DESIGN: Retrospective cohort study. METHODS: A review of 164 patients treated for oral cavity cancer at a tertiary institution with a large volume of head and neck malignancy was performed. Demographic data, rates of surgical complications and follow up information was recorded. Pathologic review of resected material in this group yielded 134 specimens in the region of the SLG. A detailed analysis of 63 specimens in which the SLG was included was carried out. RESULTS: The median age was 58 years, mean follow up was 2.2 years, and there were 44 males and 19 females. Seventeen cases (27%) demonstrated histopathological SLG invasion. In patients with SLG involvement, this was evident at the time of surgery in 15 patients (88%). Microscopic SLG invasion, without macroscopic evidence at surgery, was present in only 4.2% of patients undergoing SLG resection. Clinical and pathological T stage (p = 0.023 and 0.005) and tumor thickness (p = 0.015) predicted for SLG invasion. Total SLG resection significantly increased the post-operative wound complication rate from 14% in patients without SLG resection to 25% (p = 0.05). CONCLUSION: Total SLG resection in early stage and thin squamous cell carcinoma of the floor of mouth and oral tongue provides minimal oncologic benefit and is associated with increased perioperative morbidity due to neck wound complications.

Mucoepidermoid carcinoma of the major salivary glands: clinical and histopathologic analysis of 234 cases with evaluation of grading criteria.

BACKGROUND: The authors had previously conducted an investigation of minor salivary gland mucoepidermoid carcinoma, in which they demonstrated that certain clinical and histopathologic features were useful in predicting biologic outcome. The current study investigated the usefulness of these features in determining the prognoses of patients with mucoepidermoid carcinomas of the major salivary glands. METHODS: Clinical data and 15 histopathologic features were compared in 4 patient groups based on outcome after initial treatment. The outcome groups were 1) survival without disease, 2) survival with tumor recurrence only, 3) survival with metastasis, and 4) death related to tumor. A numeric score was assigned to each unfavorable histopathologic feature. Low grade tumors had scores of 0-4. Intermediate grade tumors scored 5 or 6. High grade tumors had scores higher than 6. RESULTS: Most patients (75%) were tumor free after the initial treatment. Twenty-one patients (9%) had local recurrence only, 12 (5%) demonstrated metastasis and survived, and 25 patients (11%) died of their disease. CONCLUSIONS: Clinical features associated with metastasis or death were more advanced age, tumor size, and preoperative symptoms. Histopathologic features that correlated with poor outcome were cystic component less than 20%, 4 or more mitotic figures per 10 high-power fields, neural involvement, necrosis, and anaplasia. All five of these histopathologic features demonstrated statistical prognostic significance when parotid gland tumors from Groups 1 and 4 were compared (P < 0.001). The point-based grading system demonstrated a statistically significant correlation with outcome for parotid tumors but not for submandibular tumors. The authors' findings indicate that patients with tumors of equal histopathologic grade have a better prognosis when their tumors are in the parotid gland than when their tumors are in the submandibular gland. Six of eight submandibular tumors that metastasized or resulted in death were low grade lesions, and none were high grade.