Submandibular Gland Tumors Research Articles

Desmoplastic small round cell tumor of the submandibular gland—a rare but distinctive primary salivary gland neoplasm

Desmoplastic small round cell tumor is a highly aggressive neoplasm that generally involves the peritoneum and pelvis of young patients. Only rare cases occur outside the abdomen. We report a case presenting as a primary submandibular gland tumor in a 24-year-old man. Histologically, although there were irregular tumor islands lying in an abundant desmoplastic stroma, there were also areas comprising large cellular islands with scanty stroma in between, raising the differential diagnosis of various salivary gland carcinomas. The tumor cells were medium sized, with hyperchromatic nuclei and moderate amounts of cytoplasm. The diagnosis of desmoplastic small round cell tumor was confirmed by the presence of a polyphenotypic immunoprofile (positive for cytokeratin, desmin, and neuron-specific enolase) and the characteristic EWS-WT1 gene fusion. Although rare, desmoplastic small round cell tumor has to be considered in the differential diagnosis of poorly differentiated neoplasms of the salivary gland, especially in young patients.

An evidence based approach to the management of salivary masses

Salivary lumps are common and constitute 13.8% of all lumps assessed at the North Glasgow neck lump clinic. We present our management plan for these conditions. Literature search using key words salivary lumps or glands or cancer or sialadenitis or sialalisthiasis in combination with diagnosis, management, investigation, treatment, intervention and surgery was performed in June 2009 using MEDLINE and Pubmed. Data from the North Glasgow neck lump clinic database are also presented. Published literature on management of salivary lumps is level 2/3 evidence. The majority of salivary gland swellings can be adequately assessed with ultrasound and ultrasound guided FNA. The majority of patients with salivary gland tumours are likely to benefit from surgical excision. Due to the higher risk of malignancy in submandibular gland tumours surgical excision should be undertaken, unless there is significant co-morbidity.

Salivary Duct Carcinoma of the Submandibular Gland Mimicking Deep Neck Infection-A Case Report

Background: Salivary duct carcinoma (SDC) is a rare highly malignant tumor, characterized by early distant metastasis and dismal prognosis. It often derives from the excretory duct reserve cell of the major salivary glands and is histologically similar to ductal carcinoma of the breast. The parotid gland is affected in most of the cases. Aim and Objectives: We report a 47-year-old man with diabetes mellitus and SDC of the submandibular gland mimicking deep neck infection. Materials and Methods: The patient underwent surgical drainage for his rapidly swollen right neck accompanied by erythema and tenderness. Results: A solid tumor of the right submandibular gland was found instead. Pathological examination confirmed the diagnosis of salivary duct carcinoma. Distant metastasis had evolved at the time of diagnosis and the patient died within two months. Conclusion: The presentation of SDC can mimic a deep neck infection. In this article, the clinical characteristics of SDC and the treatment strategies are discussed.

Pagetoid spread to oral mucosa from submandibular gland salivary duct carcinoma: a case report

Salivary duct carcinoma is a high-grade malignant tumor arising predominantly in the major salivary glands. We describe herein a rare presentation of salivary duct carcinoma arising in the submandibular gland and showing pagetoid spread to the oral mucosa. A 46-year-old man was admitted with a 2-year history of swelling in the floor of the mouth. Resection of the submandibular gland tumor was followed by mucosal biopsy around the aperture of the right Wharton's duct. The tumor recurred in the oral mucosa with pagetoid spread and extended widely in the oral cavity for the following 10 years, despite chemo- and radiotherapy. Mucosal spread, although a relatively remote possibility, can occur in the setting of salivary duct carcinoma.

顎下腺良性腫瘍から発生したと考えられた唾液腺導管癌の1例

We report the case of a salivary duct carcinoma thought to have arisen from a benign tumor of the submandibular gland. The patient was a 72-year-old man. He had a hard, elastic, painless mass measuring 30 ×20mm in the right submandibular region. CT and MRI revealed that the mass spread to the adjacent tissues with a rough margin and ipsilateral cervical lymph node metastases. He underwent excision of the right submandibular gland tumor combined with a radical neck dissection. On histological examination of the surgical specimens, malignant components were observed around hyalinized tissues, which were thought to be remnants of a benign tumor. On the basis of these histological findings, the tumor was diagnosed to be a salivary duct carcinoma, which was highly suspected to have undergone transformation from a benign tumor. The neck specimens had 17 histologically positive nodes, including 12 nodes showing extracapsular spread of tumor. He received postoperative chemoradiotherapy, but local failure with distant metastases developed, and the patient died 10 months after operation.

Gland-preserving surgery for pleomorphic adenoma in the submandibular gland

Removal of tumours arising in the submandibular gland (SMG) usually involves excision of the entire gland. This prospective study evaluated the efficacy of gland-preserving surgery in patients with benign SMG tumours. Twenty consecutive patients with pleomorphic adenomas in the SMG underwent local excision of tumours with limited tumour-free margins and preservation of the remnant glandular tissues. All patients had salivary scintigraphy before and 6 months after surgery, with ultrasonography 2 years after operation. Complications, operating time, salivary function and rates of tumour recurrence were evaluated. The mean operating time was 28 min, and no patient had neurological complications. After surgery, the function of the affected gland was equal to that of the unaffected gland. All patients had symmetrical facial contours without defects in the operated submandibular triangle. None of the patients had evidence of tumour recurrence on ultrasonographic examination and regular clinical examination at a median of 36 months after surgery. Gland-preserving surgery in patients with benign SMG tumours preserves salivation, and reduces surgical morbidity and operating time, with good cosmesis but without compromising local control.

Primary epithelial submandibular salivary gland tumours – Review of management in a district general hospital setting

Primary epithelial submandibular gland (SMG) tumours are uncommon, accounting for 8-12% of all salivary gland neoplasms, and most studies come from large specialised centres. There is little published about the relative frequency and outcome of SMG tumours treated in general hospitals. A retrospective study from two district general hospitals over a 10 year period (1996-2005). Seventeen benign (68%) and eight malignant (32%) tumours were included. A painless mass was the most common presentation. Pain and paraesthesia were not pathognomonic of malignancy. Pleomorphic adenoma accounted for all 17 benign tumours and adenoid cystic carcinoma was the commonest malignant tumour. Fine needle aspiration cytology (FNAC) accurately identified 78% of the benign tumours but none of the malignant tumours. Pre-operative imaging was also unable to distinguish malignant from benign tumours. Incomplete excision was reported in 20% of cases and was more common for malignant tumours. It may be difficult to distinguish benign from malignant SMG tumours on clinical examination and pre-operative investigations. Any suspected submandibular tumour should be considered for early treatment even when FNAC is suggestive of a benign tumour.

MRI of chronic sclerosing sialoadenitis

The purpose of our study was to describe the MR appearance of Kuttner's tumours and to interpret their differences in appearance from other submandibular gland tumours. MR studies of 7 Kuttner's, 8 malignant and 12 benign submandibular gland tumours were reviewed. MR sequences obtained included T(1) weighted, short inversion time inversion recovery (STIR), T(2) weighted, diffusion-weighted (DW) and dynamic contrast-enhanced MR (dynamic MR) images. In all cases of Kuttner's tumour, the affected submandibular glands were swollen with slightly higher intensity on T(2) weighted, STIR and DW images, but the tumour margin could not be defined. Conversely, the margins of the other tumours could be detected. On T(2) weighted, STIR and DW images, the mean signal intensity ratios and the mean apparent diffusion coefficient (ADC) values for Kuttner's tumours and malignant tumours were significantly lower than those of benign tumours, but there were no significant differences between those of Kuttner's tumours and those of malignant tumours. All benign tumours showed late enhancement, with peak enhancement later than 120 s on dynamic MR images. Kuttner's tumours and malignant tumours showed variable enhancement patterns. In conclusion, signal intensity ratios for T(2) weighted and STIR images, ADC values and patterns of enhancement may help distinguish Kuttner's tumours from benign submandibular gland tumours, but not from malignant tumours. Although the intensities, ADC values and enhanced patterns of Kuttner's tumours were similar to those of malignant tumours, there were some morphological differences.

大唾液腺腫瘍１９８例の臨床検討

Between 1995 and 2005, 198 cases of major salivary gland tumors underwent surgery in our department, including 158 parotid gland tumors, 38 submandibular gland tumors and 2 sublingual gland tumors.The number of parotid tumors increased annually, although submandibular gland tumors did not demonstrate any increasing trend.The ratio of malignant tumors among submandibular gland tumors was 24%, which was higher than among parotid tumors (11%). In addition, the ratio of the malignant tumors among submandibular gland tumors in males (42%) was significantly higher than that in females (5%).Fine needle aspiration biopsy was the most useful for diagnosis of the malignant tumors on preoperative examinations.Permanent paralysis of the facial nerve after resection of parotid tumor was 29% in malignant tumors, 6% in benign tumors and 8% among parotid tumors overall. Frey's syndrome was occurred in 12%.

Submandibular Gland Carcinoma With Unusual Widespread Bone Marrow Metastases Detected by Tc-99m MDP Bone Scan and FDG PET

We report a case of a 33-year-old woman with undifferentiated submandibular gland carcinoma, status post excision and external radiotherapy. Three years after radiotherapy, primary tumor control was considered to be satisfactory, but she complained of upper back pain. A Tc-99m MDP whole body bone scan was done and exhibited diffuse uptake in the axial skeleton and relatively decreased tracer uptake in the extremities. Positron emission tomography demonstrated increased F-18 FDG uptake over the whole spine and pelvis. Immunohistochemical staining of an iliac crest bone marrow biopsy corresponded to features of submandibular gland tumor cells.

Malignant Submandibular Gland Tumors: Failure Analysis and Role of Postoperative Radiotherapy

Malignant Submandibular Gland Tumors: Failure Analysis and Role of Postoperative Radiotherapy

Submandibular Gland Excision: 15 Years of Experience

The surgical management of submandibular gland diseases has always been a challenge because it carries a considerable risk of nerve injury. The aim of this study was to review a single institution's experience of a nonselected case series of submandibular gland excision over 15 years. We retrospectively analyzed 258 unselected submandibular excisions of a tertiary university center for the histopathologic diagnosis and postoperative morbidity; 119 patients (46%) with sialolithiasis, 88 patients (34%) with sialadenitis, and 51 patients (20%) with submandibular tumors were operated. We found a high rate of malignant tumors (42%) in the group of submandibular gland tumors. A low rate of transient palsies of the mandibular branch of the facial nerve (9%) and lingual nerve (2%) was observed. One patient developed a permanent paresis of the mandibular branch (<1%). Our large series has shown that standardized submandibular sialadenectomy is a safe operation with a low rate of complications. Malignant disease is frequent in tumors of the submandibular gland.

腎細胞癌顎下腺転移の２例

Renal cell carcinoma can metastasize to any part of the body. We encountered two cases of renal cell carcinoma that metastasized to the submandibular gland.The first case was a 74-year-old man and the second case was a 78-year-old man. Each patient complained of a painless swelling of the right submandibular gland after nepfrectomy for renal cell carcinoma. In both cases, we performed resection of the submandibular gland. Pathological examination demonstrated metastatic renal cell carcinoma to the submandibular gland. Metastatic renal cell carcinoma as a submandibular gland tumor is rare, and we discussed metastasis of renal cell carcinoma especially to the submandibular gland.

Czy zabiegi endoskopowe stanowią przyszłość chirurgii szyi?

Summary The aim of the paper is to present the minimally invasive endoscopic video-assisted surgery for the neck on the basis of current literature. The different techniques include pure endoscopic procedures, open video-assisted approach and miniaccess. The preliminary material constitute of three patients who undergone endoscopic sentinel lymph node biopsy and one with colli lipoma. The authors describe the operating technique, instrumentation, indications, contraindications and advantages of this method. The endoscopic surgery offered a valuable approach for chosen lesions of the neck (solitary parathyroid adenoma, thyroid nodules less then 35 mm, submandibular gland sialolithiasis or tumors). The advantages include lower pain intensity within first hours following surgery, lower analgesia request rate, shorter scar length, higher early cosmetic satisfaction.

Neurofibroma of the Lingual Nerve: A Case Report

A neurofibroma of the lingual nerve is a rare clinical finding, the most common lesion site of a lingual neurofibroma being the tongue. In most situations, it is difficult to determine the precise nerve origins. Herein, we report a case of lingual nerve neurofibroma that presented as a submandibular mass, mimicking a submandibular gland tumor or solitary lymphadenopathy. Complete surgical excision of such a lesion for histopathologic examination provides a better treatment and final diagnosis. For a patient presenting with neurofibromatosis and a submandibular mass, a neurofibroma of nerve origin should be considered in the differential diagnosis.

A clinical analysis of patients with submandibular gland tumors

A retrospective investigation was performed on 33 patients with submandibular gland tumors treated between 1998 and 2005. The tumors consisted of 17 benign and 16 malignant types. Fifteen of 16 malignant tumors were cancerous, and the most common histological type was adenoid cystic carcinoma (33.3). The accuracy of FNAB (fine-needle aspiration biopsies) was 84%, eight of 12 aspirates from malignant tumors were correctly diagnosed by FNAB, for a sensitivity of 66.7%, while all of 13 aspirates from benign tumors were correctly diagnosed by FNAB, for a specificity of 100%. The five-year survival rate calculated by the Kaplan-Meier method was 81.3% in 15 patients with submandibular gland cancer. Pathological neck lymph node metastasis in all NO cases was detected in level 2, so we concluded that supraomohyoid neck dissection might be suitable for NO cases of submandibular gland cancer. To improve the prognosis of submandibular gland cancer, postoperative chemotherapy may be necessary for T3 and T4 cases.

唾液腺腫瘍１５１例の臨床統計的検討

One hundred fifty-one patients with salivary gland tumors histologically diagnosed at the Department of Oral and Maxillofacial Surgery, Kyushu University Hospital from 1982 through 2001 were clinicostatistically studied. The following results were obtained: 1. Of the total, 56.3% had benign tumors and 43.7% had malignant tumors.2. Pleomorphic adenomas were most common, comprising 47.8% of all tumors and 84.7 % of benign tumors.3. Both benign tumors and malignant tumors occurred more frequently in women.4. The average age of the patients with malignant tumors (56.6 years) was higher than that of the patients with benign tumors (49.3 years).5. With respect to the primary site, parotid gland tumors were most common, comprising 33.8% of all tumors. All sublingual gland tumors (100%) were malignant.6. The cause-specific cumulative survival rate for all patients with malignant tumors was 67.6% at 5 years and 61.7% at 10 years. Mucoepidermoid carcinoma had a good outcome, but adenoid cystic carcinoma and carcinoma in pleomorphic adenoma had poor outcomes. According to primary site, submandibular gland tumors were associated with a significantly lower cumulative survival rate than minor salivary gland or parotid gland tumors.

Distant metastasis of parotid gland tumors

The incidence of distant metastasis in head and neck cancer and especially in salivary gland cancer is relatively low in comparison to other malignancies. However, the presence of distant metastasis heralds a poor prognosis in head and neck cancer, with a median survival of 4.3–7.3 months. Treatment of these patients is usually performed in a palliative setting. Patients with malignant salivary gland tumors should have an X-ray or CT scan of the chest at their initial assessment to exclude the possibility of distant metastasis. The likelihood of developing distant metastasis is associated with high-grade tumors, such as adenoid cystic carcinoma, salivary duct carcinoma, high-grade mucoepidermoid carcinoma and tumors located in the submandibular gland, posterior tongue and pharyngeal tumors. A lower risk of developing distant metastasis is known for all other histological entities of salivary gland tumors. Nevertheless all patients who have a histologically confirmed malignant salivary gland tumor should have lifelong follow-up. On the basis of a clinical case regarding a patient with metastatic parotid gland cancer we present a review of the literature.

Molecular characterization of salivary gland malignancy using the Smgb-Tag transgenic mouse model

The molecular mechanisms underlying salivary gland tumorigenesis remain unclear. In order to identify genetic changes that occur during the development of invasive adenocarcinoma from normal salivary gland, we used the Smgb-Tag transgenic mouse model. This transgene induces the progressive development of dysplasia to invasive adenocarcinoma in the submandibular salivary gland. Gene expression patterns from 20 submandibular glands (two normal, nine dysplasia and nine adenocarcinoma samples) were assessed using a mouse 15 K cDNA array. Unsupervised hierarchical clustering was used to group gene expression based on 157 differentially expressed genes distinguishing between dysplasias and adenocarcinomas. Further analysis identified 25 significantly overexpressed and 28 underexpressed cDNA sequences in adenocarcinoma as compared to dysplasia. Differential expression of five genes (Lcn2, Ptn, Cd24a, Mapk6 and Rnps1) was validated by quantitative real-time RT-PCR in a total of 48 mouse salivary gland tissues (seven histologically normal, 13 dysplasias and 28 adenocarcinomas), including the 20 samples analyzed by cDNA arrays. Immunohistochemical analysis was used to validate the expression of Ptn and Cd24a at the protein level in a subset of 16 mouse salivary glands (four normal, five dysplasia and seven adenocarcinoma samples), as well as in 23 human submandibular gland tumors (16 pleomorphic adenomas, three adenoid cystic carcinomas, one acinic cell carcinoma, one adenocarcinoma NOS, one myoepithelial and one mucoepidermoid carcinoma). We thus demonstrated that the Smgb-Tag transgenic mouse model is a useful tool for the identification of genes that are deregulated in salivary gland adenocarcinomas. Our data suggest that Ptn and Cd24a may be genetic markers associated with salivary gland tumorigenesis and/or progression.

Differentiation of a mouse submandibular gland-derived cell line (SCA) grown on matrigel

SCA-9 cell line was developed from an induced tumor of mouse submandibular gland ( T. Barka, H. van der Noen, A.M. Michelakis, I. Schenkein, Epidermal growth factor, renin, and peptidase in cultured tumor cells of submandibular gland origin. Lab. Invest., 42 (1980) 656–662). We have studied some of the phenotypic characteristics of SCA cells cultured on different matrices. On plastic surface, the cells grow as a monolayer; on matrigel, they form branching structures and tubes, a phenomenon termed branching morphogenesis. EGF and HGF promoted cellular growth and branching morphogenesis which was inhibited by anti-EGF antibodies. We have performed RT-PCR and real-time quantitative RT-PCR of cells grown on plastic surface or on matrigel. Grown on plastic, the cells express EGF and renin 2, but no or only trace amounts of NGF. Growth on matrigel for 24 h resulted in a transient 21-fold increase in EGF mRNA and a 3371-fold increase in renin 2 mRNA. There was no change in NGF mRNA level. SCA-9 cells express mRNAs for receptors for the EGF family of ligands. On plastic, mainly ErbB1 and ErbB2 are expressed. Culture on matrigel resulted in 11-fold increase in mRNA levels for ErbB1 and ErbB2, and a 221-fold and 85-fold increase in the mRNA levels for ErbB3 and ErbB4, respectively. Small interfering RNAs siErbB3 and siErbB4 inhibited the growth of the cells grown on plastic or matrigel. Significant growth inhibition was seen also with siErbB1 + siErbB3 and siErbB2 + siErbB3. siErbB1 and siErbB2 also inhibited branching morphogenesis. Since SCA cells express EGF and receptors for EGF, EGF acts an autocrine regulator in promoting growth and branching morphogenesis. We conclude that SCA cells provide a useful model to analyze the mechanism of branching morphogenesis and the role of matrix in regulating expression of phenotypic characteristics of cultured cells.