Epithelial–myoepithelial carcinoma of a minor salivary gland in the buccal mucosa

Epithelial–myoepithelial carcinoma (EMC) is a rare tumor constituting less than 1 % of all salivary gland tumors. This neoplasm most often involves the parotid glands, with less frequent involvement of the submandibular or minor salivary glands. This case report describes EMC arising from a minor salivary gland in the buccal mucosa. A 71-year-old Japanese man with a painless submucosal mass in the left buccal region was referred to our hospital. The patient stated that the tumor had been present for more than 4 years and was painless but increasing in size. Oral examination showed a mobile, well-defined mass of approximately 19 × 15 mm; its surface was covered mostly by normal mucosa. Magnetic resonance imaging showed a mass with well-defined borders and relative homogeneity internally. Incisional biopsy led to a histological diagnosis of a suspected pleomorphic adenoma or low-grade malignant tumor originating from the salivary gland. The patient underwent surgical excision under general anesthesia to obtain a definitive diagnosis, and EMC was diagnosed based on histopathological and immunohistochemical examination of the whole specimen. Because the surgical margins were positive in some areas, the patient underwent external postoperative radiotherapy (total of 60 Gy at 2 Gy/day). Recovery was uneventful, and good function returned after postoperative treatment. Four years after tumor resection and radiotherapy, no recurrence had been detected and no other lesions had developed.     

Epithelial-myoepithelial carcinoma of the submandibular gland with a high uptake of 18F-FDG: a case report and image diagnosis

Epithelial-myoepithelial carcinoma (EMC) is a rare low-grade malignant salivary gland neoplasm typically found in the parotid gland. This report describes a case of EMC that arose from the submandibular gland, and gives special emphasis to the preoperative diagnostic value of positron emission tomography (PET) in combination with computed tomography (CT) and magnetic resonance imaging (MRI). Although a preoperative diagnostically specific image of EMC could not be established, the malignant nature of this tumor was detected by these combined examinations.     

Iodine-125 brachytherapy for the treatment of a large parotid epithelial-myoepithelial carcinoma in a child

Epithelial-myoepithelial carcinoma (EMC) in the salivary glands is rare. Tumours originating from salivary glands are also rare in children. Radical resection is the main treatment used for EMC in salivary glands. However, this surgery is commonly associated with functional and/or cosmetic deficits. There is also a high percentage of local recurrence after a tumourectomy. We present a typical case of recurrent EMC in the left parotid gland of an 8-year-old girl. The patient was treated with iodine-125 brachytherapy. At the 2-month follow-up, the tumour size was reduced by 80%, and at 1 year, no tumour tissue was detected on positron emission tomography/computed tomography. At the 6-year follow-up, no local recurrence or distant metastasis was found and no complications had occurred. Iodine-125 brachytherapy is a potentially appropriate alternative treatment for EMC in the salivary glands, especially for paediatric patients.     

Epidermal growth factor in the submandibular salivary glands of congenitally athymic mice.

The submandibular salivary glands of a group of congenitally athymic (“nude”) mice were assayed for their epidermal growth factor (EGF) content and their histology was examined by light microscopy. The ability of the submandibular salivary glands from athymic mice to respond to an androgenic agent was assessed. The histology of the submandibular salivary glands resembled that of normal mice. Athymic mice had concentrations of EGF in their submandibular salivary glands which were similar to those reported previously for normal mice As in normal mice, male athymic mice had more prominent granular convoluted tubules than female mice, but, as in normal mice, testosterone treatment of female athymic mice resulted in an increase in both the EGF content and in the number of granules within the cells of the granular tubules of the duct system of the submandibular salivary gland.These results provide no evidence for a relationship between the level of submandibular salivary gland EGF and the immunological deficiency of nude mice, and show that the abnormalities in development of nude mice do not extend to their submandibular salivary glands.     

Ipsilateral removal of sublingual gland after excision of submandibular gland for benign disease: 10-year prospective study and comprehensive review 1978–2008

The removal of the submandibular salivary gland for non-neoplastic disease is a common procedure that has well documented risks and postoperative complications. Persistent symptoms of pain and swelling in the floor of the mouth that can occur after excision of the submandibular gland may require removal of the sublingual gland, but a causative association between the two has not, to our knowledge, been comprehensively established. We prospectively studied 77 patients who had had excision of the submandibular gland for benign disease, six of whom (8%) returned to theatre for ipsilateral sublingual sialadenectomy within a 5-year period after the initial operation (mean 24 months). These findings suggest that the association is under-reported, and may need to be considered during the consent process for excision of the submandibular salivary gland.     

Fine needle aspiration cytology in the diagnosis of amyloid in the submandibular gland

This paper describes how fine needle aspiration cytology (FNAC) was used to diagnose amyloid in a patient who had multiple myeloma and had developed xerostomia and submandibular salivary gland enlargement.     

Prognostic factors in primary salivary gland mucoepidermoid carcinoma: an analysis of 376 cases in an Eastern Chinese population

Mucoepidermoid carcinoma (MEC) is an infrequent malignant neoplasm that originates most commonly in the salivary glands. The present study aimed to provide new information on prognostic factors in patients with salivary gland MEC. A retrospective analysis of the medical records of patients diagnosed with primary salivary gland MEC between 2003 and 2010 was conducted. The incidence of MEC in the minor salivary glands (62.2%) was almost twice that in the major salivary glands (37.8%). The most frequently affected sites were the parotid gland and palate. Lymph node metastasis was reported more frequently in male than female patients (P = 0.02), in high-grade than low/intermediate grade lesions (P < 0.001), and in lesions involving the submandibular gland (P < 0.001). The disease-free survival (DFS) at 5 years was 80.47%, with rates of 98.0%, 86.5%, and 38.5% for low-, intermediate-, and high-grade tumours, respectively. Among various clinicopathological factors, the only independent prognostic factor was histological grade (P < 0.001). Primary tumour site and histological grade are two important factors affecting cervical lymph node metastasis. Histological grade is the only independent factor affecting survival beyond tumor lymph node metastasis (TNM) staging in salivary gland MEC. Further advances in therapy are needed to improve the outcomes for patients with high-grade lesions.     

Sialolithiasis: an unusually large submandibular salivary stone

Salivary gland calculi account for the most common disease of the salivary glands. The majority of sialoliths occur in the submandibular gland or its duct and are a common cause of acute and chronic infections. This case report describes a patient presenting with an unusually large submandibular gland sialolith, the subsequent patient management, the aetiology, diagnosis and various treatment modalities available for management of salivary gland calculi depending on their site and size.     

Synchronous lymphomas arising in ipsilateral parotid and submandibular glands: video-assisted surgery

The incidence of lymphomas in salivary glands is 5%, and it represents 10% of malignant salivary tumors. Synchronous tumors arising in different major salivary glands are not only very infrequent but also extremely rare; on the other hand, synchronous lymphomas in ipsilateral major salivary glands have not been reported previously. This article reports a case of synchronous lymphomas in mucosa-associated lymphoid tissue in the ipsilateral parotid and submandibular glands. Parotidectomy with facial nerve conservation and resection of the submandibular gland by video-assisted surgery using only a Blair-modified classic incision was performed. One year after the surgical procedure, the patient was followed up at regular intervals, and residual mucosa-associated lymphoid tissue lymphoma did not appear. This case illustrates the importance of the diagnostic possibility of synchronous primary tumors after identification of a simple one; this also reports the video-assisted resection of the submandibular gland using only a Blair incision, when a parotidectomy must be also made.     

Alteration in salivary function in early HIV infection

The etiology of salivary gland hypofunction in HIV(+) patients is unclear. This study was designed to determine the effect of early-stage HIV(+) infection (CD4(+) > 200 cells/ micro L; n = 139) on salivary gland function and the relationship of this dysfunction to the taking of xerostomic medications. Salivary flow rates and the content of electrolytes and antimicrobial proteins in stimulated parotid and submandibular/sublingual saliva were determined. Compared with healthy controls (n = 50), the HIV(+) group showed significant reductions in flow rates of unstimulated whole (35%), stimulated parotid (47%), unstimulated submandibular/sublingual (23%), and stimulated submandibular/sublingual (39%) saliva. The flow rates for the HIV(+) patients taking xerostomic medications did not differ from those of patients who did not. Concentrations of some salivary gland components were altered in the HIV(+) group. Analysis of these data suggests that salivary gland function is adversely affected early in HIV infection and that these changes do not appear to be compounded by the taking of xerostomic medications.     

Dedifferentiated epithelial-myoepithelial carcinoma of the parotid gland: a rare case report of immunohistochemical analysis and review of the literature

Dedifferentiation of salivary gland neoplasms is a rare event, unlike bone and soft part sarcomas, which was first described by Stanley et al. in 1988. An additional case of dedifferentiated epithelial-myoepithelial carcinoma (EMC) is reported here. The patient was a 70-year-old Japanese man who requested examination of the rapid growth of a mass in the right parotid region, which he had first noticed 25 years previously. Clinical examination showed an ill-circumscribed, 6.8 x 4.7 x 7.0-cm lesion. Histologically, most parts of the lesion were high-grade carcinoma (HGC) with sheetlike and nestlike growth of markedly atypical cells and comedonecrosis, whereas the minor part consisted of typical EMC. The outer clear cells of EMC were positive for alpha-smooth muscle actin (ASMA), p63, cytokeratin (CK) 14, and vimentin, and the inner ductal cells of EMC were positive for CKs and epithelial membrane antigen. HGC was negative for ASMA, CK14, and vimentin, but diffusely positive for p53 protein and cyclin D1. The Ki-67 labeling index of EMC was 11.5%, whereas that of HGC was 67.1%. These findings and a review of literature indicate that HGC arose from preexisting EMC, and this phenomenon is the dedifferentiation of EMC. Dedifferentiated EMC is extremely rare.     

4种涎腺肿瘤及人胚颌下腺免疫组化对比研究

目的：探讨涎腺肿瘤细胞的形态分化、组织发生及其异质性。方法：采用光镜、免疫组化技术对８例腺淋巴瘤、３例嗜酸性腺瘤、１２例多形性腺瘤及２例上皮－肌上皮癌进行研究,同时观察了５６例胚胎颌下腺组织作为对照。结果：多形性腺癌中导管样结构系肿瘤性闰管结构和肿瘤性分泌管结构;腺淋巴瘤和嗜酸性腺瘤中导管结构系肿瘤性分泌管结构;上皮－肌上皮癌中导管结构系肿瘤性闰管结构;涎腺肿瘤组织结构及抗原表达复杂多样,与人胚涎腺发生过程中结构及抗原表达有相似之处。结论：涎腺肿瘤的复杂性系致瘤因素作用于不同干细胞或同一干细胞不同分化阶段所致。     

A prospective study of salivary gland function in patients undergoing radiotherapy for squamous cell carcinoma of the oropharynx

OBJECTIVE: We sought to investigate the impact of head and neck cancer treatment on salivary function. STUDY DESIGN: The study was conducted on 54 patients with advanced squamous cell carcinoma with confirmed (n = 50) or suspected (n = 4) primary oropharyngeal localization who were treated with radiation alone or in combination with surgery or chemotherapy, or both. The following groups were considered in the evaluation: 1, the entire pool of patients; 2, those undergoing surgery and those not undergoing surgery before radiation; 3, those undergoing resection and those not undergoing resection of the submandibular gland. The flow rates, pH, and buffering capacity were determined before, during, and up to 12 months after the completion of radiation. RESULTS: Head and neck surgery, particularly when submandibular gland resection was performed, had a negative impact on salivary flow rates but did not influence pH or buffering capacity. Nonetheless, the effect of surgery on salivary flow rates decreased progressively and disappeared at 3 to 6 months after radiotherapy. More than two thirds of the salivary output was lost during radiation treatment. All patients were experiencing salivary dysfunction at 1 year after completion of radiotherapy, with average decreases of 93% (P < .0001) and 95% (P < .0001) for whole resting salivary flow and whole stimulated salivary flow, respectively, compared with the preradiotherapy values. The buffering capacity decreased to 67% of its preradiotherapy value, and whole stimulated saliva became acidic. CONCLUSIONS: The result of this study confirms that cancer treatment involving full-dose radiotherapy (RTH) to all major salivary glands for locally advanced squamous cell carcinoma of the oropharynx induces severe hyposalivation with alteration of salivary pH and buffering capacity. Head and neck surgery has a negative impact on salivary flow rates, especially when the submandibular gland is removed. However, surgery before irradiation is not a factor aggravating hyposalivation when postoperative radiotherapy includes all the major salivary glands.     

Epidermal growth factor concentration in hyperplastic and hypertrophic submandibular salivary glands of mice.

Submandibular salivary glands of mice contain high concentrations of epidermal growth factor (EGF). The EGF content of mouse submandibular salivary glands undergoing hyperplastic and hypertrophic changes was measured and compared to that in the glands of control mice. Salivary gland hyperplasia was induced by giving mice a single injection of isoprenaline and hypertrophy was produced either by repeated, daily injections of isoprenaline, repeated amputation of a lower incisor tooth or by removing the right submandibular salivary gland and thus producing a compensatory hypertrophy of the left submandibular gland. The EGF content of the hyperplastic submandibular salivary glands was not different from that of the control glands. While the EGF content of the hypertrophied glands resulting from either repeated isoprenaline injections or partial sialoadenectomy did not differ from that of the control glands, the concentration of EGF was significantly lower. This reduced concentration is probably a reflection of acinar hypertrophy with a resultant smaller proportionate contribution of the granular tubules to the mass of the gland. Incisor-amputation-induced hypertrophy did not result in a reduced concentration of EGF in the submandibular salivary glands, but the reason for the different response is unknown.The findings provide no evidence for the involvement of EGF in the induced changes of submandibular salivary gland hyperplasia or hypertrophy resulting from either isoprenaline treatment or partial sialoadenectomy. The reason for the higher concentration of EGF in hypertrophied submandibular salivary glands resulting from incisor amputation compared to that measured in hypertrophied glands resulting from the other stimuli used remains unresolved.     

Metastasis to the submandibular gland as the initial presentation of small cell ("oat cell") lung carcinoma

A 45-year-old man had a submandibular mass, which was resected. Histopathologic evaluation and the subsequent course revealed it to be a metastasis from a small cell carcinoma of the lung. This is the first case in which a metastatic lesion in the submandibular gland was the presenting manifestation of an extrasalivary neoplasm, predating discovery of the primary tumor. Metastases to the salivary glands in general and to the submandibular gland in particular are discussed, as is the differential diagnosis with primary salivary gland carcinoma.     

下颌下腺涎石病伴发黏液表皮样癌1例报告

黏液表皮样癌是最常见的唾液腺恶性肿瘤之一,最常发生于腮腺（约占2/3）,其次为腭部、小唾液腺（特别是磨牙后腺）及下颌下腺,而发生于由涎石病导致的慢性炎性下颌下腺者极为罕见。本文报告1例发生于慢性炎性下颌下腺中的黏液表皮样癌病例,并对其诊断和治疗方法进行讨论。     

The effect of food consistency and dehydration on reflex parotid and submandibular salivary secretion in conscious rats

Changes in salivary secretion with different consistency of diet and dehydration were studied in male Wistar rats under unrestricted conditions. To measure the salivary secretion, a stop-flow method was used. There was little unstimulated salivary secretion from the parotid and submandibular glands, but eating solid, powdered, and liquid diets induced parotid and submandibular saliva. There was no significant change in the volume and flow rate of saliva in bilateral parotid glands during the eating of solid diets. The solid and powdered diets induced significantly more salivary secretion from the parotid gland than did the liquid. The salivary flow rate with solid diets was significantly greater from the parotid gland than from the submandibular. On the other hand, the salivary flow rate with the liquid diet was significantly smaller from the parotid gland than from the submandibular. Appreciable amounts of submandibular saliva, but little parotid saliva were secreted during grooming. Clearly, parotid and submandibular saliva have different roles in the rat. When injected intraperitoneally with 1.5 M NaCl solution or water-deprived for 24 h, rats took longer to eat the solid diets, and had increased salivary volume and decreased flow rate from the parotid gland. These results indicate that the moisture content of the diet and the dryness of the mouth alters the volume of parotid saliva secreted in rats and show that parotid saliva plays an important part in mastication and swallowing.     

Clinicopathological factors are predictors of distant metastasis from major salivary gland carcinomas

The risk of distant metastasis of salivary gland cancers has usually been associated with histological type, tumour size, and site. The aim of this study was to evaluate a series of patients with major salivary gland carcinomas in order to identify potential risk factors associated with distant metastasis. 255 patients treated for major salivary gland carcinoma in Brazil from 1953 to 2004 were reviewed. Clinical and treatment data were obtained from the medical records and histological features reviewed. 57 (22%) of 255 patients had distant metastasis. The lungs were the most common metastatic site (40 cases, 65%) and adenoid cystic carcinoma the most frequent histological type involved (27 cases, 47%). The percentage of tumours in the submandibular, parotid, and sublingual glands that presented distant metastasis was 42%, 20%, and 17%, respectively. These results provide evidences that clinicopathological factors (tumour site and histology) are significant predictors of distant metastasis in patients with major salivary gland carcinomas.     

Synchronous pleomorphic adenomas of the major salivary glandsA case report

The presentation of multiple distinct tumors in major salivary glands is rare. Although the most common tumor with bilateral synchronous or metachronous development is the Warthin tumor, pleomorphic adenomas have been diagnosed simultaneously as well. We report the case of a female patient who was diagnosed with pleomorphic adenomas in the right parotid and submandibular glands, concomitant with sialolithiasis affecting the submandibular gland. This patient had been exposed to head and neck radiotherapy in childhood, which may have played a role in the development of her tumors. A review of the relevant literature is included.