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.

Analysis of local invasion and regional spread in malignant sublingual gland tumour: Implications for surgical planning.

Malignant tumours arising from the sublingual glands are very rare, and the extent and frequency of local invasion or regional spread in malignant sublingual gland tumour (MSLT) has not been fully studied due to the disease rarity. To provide comprehensive features of local and regional spread of MSLT, we reviewed 20 surgical cases for detailed pathological analyses among 26 cases diagnosed as having primary MSLT. Adenoid cystic carcinoma (ACC) was the most common pathological subtype, followed by mucoepidermoid carcinoma. Disease-free and overall survivals at 5 years were 76.1 % and 77.7 %, respectively. High-grade malignant tumours and grade 2-3 ACC accounted for 41.7 % and 85.7 %. Clinical and pathological extraparenchymal extensions were found in 34.6 % and 80.0 %, respectively. Tumour invasion to the lingual nerve and submandibular gland/ductal system were also detected in 40.0 % and 28.6 %. The incidences of lingual nerve invasion in ACC and ACC ≥4 cm were 30.8 % and 42.9 %. Regional nodal involvement occurred in seven of 26 cases, and all metastatic lymph nodes were found in neck levels Ib and IIa. In summary, a significant portion of MSLT cases consisted of high-grade tumours and grade 2-3 ACC; therefore local invasion into adjacent structures should be cautiously evaluated in cases of MSLT.

Schwannoma arising from the sublingual glandular branch of the lingual nerve radiologically masquerading as sublingual gland tumor.

We report a rare case of schwannoma arising from the sublingual glandular branch of the lingual nerve radiologically masquerading as sublingual gland tumor. A 42-year-old female was referred to our department with a painless swelling in the left submandibular region. Contrast-enhanced computed tomography showed a well-circumscribed, heterogeneous low-density tumor with cystic change in the left sublingual region. Magnetic resonance imaging showed a well-circumscribed, heterogeneous sublingual tumor with low-signal intensity on T1-weighted image and high-signal intensity in T2-weighted image. The lesion was diagnosed radiologically as benign sublingual gland tumor. The patient underwent resection of sublingual gland tumor under general anesthesia. There was no definitive continuity between the tumor and the sublingual gland, and the tumor originated from sublingual glandular branch of the lingual nerve. Pathological examination of the specimen showed schwannoma with highly cellular areas (Antoni A) and hypocellular areas (Antoni B). The postoperative course was uneventful without lingual nerve palsy, and there was no recurrence 4 years after surgery.

Development of sentinel lymph node biopsy technique in patients with salivary gland cancer using the IDEAL framework.

BACKGROUND: Salivary cancer is rare and comprises a variety of histological subtypes and clinical behaviors. There is no agreed method of estimating the risk of occult metastasis or managing the clinically N0 neck.Sentinel node biopsy (SNB) may offer a solution but previous studies have not produced a reliable imaging protocol. This study uses novel technology and trial methodology to develop a reliable SNB technique, with primary aim to identify peri-and intraglandular sentinel nodes. METHODS: IDEAL framework was used to undertake SNB in clinically node negative salivary gland cancer. Patients with cT1-2 N0 salivary cancer were eligible. Lymphoscintigraphy was undertaken using Tc-99 m labelled nanocoll. Injection technique as well as adjunctive use of freehand SPECT (fhSPECT), near-infrared (NIR) fluorescence imaging, and navigation-guided surgery were used and optimisied during the study protocol. RESULTS: 10 patients were recruited. Initial protocol of peritumoural injection of Tc99 m nanocoll showed poor image resolution. Subsequent adjustment to single intratumoural injection allowed identification of intraglandular sentinel nodes. Fh/SPECT and NIR fluorescence imaging found intraglandular lymph nodes otherwise not recognizable to the naked eye. In two cases occult lymph node metastasis were identified. CONCLUSION: This study has shown the IDEAL framework is vital in allowing iterative changes in surgical protocol in the light of experience. This study has produced a reliable method for detection of sentinel nodes, in particular the ability to identify intra- and periglandular nodes with diagnosis of occult metastatic deposits and no false negative results. Our protocol can be readily transferred in to larger scale studies.

Malignant extrapleural solitary fibrous tumor arising in the sublingual gland: A case report and review of literature.

Solitary fibrous tumor is an uncommon neoplasm with unpredictable clinical behavior. Malignant solitary fibrous tumor is a rare morphological variant with more aggressive behavior and higher rates of local recurrences and distant metastasis, exceeding rare in oral cavity; our case occurred in the floor of the mouth in the sublingual gland.

Margins for Benign Salivary Gland Neoplasms of the Head and Neck.

The proper ablation of any neoplasm of the head and neck requires the inclusion of linear and anatomic barrier margins surrounding the neoplasm. Extirpative surgery of the major and minor salivary glands is certainly no exception to this surgical principle. To this end, the selection and execution of the most appropriate ablative surgical procedure for a major or minor benign salivary gland neoplasm is an essential exercise in oral and maxillofacial surgery. Of equal importance is the intraoperative identification and preservation of the pseudocapsule surrounding the benign neoplasm. This article reviews these important elements specifically related to ablative surgery of benign neoplasms of the parotid, submandibular and minor salivary glands with strict attention to observed nomenclature.

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.

Cervical lymph node metastasis in adenoid cystic carcinoma of the major salivary glands.

OBJECTIVE: To verify the prevalence of cervical lymph node metastasis in adenoid cystic carcinoma of major salivary glands, and to establish recommendations for elective neck treatment. METHODS: A search was conducted of the US National Library of Medicine database. Appropriate articles were selected from the abstracts, and the original publications were obtained to extract data. RESULTS: Among 483 cases of major salivary gland adenoid cystic carcinoma, a total of 90 (18.6 per cent) had cervical metastasis. The prevalence of positive nodes from adenoid cystic carcinoma was 14.5 per cent for parotid gland, 22.5 per cent for submandibular gland and 24.7 per cent for sublingual gland. Cervical lymph node metastasis occurred more frequently in patients with primary tumour stage T3-4 adenoid cystic carcinoma, and was usually located in levels II and III in the neck. CONCLUSION: Adenoid cystic carcinoma of the major salivary glands is associated with a significant prevalence of cervical node metastasis, and elective neck treatment is indicated for T3 and T4 primary tumours, as well as tumours with other histological risk factors.

Salivary gland malignancies - an update on current management for oral healthcare practitioners.

Salivary gland tumours represent a diverse range of tumours with many histological subtypes which occur in major and minor salivary glands. The management of these tumours is complex owing to their heterogeneity. Surgery together with radiotherapy and/or chemotherapy remains the treatment strategy for these tumours. The aim of this review is to examine the current management of these tumours.

Resected tumours of the sublingual gland: 15 years' experience.

Sublingual gland tumours are rare, and we have evaluated the clinical features and prognosis of patients treated at a tertiary medical centre in eastern Taiwan. We retrospectively reviewed the cases of nine patients with sublingual gland tumours that were resected from December 1993 to November 2008, four of whom were men and five women. The median (range) age at diagnosis was 52 (39-63) years. Seven had malignant tumours, of which adenoid cystic carcinoma was the most common. All patients with malignant tumours had neck dissections, and four had cervical lymph node metastases. The incidence of lymph node metastases was much higher in patients with advanced primary tumours (T1/2 compared with T3/4: one out of three compared with three out of four). All patients with malignant tumours were given adjuvant radiotherapy. There were no local failures. One patient had regional recurrence in the neck and had a successful further resection. Three patients developed distant metastases, and two died during the follow-up period. Our results suggest that radical resection with postoperative radiotherapy offers adequate local and regional control for malignant sublingual gland tumours. Neck dissection is beneficial, especially for T3/4 disease.

Surgery for Malignant Sublingual and Minor Salivary Gland Neoplasms.

Malignant sublingual gland neoplasms are rare, early-stage neoplasms presenting as painless non-ulcerated masses in the antero-lateral floor of the mouth. The majority of patients present with advanced disease, with symptoms of pain or anaesthesia of the tongue. Malignant minor salivary gland neoplasms are more common, the majority (>80%) of which present in the oral cavity, most frequently in the palatal area, as painless masses or as obstructive symptoms in the head and neck region. The most frequent pathologies are adenoid cystic carcinoma and mucoepidermoid carcinoma (>85%), with the majority presenting at an advanced stage (III/IV). Wide tumour-free surgical margin excision is the treatment of choice, followed by radiotherapy, after discussion of the multidisciplinary head and neck cancer tumour board. Improvements in survival and quality of life have been achieved since the introduction of endoscopic and robotic surgeries for many minor salivary gland malignancies.

Extracardiac rhabdomyomas--presentation of two cases with analysis of AKT/mTOR and ERK1/2 signaling.

Extracardiac rhabdomyomas (RM) are very rare benign tumors with a poorly understood pathogenesis. In this report we describe two RM cases--a sublingual adult type tumor and a genital type tumor involving the uterine cervix. The patho-clinical characteristics, as well as the pioneer immunohistochemical analysis of ERK1/2 and AKT/mTOR pathway status is included. The expression of key proteins involved in above signaling gives new insight into the biology of extracardiac RM.

Recurrent sublingual ranula or saliva leakage from the submandibular gland? Anatomical consideration of the ductal system of the sublingual gland.

PURPOSE: Some believe that the recurrence of sublingual ranula results from incomplete removal of the sublingual gland (SLG), but recurrence remains in some patients who undergo repeated excision of the remnant SLG, and the final solution to the recurrence is to remove the ipsilateral submandibular gland (SMG). In the authors' experience, preoperative aspirate from a sublingual ranula was a thick mucus-like fluid resembling egg white, whereas that from recurrent cyst after removal of the SLG was thin serous fluid. Based on the difference of the aspirated fluids, the authors speculated that the recurrent cystic mass might not be a ranula, but rather iatrogenic saliva leakage from the SMG through the previous surgically damaged excretory duct of the SLG (Bartholin duct) that opens into the SMG duct (Wharton duct). MATERIALS AND METHODS: A gross anatomic study was performed of the ductal system of the SLG and the anatomic communication between the Bartholin duct and Wharton duct. RESULTS: Four anatomic SLG duct variants were found. 1) The SLG has 1 Bartholin duct that seems to fuse with the Wharton duct but does not join the Wharton duct, running parallel to the Wharton duct and opening at its own orifice adjacent to and a short distance from the orifice of the Wharton duct (35.8%). 2) The SLG has 1 Bartholin duct that empties into the middle section of the Wharton duct (32.1%). 3) The SLG has 2 Bartholin ducts, one of which joins the Wharton duct and the other opens at its own orifice near that of the Wharton duct on the floor of the mouth (7.1%). 4) The SLG has many fine ducts (Rivinus ducts) that open at the floor of the mouth with no relation to the Wharton duct (25.0%). Of the 4 anatomic SLG duct variations, types 2 and 3 form immediate anatomic communication between the Bartholin duct and Wharton duct (39.2%). CONCLUSIONS: Several conclusions can be made from the present anatomic findings. 1) A certain proportion of Bartholin ducts open into the Wharton duct, and "recurrent ranula" after removal of the SLG can result from iatrogenic saliva leakage from the SMG through the surgically severed Bartholin duct if the aspirated fluid from the "recurrent cyst" is a thin seriflux. 2) The Bartholin duct emptying into the Wharton duct should be ligated during removal of the SLG to prevent local saliva accumulation from the SMG or even caudal "recurrence" as plunging ranula. 3) The surgical incision for SLG removal should be sutured loosely so that the surgically severed Bartholin duct with communication to the Wharton duct can reopen at the floor of mouth as an outlet for possible saliva leakage from the SMG and saliva accumulation can be avoided.

Distant Metastases in Patients with Carcinoma of the Major Salivary Glands.

BACKGROUND: This study aimed to show the incidence of distant metastases (DM) in salivary gland cancer as well as the types of histology most commonly associated with it and to identify factors predictive of DM. METHODS: The study identified 301 patients who underwent surgery for cancer of the major salivary glands at Memorial Sloan-Kettering Cancer center between 1985 and 2009. Clinical, tumor, and treatment characteristics were recorded. Tumors were categorized as low-, intermediate-, and high-risk pathology based on histologic subtype and grade. Factors predictive of distant recurrence-free probability (DRFP) were determined by uni- and multivariable analyses. RESULTS: The primary tumor was parotid in 266 patients (88 %), and 96 tumors (32 %) were clinical T3/T4. For 57 patients (18.9 %), DM developed with a 5-year DRFP of 72.7 %. The most common site of metastasis was the lung (50 %). The clinical predictors were male gender, cT4 stage, cN+ stage, and clinical overall stage. The multivariable analysis of clinical variables showed male gender (p = 0.018), cT4 stage (p < 0.001), and cN+ stage (p = 0.004) to be significant. The pathologic predictors were high-risk and high-grade pathology, vascular invasion, perineural invasion, positive margins, pT4 stage, pN+ stage, and overall stage. The multivariable analysis of pathologic variables showed high-grade pathology (p < 0.001), perineural invasion (p = 0.005), and pN+ stage (p = 0.002) to be significant. CONCLUSIONS: Distant metastases developed in approximately 20 % of the patients with salivary gland cancer. The most common site of metastases was the lung. The significant predictors of DM were cT4, cN+, male gender, high-grade pathology, perineural invasion, and positive nodal disease.

Lymphoepithelial carcinoma--review of the treatment modalities and report of a rare case in the sublingual gland.

Controversies exist in the current literature regarding the treatment of lymphoepithelial carcinoma (LEC). The presented study summaries the literature controversies in the treatment of LEC and present a case of a rare LEC diagnosed in the sublingual gland of an otherwise healthy 21-year-old Arab woman. The patient was admitted for swelling in the sublingual area. A biopsy indicated a diagnosis of LEC of the sublingual gland. Treatment included a wide resection with safe margins, marginal mandibulectomy, functional neck dissection, and a radial forearm free flap for reconstruction. The patient received concomitant radiotherapy and has remained disease free since her last follow-up. The recommended primary treatment, based on the literature review, is primary surgical excision with neck dissection and optional adjuvant radiotherapy to the gland area. Chemotherapy was not found to be beneficial according to the literature.

Carcinoma adenoquístico de glándula sublingual: a propósito de un caso / Adenocystic carcinoma of the sublingual gland: on the purpose of a case

Los tumores de glándulas salivales son relativamente infrecuentes, constituyendo el 1 por ciento de los tumores de cabeza y cuello, siendo aún más raros los tumores de glándulas sublinguales, aunque frecuentemente su diagnóstico es maligno. El propósito de este trabajo es la presentación de un caso clínico que presentó un carcinoma adenoquístico de glándula sublingual en suelo de boca lado izquierdo. Clínicamente apareció una lesión alargada, sobre el trayecto del conducto de Wharton, con una coloración normal, lisa, brillante y asintomática. Se le realizó exéresis simple de la lesión y el Departamento de Anatomía Patológica confirmó el diagnóstico. Se procedió a revisar la bibliografía nacional e internacional sobre estas lesiones encontrándose escasas referencias debido a su rareza. La evolución de la paciente después de 16 meses ha sido satisfactoria(AU)

The salivary gland tumors are relatively infrequent, being only 1 per cent of the head and neck tumors; the sublingual gland tumors are even more unusual, although their diagnosis is frequently malignant. The purpose of the current work is presenting a clinical case carrying a sublingual gland adenocystic carcinoma on the left side of the mouth bottom. Clinically, there it was a long lesion, over the course of the Whartons duct, with an asymptomatic, normal, brilliant and smooth color. The lesion was simply removed and the Department of Pathologic Anatomy confirmed the diagnosis. We reviewed the national and international literature on these lesions, finding scarce references because of its unusualness. The patients evolution is satisfactory when 16 months have passed from the surgery(AU)

Hemangioma cavernoso sublingual / Sublingual cavernous haemanginoma

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Computer guided surgery for implant placement and dental rehabilitation in a patient undergoing sub-total mandibulectomy and microvascular free flap reconstruction.

A 58-year-old patient presented with an extensive, destructive, recurrent pleomorphic adenoma occupying the mandibular body and the soft tissues of the mouth and neck. Resection of the mandible from right ramus to left condylar process, and implant rehabilitation in both jaws with fixed bridgework was planned. Comprehensive presurgical prosthetic work up was carried out to record the existing dental relationship and guide all stages of the reconstruction. The jaw was first grafted with a segmented, fibular microvascular free-flap, which was fixed in place with a fixation plate prebent on a Rapid Prototype Anatomical Model of the jaw. Reconstruction with implant supported fixed partial dentures took place to the dental scheme planned preresection, using a computer guided approach to implant placement in the complex and unfamiliar anatomy of the extensively grafted mandible. This approach facilitated and expedited implant surgery such that treatment could take place using a minimally invasive approach relatively soon after surgery, prior to commencement of radiotherapy, and highlights the importance of a multidisciplinary approach to treatment for patients having extensive surgery to the jaws. The patient's personal assessment 2 years post surgery was recorded using 1999 University of Washington Quality of Life Questionnaire.