Malignant salivary gland tumors: squamous cell carcinoma of the submandibular gland in a child.

PURPOSE: Malignant neoplasms of the salivary glands are extremely unusual in the pediatric age group. METHODS: We report an 11-year-old boy who presented with a mass in the left submandibular region and a second mass in the jugularfacial venous angle. RESULTS: Histologic evaluation determined that this was a squamous cell carcinoma of the submandibular gland with metastasis to a cervical node. Review of the literature was undertaken to identify the rate of malignant salivary gland tumors in children. Malignant salivary gland tumors are extremely rare in children. When malignancy does occur, nearly 90% are present in the parotid gland. The submandibular gland is effected in 7.7% of cases. The most common malignancy in salivary glands of children is the mucoepidermoid carcinoma. Squamous cell carcinoma occurs in less than 2% of cases. CONCLUSIONS: Salivary gland tumors are very rare in children in contrast to adults. The proportion of malignant tumors in children is higher than that observed in adults; however, it is not possible to draw conclusions concerning treatment from the few case reports that exist. A higher rate of local recurrence and cervical lymph node metastasis may be expected in children.     

Histology of submandibular gland tumours, 10 years' experience

Introduction and objectivesThere are few reports focused on neoplasms in the submandibular gland because they are uncommon and are frequently grouped with the other salivary glands tumours. In the classical studies, the proportion of malignancy reported in these cases is around 50%.ObjectivesDetermining the proportion of malignancy, the most frequent histological types, the gender distribution and average age at diagnosis in patients with submandibular gland neoplasms who were treated in our hospital from 2000 year to 2010.MethodsA retrospective review of our department database of the patients who underwent surgery for submandibular gland neoplasm in a ten-year period was performed.ResultsTwenty-two patient records were included, in which 19 (86%) patients presented benign disease and 3 were of a malignant type (adenocarcinoma, well-differentiated epidermoid carcinoma, follicular lymphoma). Two of the 3 cases of malignant neoplasms were in men.ConclusionsA greater proportion of benign neoplasm was found in submandibular tumours. The most frequent benign tumour is the pleomorphic adenoma. Women are more commonly affected (76%). Benign tumours appear in younger patients than do malignant ones.     

Non-neoplastic salivary gland lesions: a 15-year study

The spectrum of salivary gland lesions is wide and the relative incidence of neoplastic versus non-neoplastic lesions is variable in different studies. A series of non-neoplastic salivary gland lesions is reviewed to analyze their spectrum and their relative frequency. This is a retrospective study of salivary gland excisions and biopsies received in our department from January 1994 to December 2008. Routine hematoxylin and eosin-stained sections of all the salivary gland excisions and biopsies received were analyzed. Of the 393 salivary gland excisions and biopsies received, 216 cases were reported as non-neoplastic (55%) and formed our study group; 177 (45%) were neoplastic. Non-neoplastic lesions were more frequent in major salivary glands (65.7%) and submandibular gland was the most commonly involved (66.2%). Lip was the most frequent site (81.7%) for minor salivary gland lesions. Inflammation was the predominant pathological finding (49.5%), of which non-specific chronic sialadenitis constituted the majority (86.9%). Sialolithiasis was present in 22 cases (20.6%); all of these cases were of non-specific chronic sialadenitis. Cysts were second in frequency (36.6%), of which mucocele was the most common (54.5%). There were 5.6% cases of benign lympho-epithelial lesions, while normal salivary gland tissue was seen in 6.5% cases. Non-neoplastic salivary gland diseases are more common than neoplastic diseases and have a wide disease spectrum.     

Myoepithelial neoplasia of the submandibular gland: case report and therapeutic considerations

Tumors of the submandibular gland typically arise from the seromucinous acini, which make up the majority of the gland. The most common benign tumor of this structure is the pleomorphic adenoma, whereas the most common malignancy of the submandibular gland is adenoid cystic carcinoma. We describe an unusual case of a neoplastic process of the myoepithelial cells of the submandibular gland in a middle-aged woman. This rare tumor is most commonly diagnosed in the parotid gland and in the minor salivary glands of the hard palate; a review of the literature uncovered only 5 previous reports of myoepithelioma of the submandibular gland. Distinguishing myoepithelioma from benign pleomorphic adenoma and malignant myoepithelial carcinomas can be challenging. Immunohistochemical staining can help to distinguish between the benign neoplasms, but histologic features remain the "gold standard" for diagnosing the malignant tumors. Increasing use of immunohistochemistry panels to assess parotid neoplasms also suggests that myoepithelioma may be underrecognized.     

Submandibular gland excision

A review of 107 submandibular gland excisions was made to determine the relative frequency of neoplastic versus non-neoplastic disease. The results show a preponderance of non-neoplastic pathology. Benign tumors were found in 6% of glands, primary glandular and metastatic carcinoma in 3%. Timely treatment of a submandibular gland mass is indicated due to the relatively aggressive behavior of malignancies originating in this location.     

Paediatric salivary gland epithelial neoplasms

Salivary gland epithelial neoplasms in children are rare. Clinicians of all disciplines need to have a high level of suspicion when a non-inflammatory single mass lesion presents in the parotid or submandibular glands. There is high likelihood of malignancy when such a mass presents. Surgery is the primary treatment of choice in the majority of cases, with the addition of adjuvant radiotherapy +/- chemotherapy when the diagnosis is a high-grade tumour and/or when the malignancy presents as a large mass or is associated with local tissue invasion. Minor salivary gland neoplasms also present, the oral cavity is most frequent, with pleomorphic adenoma and mucoepidermoid carcinoma being most common, other malignant neoplasms have been reported in other sites.     

Kaposi's sarcoma of an intraparotid lymph node leading to a diagnosis of HIV

OBJECTIVE: Kaposi's sarcoma is a common malignancy in patients infected with HIV but is rarely seen in the major salivary glands. If a patient is known to be HIV-positive, however, Kaposi's sarcoma must be considered in the differential diagnosis of salivary gland masses in addition to the benign and malignant neoplasms that occur in immunocompetent patients. We present a unique case in which an otherwise healthy patient was diagnosed with HIV after resection of his enlarged parotid gland revealed Kaposi's sarcoma. STUDY DESIGN: Case report. METHODS: A 58-year-old man presented with slowly enlarging bilateral parotid masses of approximately 3 years' duration. The patient's presentation, workup, and final diagnosis of Kaposi's sarcoma are discussed. RESULTS: A magnetic resonance imaging scan of the neck showed two right parotid lesions and one left parotid mass. The patient underwent a right superficial parotidectomy for a suspected diagnosis of Warthin's tumor, given the bilaterality of the lesions. Histologic evaluation of the surgical specimen revealed spindle-shaped cells with extravasated erythrocytes typical of Kaposi's sarcoma. After discussion of the results with the patient, HIV risk factors were elucidated, and subsequent testing revealed the patient to be HIV-positive. CONCLUSIONS: Although Kaposi's sarcoma is common in AIDS patients, there are few case reports of this malignancy arising in the salivary glands. Previously reported cases include salivary gland Kaposi's sarcoma in known HIV-positive patients and a handful of reports in patients without confirmed immunocompromise. The patient presented here is unique because the diagnosis of parotid gland Kaposi's sarcoma led to a new diagnosis of HIV. This interesting case reiterates the need for complete history taking and the inclusion of Kaposi's sarcoma in the differential diagnosis of salivary gland masses in the appropriate patient population.     

Mucoepidermoid carcinoma of the submandibular gland after chemotherapy in a child

Second malignant neoplasms (SMNs) have become a concern in survivors of childhood malignancy. Although there are many reports describing SMN in patients treated for childhood cancer, salivary gland tumors rarely appear in these reports. Radiotherapy is a well-known risk factor for the development of secondary salivary gland malignancies after the treatment of childhood cancer. However, it is not well known whether chemotherapy itself treatment increases the risk of salivary gland malignancies. We report a child case with mucoepidermoid carcinoma of the submandibular gland as a SMN after chemotherapy alone for acute myeloid leukemia.     

An analysis of salivary gland neoplasms: a 12-year, single-institution experience in Turkey

The epidemiology of salivary gland tumors worldwide is not very well defined. Although many studies on this subject have been undertaken, the data are generally focused on specific topics such as parotid gland neoplasms or tumors of the major salivary glands. We conducted a study to establish the prevalence and distribution of benign and malignant neoplasms of both the major and minor salivary glands at a single institution. We reviewed 244,204 cases that had come through our pathology department from January 1994 through December 2005 and found 235 cases of a salivary gland neoplasm (0.09%). The female-to-male ratio was 1.04:1, and the mean age of the patients was 47 years. Of the 235 neoplasms, 159 (67.66%) were located in the parotid gland, 34 (14.47%) in the submandibular gland, and 42 (17.87%) in the minor salivary glands. A total of 146 tumors (62.13%) were benign and 89 (37.87%) were malignant. Pleomorphic adenoma was the most common neoplasm, occurring in 98 cases (41.70%). The most common malignancy was mucoepidermoid carcinoma, with 27 cases (11.49%). Our data demonstrate that the characteristics of salivary gland tumors in a Turkish population at a single institution are similar to those reported in the literature worldwide.     

Cystadenocarcinoma (papillary cystadenocarcinoma) of the submandibular gland

Cystadenocarcinoma (papillary cystadenocarcinoma) of the salivary gland is a rare malignant neoplasm. Major locations of this neoplasm are the parotid gland, the sublingual gland, and minor salivary glands, while occurrence in the submandibular gland is extremely rare. As far as we know, only one report, written in the French language, has been published concerning cystadenocarcinoma of the submandibular gland, but no report is available in the English language. In this report, we describe a case of cystadenocarcinoma arising from the submandibular gland of a 54-year-old male patient. The neoplasm is a low-grade carcinoma, and pre-operative examination may not show typical characteristics of malignant neoplasms. Therefore, as was true in this case, the differential diagnosis from benign lesions is sometimes difficult. This is the first report on cystadenocarcinoma of the submandibular gland in the English language and the first to show a computed tomography (CT) scan and magnetic resonance imaging (MRI) of this neoplasm in the submandibular gland.     

Masses of the salivary gland region in children

BACKGROUND: Noninflammatory masses of the salivary gland region in children are extremely rare. Therefore, very few published individual and institution-based experiences exist. DESIGN: Retrospective chart review from 1990 through 1997. SETTING: University-based children's hospital. DESIGN: Patients 18 years of age or younger with a tumor in the salivary gland region. Masses of infectious origin were excluded. Hemangiomas and lymphangiomas were tallied for relative incidences only. RESULTS: Three hundred twenty-four consecutive cases of salivary gland masses were found: 192 hemangiomas (59.2%), 89 lymphangiomas (27.5%), and 43 (13.3%) solid masses. No significant difference was found between the age at presentation of the patients with benign solid tumors and the patients with malignant solid tumors (mean + SEM age, 7.2 + 0.7 years). Sixty-one percent of the masses were found in the parotid region; 18% were localized to the submandibular gland region; and the remaining 21% were located in a minor salivary gland site. The most common benign perisalivary masses were pilomatrixomas (20.9%), followed by pleomorphic adenomas (11.6%). The most common malignant masses were mucoepidermoid carcinomas (9.3%), followed by rhabdomyosarcomas (7.0%). Treatment was individualized to the disease. Twenty-two patients had adequate data for follow-up analysis (mean + SEM follow-up, 30.0 + 8.4 months). Four patients (18.2%) experienced recurrent or residual disease and were alive with disease at last follow-up, and 100% of our population demonstrated disease-specific survival at last follow-up. CONCLUSIONS: Vascular lesions outnumber solid tumors of the salivary gland region. The most common salivary tumors were pleomorphic adenomas, followed by mucoepidermoid carcinomas. Although certain solid salivary masses may demonstrate locally aggressive behavior, the overall prognosis is favorable. Arch Otolaryngol Head Neck Surg. 2000;126:1435-1439     

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.     

Metachronous bilateral submandibular gland metastases from carcinoma of the breast

Metastases to the salivary glands from distant neoplasms are unusual, with most reported cases involving the parotid gland. Metastatic deposits in the submandibular gland are extremely rare with bilateral involvement not previously reported. We present the case of a patient with advanced breast carcinoma who has had metachronous involvement of both submandibular glands and review the literature on this subject.     

Absence of Epstein-Barr virus encoded RNA and latent membrane protein (LMP1) in salivary gland neoplasms

A series of 55 (42 benign and 13 malignant) salivary gland tumours were investigated by immunohistochemistry, to detect Epstein-Barr virus (EBV) latent membrane protein (LMP1) and by in situ hybridization for EBV-encoded RNA. Non-neoplastic gland from all the patients with tumours and 15 control glands were also examined. All cases, both neoplastic and non-neoplastic were negative for LMP1 and failed to show any positive signal by in situ hybridization for EBV RNA. One undifferentiated carcinoma from a European patient was included in the group. These results confirm previous reports of an ethnic association between EBV and undifferentiated carcinomas of the salivary gland. They do not support an aetiological role for EBV in other salivary gland tumours.     

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.     

Clinical approach and treatment of benign and malignant parotid masses, personal experience

Parotid gland tumours account for 80% of all salivary gland neoplasms, 20% of these are malignant, but in daily clinical practice most parotid masses are operated on before obtaining the final histological diagnosis. This clinical setting further complicates the critical point of parotid surgery, which is the management of the facial nerve. In the present study, data were evaluated referring to 540 patients who underwent parotidectomy for a mass which was discovered to be a benign (470 cases) or a malignant (70 cases) neoplasm, between November 1994 and December 2007, at our Institution. The most significant single parameter in this series of malignancies regarding disease specific survival was the clinical involvement of the facial nerve at diagnosis (p = 0.006). Also for this reason, as there is no evidence that liberal VIIth nerve sacrifice improves prognosis, when it is not clinically involved, every attempt is made to dissect and preserve it. At present, the most complicated situation concerning nerve preservation may be, on the other hand, recurrence of a benign tumour, in particular pleomorphic adenoma, which, in our series, has a higher incidence (8.3%) of permanent facial dysfunction, than surgery with nerve preservation for malignancy (3.7%).     

Fine-needle aspiration biopsy of salivary gland lesions in a selected patient population

OBJECTIVE: To report the role of selective use of preoperative fine-needle aspiration biopsy (FNAB) in patients with major salivary gland lesions at a tertiary care cancer center. DESIGN: Retrospective review of FNAB results compared with final histologic diagnosis as the criterion standard. SETTING: An academic tertiary care cancer center. PATIENTS: A consecutive series of 258 patients who underwent FNAB of major salivary gland lesions between 1996 and 2000, of whom 169 had surgical resection. MAIN OUTCOME MEASURES: Predictive value, sensitivity, specificity, and accuracy. RESULTS: FNAB was performed in 169 (37%) of 463 salivary gland lesions undergoing surgical procedures. A total of 126 lesions were in the parotid gland and 44 in the submandibular gland. Seventy-nine lesions (46%) were malignant. There were 150 FNAB specimens (89%) that were satisfactory for evaluation. The FNAB diagnosis of malignant or suspicious lesion had positive and negative predictive values of 84% and 77%, respectively. Ten of 20 false-negative FNAB results were low-grade lymphoma on final histologic assessment. Fine-needle aspiration biopsy diagnosis of a benign neoplasm had positive and negative predictive values of 83% and 88%, respectively. A cytopathologic diagnosis of a nonneoplastic lesion was predictive in only 47% of cases. Fifteen (47%) of 32 lymphocyte-predominant FNAB specimens were lymphoma on final histologic assessment. Ten (20%) of 49 patients with history of a solid, non-head and neck malignancy had evidence of distant metastasis to the salivary gland by histologic and/or cytopathologic assessment. CONCLUSIONS: An FNAB diagnosis of malignant or neoplastic major salivary gland disease is generally predictive of final histologic diagnosis. The predictive value of a negative FNAB finding is low, and should not supersede clinical suspicion. Cytologic findings of a lymphocyte-predominant lesion should prompt further workup to rule out lymphoma.     

Large cell neuroendocrine carcinoma of the submandibular gland: Case report and literature review

Large cell neuroendocrine carcinoma (LCNEC) of the salivary gland is extremely rare. We report on a case of LCNEC in the submandibular gland. A 58-year-old male had a four-month history of an enlarging mass in his left submandibular region. He underwent lymph node resection and metastasis of LCNEC was suspected. Magnetic resonance imaging of the neck showed a solid submandibular gland tumor with marginal blurring. Positron-emission tomography and upper gastrointestinal endoscopy showed no evidence of malignancy other than in the left submandibular gland. He underwent left submandibular gland resection and left upper neck dissection. The final diagnosis was LCNEC of the submandibular gland; surgical margin was negative. Fourteen months later he is free of tumors. This is the first report of LCNEC of the submandibular gland. LCNEC of the salivary gland shows high-grade malignancy like that of the lung. According to past reports, two of four patients died despite multidisciplinary treatments. There are no standard treatments for LCNEC of the salivary glands. More studies are needed to define prognostic factors and establish therapeutic methods.     

Malignant epithelial salivary gland tumors in northern Finland: incidence and clinical characteristics

A population-based survey was conducted in Northern Finland to evaluate the incidence rate as well as the patient and tumor characteristics of malignant epithelial salivary gland tumors diagnosed between 1988 and 1998. A total of 40 new cases of malignant epithelial salivary gland tumors were diagnosed and separately evaluated. The age-standardized incidence rates (per 100,000 years) were 0.51 among men and 0.35 among women, giving an overall incidence rate of 0.43. About half of the patients suffered from pain, which was the most common symptom along with palpable resistance, but the duration of symptoms varied widely (median: 11 months). Of all the malignant epithelial salivary gland tumors analyzed in our study, fine-needle aspiration cytology correctly indicated malignancy in only 28% of the cases. Received: 19 February 2001 / Accepted: 27 September 2001     

Neck masses: diagnostic analysis of 630 cases in Turkish population

Neck masses can be classified into three main categories: congenital, inflammatory and neoplastic. Our aim was to determine the distribution of diagnosis in patients who were followed-up for a neck mass and had undergone surgery for diagnostic indications. Six hundred and thirty cases referred to the Otorhinolaryngology and Head Neck Surgery Department of Haseki Research and Training Hospital between January 2005 and February 2012 with a neck mass who underwent excisional or incisional biopsy to establish a histopathologic diagnosis were retrospectively evaluated. Patients with a diagnosis of upper aerodigestive tract malignancy were excluded from the study. As well as the patients with thyroid masses were excluded. Only unknown primary neck masses were included in the study. The neck masses were categorized as inflammatory (33.49 %), congenital (18.9 %) or neoplastic (47.6 %). Neoplastic masses were either benign (51 %) or malignant (49 %) tumors. The most common causes were tuberculous lymphadenitis (40.28 %) among inflammatory masses, thyroglossal duct cysts (32.77 %) among congenital masses, pleomorphic adenoma (22.33 %) among benign neoplastic masses, and lymphoma (20 %) among malignant neoplastic masses. The most common types of mass were congenital in the 0–20 year age group, benign neoplastic in 21–40-year-old and malignant neoplastic in the >40-year group. Any neck mass, especially in an elderly patient, should be managed with caution as a considerable proportion may be malignant. In children and adolescents, a neck mass requiring surgery is most likely to be congenital. Tuberculosis should be considered as a cause of a neck mass due to a long-term inflammatory process in a developing country.