Malignant lymphoepithelial lesion of the parotid gland: a case report and review of the literature

Malignant lymphoepithelial lesions are rare tumors of the major salivary glands. They most often occur in Asians and Greenland Eskimos and are strongly associated with Epstein-Barr virus infection. We report a case of a malignant lymphoepithelial lesion of the parotid gland that developed in an Italian-American woman whose serology was positive for Epstein-Barr virus antibody. The patient underwent a left total parotidectomy and upper neck dissection, followed by radiation therapy. At the 2-year follow-up, she remained free of disease.     

Malignant lymphoma of the parotid gland

A case of malignant lymphoma of the parotid gland was described. A 15-year-old male developed a mass which was a firm and non-tender. The patient underwent a superficial parotidectomy with preservation of the facial nerve. The tumor was a grey, soft mass, 3 X 4 X 4 cm in size. On microscopic examination, the tumor was intraglandular lymph node with invasion of salivary gland tissue. The tumor cells were seen between acini and ducts. A diagnosis of lymphosarcoma, prolymphocytic, nodular type was made according to the WHO's classification system and the patient was refered for radiation therapy. Further investigations, including radio-isotope scanning of liver, spleen and bone and a biopsy of bone marrow revealed no sign of the disease. The patient was treated with 5380R of cobalt irradiation and in follow-up of a one year's duration there was no recurrence. Our case fulfilled the criteria which was represented by Hyman and Wolff and we reviewed the literature of this condition.     

Management of benign lymphoepithelial lesions of the parotid gland in human immunodeficiency virus-positive patients

The diagnosis and radiotherapeutic treatment of these HIV-associated benign lympepithelilial lesions in the parotid gland are discussed. As an example of these lesions, a case is presented involving a 43-year-old HIV-infected man with a 2-year history of enlargements in both parotid glands. After evaluation by computer tomography and cytology, the diagnosis of benign lymphoepithelial lesions was made. Treatment by low-dose radiotherapy (15 Gy) caused regression of both lesions. The lesion on the left regressed completely, but the one on the right side responded only partially . A second course of high-dose radiotherapy (24 Gy) to the right lesion caused regression to a cosmetically acceptable size. Received: 20 March 1998 / Accepted: 1 April 1998     

Malignancy and the benign lymphoepithelial lesion.

The benign lymphoepithelial lesion of salivary glands is now considered the histological hallmark of a variety of clinical and pathological disorders affecting salivary tissues. Malignancy arising in the lesion is uncommon, but may take origin in either the epithelial or lymphoreticular components. Lymphomas and pseudolymphomas associated with salivary gland lymphoepithelial lesions have been predominately extra-salivary and strongly correlated with Sjogren's syndrome. Epithelial malignancy has not been associated with autoimmunity and with few exceptions has been of the anaplastic type. This report presents two patients with intra-salivary lymphomas arising in a benign lymphoepithelial lesion of salivary glands and a patient with anaplastic carcinoma arising in the epithelial islands of the lesion. The fourth patient manifested pseudolymphomatous lymphoreticular hyperplasia in lung and submandibular gland and illustrates the possible multiple organ involvement that may occur in patients with benign lymphoepithelial lesion, even without clinical evidence of concommitant autoimmune disorders.     

涎腺淋巴上皮病诊断和治疗

目的探讨涎腺淋巴上皮病的诊断和治疗方法。方法回顾收治的16例涎腺淋巴上皮病患者资料,分析发病特点、临床表现、常规体检和影像检查,所有病例均行手术治疗,发生于腮腺者行腮腺浅叶及肿瘤切除5例,肿瘤加腮腺浅叶深叶部分切除4例。发生于颌下腺和舌下腺者均行全部腺体切除。结果均以腺体肿大或腺体内发现肿瘤为主要症状。术前误诊为混合瘤者12例（75％）。术后病理诊断良性淋巴上皮病14例,淋巴上皮癌2例。所有病例均行手术治疗,2例淋巴上皮癌术后行补充放射治疗。术后随访6个月～15年,无1例复发。结论涎腺淋巴上皮病缺乏临床特征性表现,容易误诊。手术治疗是本病主要治疗方法。     

The benign lymphoepithelial cyst and a classification system for lymphocytic parotid gland enlargement in the pediatric HIV population

OBJECTIVES/HYPOTHESIS: The objectives of this study are to present a series of parotid gland benign lymphoepithelial cysts (BLEC) in HIV-positive children and to propose a three-tiered classification system for HIV-associated lymphocytic parotid gland enlargement. STUDY DESIGN: The authors conducted a retrospective case series and literature review. METHODS: The authors conducted a retrospective chart review of four pediatric patients with HIV-associated parotid gland BLEC who presented to a tertiary care university medical center. RESULTS: Four pediatric HIV-positive patients (four girls; age range, 7-17 years [mean age, 12.8 years]) were diagnosed with parotid gland BLEC. Two patients presented with acute parotitis and the others presented with asymptomatic enlargement of the parotid glands. Three patients had bilateral parotid gland BLEC. The other patient demonstrated persistent generalized lymphadenopathy (PGL) of the intraparotid and cervical lymph nodes and early BLEC limited to the left parotid gland. One patient also displayed parotid gland microcalcifications and cystic changes in the adenoids, neither of which have been described previously in the setting of HIV-associated BLEC. Computed tomography was performed on all patients, and one patient underwent fine needle aspiration to confirm the diagnosis. All patients opted for observation and antiretroviral medication therapy as long-term treatment. Based on these findings and a review of the literature, we propose a three-tiered classification system for lymphocytic parotid gland enlargement in the HIV population: 1) PGL, 2) benign lymphoepithelial lesions (BLEL), and 3) BLEC. CONCLUSIONS: This series equals the largest pediatric series of HIV-associated parotid gland BLEC in the English literature. One patient in our series also demonstrated PGL; there were no cases of BLEL. A classification system based on morphology is proposed to help resolve the confusion in terminology used to describe this entity. Most pediatric HIV-infected patients with parotid gland BLEC can be treated with observation and antiretroviral medication therapy. For others, who are symptomatic or more concerned about their cosmetic appearance, sclerotherapy may offer a reasonable option. Radiation therapy and surgery should be reserved for select cases.     

A case report of lymphoepithelial cyst in the parotid gland]

A 57 year-old Japanese male was seen with the chief complaints of left parotid gland swelling, pain, trismus. At the initial visit, a subcutaneous mass, measuring 50 x 70mm, was seen in the left parotid region. The mass was moderately tender and the consistency was soft anteriorly and elastic firm posteriorly. The mobility was restricted with no change in the skin covering the mass. The saliva from the left parotid gland was normal in colour and contents. Various imaging modalities were done preoperatively including CT, MRI, ultrasonography and aspiration biopsy under the ultrasonographic guide. None was thus conclusive for the differential diagnosis and our tentative diagnosis was a cystic lesion in the parotid gland. A partial parotidectomy was performed under general anesthesia and the cystic lesion, measuring 20mm in diameter, was found in the deep lobe. The cyst showed no connection to the external ear canal, and tracts and fistulae were absent. Histopathological report lead to the definite diagnosis of lymphoepithelial cyst. A mild and incomplete facial palsy on the left had been noted for more than 10 years and this, however, improved after the surgery.     

Malignant tumors of the parotid gland

We review patients operated of parotidectomy in our ENT department of Dr. Peset Hospital from Valencia during the period 1975-2001. We sheltered an amount of 147 cases. The pathologic distribution between benign and malign tumors was as following: 121 benign tumors (83%) and 26 malign tumors (17%). Here we study malign parotid tumors, which represent 17% of total parotid tumors. Sensibility and specificity of FNA was 54% and 96.5% respectively. We performed almost with similar frequencies total and suprafacial parotidectomies in 46% and 42% of cases respectively. Total parotidectomy with facial resection is indicated in cases with tumor nerve infiltration and has been done in 8% of cases. The main complications of surgery were facial nerve dysfunction and Frey syndrome. Permanent facial nerve dysfunction and and transitory facial nerve disfunction occurred in 3.8% and 30.8% of patients respectively. Frey syndrome appeared in 10% of patients operated of parotidectomy. Recurrences occurred in 15.4% of patients.     

Role of cytomegalovirus, Epstein-Barr virus, and human herpes virus-8 in benign lymphoepithelial cysts of the parotid gland

OBJECTIVE: To provide background and evaluate the role of herpesviruses in benign lymphoepithelial cysts (BLC) of the parotid gland. STUDY DESIGN: Case series derived from review of pathology specimens. METHODS: Radiolabeled polymerase chain reaction (PCR) analysis was used to detect for the presence of cytomegalovirus (CMV), Epstein-Barr virus (EBV), and human herpes virus 8 (HHV-8) DNA sequences in 14 paraffin embedded specimens and 1 freshly aspirated BLC specimen. Thirteen normal parotid tissue specimens obtained from paraffin embedded blocks were used as a control group. RESULTS: CMV was detected with nearly equal frequency between the two groups (23% of normal vs. 20% in BLC). HHV-8 was found in 13% of the BLC group and in none of the normal group (P =.4841). There was significant difference in EBV detection between the normal (0%) and the BLC (33%) groups (P =.0437). CONCLUSION: CMV and HHV-8 does not appear to be associated with BLCs. Although EBV is found more frequently in BLC than in normal parotid controls, further studies are needed to elucidate the role of this virus in BLC pathogenesis.     

Non-Hodgkin's lymphoma of the parotid gland

Since 1969 we have seen 17 patients with non-Hodgkin's lymphoma of the parotid gland. Retrospective analysis of these patients reveals that the primary diagnosis was established in 14 patients by parotid surgery. In 10 (58.8%) cases there was a single focus of NHL in the parotid gland proven by extensive medical and radiological examinations. Histology showed that 14 (82.3%) of the tumors were low grade and 3 (17.7%) were high grade (Kiel classification). In 5 specimens myoepithelial sialadenitis was found in the parotid gland tissue. Six patients died within 1.5 years on average, 4 of them without evidence of disease. Four of the 7 patients alive have been followed for an average of 3.1 years without evidence of disease. Primary radiation of the parotid region in stage IE in cases of parotid gland NHL seems to be an effective therapy. NHL presenting in myoepithelial sialadenitis seems to be rare, but offers the opportunity of observing an organ specific malignant process.     

Malignant fibrous histiocytoma of the parotid gland

Primary malignant fibrous histiocytoma (MFH) arising in a major salivary gland is rare. We encountered a case of MFH affecting the parotid gland. The patient was a 54-year-old man diagnosed as having pleomorphic type of MFH after extended total parotidectomy. Examination of the resected specimen revealed the tumor had not been completely removed. Accordingly treatment consisted in the resection of MFH and radiotherapy in combination with chemotherapy using carboplatin (CBDCA). This postoperative therapy was effective in controlling the growth of the remaining tumor tissue. As the patient showed no signs of local recurrence and distant metastasis for 5 years, plastic surgery was performed to improve the serious deformation of the face with a free anterolateral thigh flap. Our literature review yielded 17 cases of parotid MFH and the 2-year survival rate was 33%.     

Cutaneous mucormycosis of the head and neck with parotid gland involvement: first report of a case

Cutaneous mucormycosis is an uncommon, life-threatening, opportunistic fungal infection that is a distinctly different entity from the more frequent although still uncommon rhinocerebral form that is better known to otolaryngologists. We describe what to our knowledge is the only reported case of cutaneous mucormycosis of the face with parotid gland involvement, which occurred in a 56-year-old man. The diagnosis was established by tissue biopsy. The patient was treated with antifungal medications and wide local debridement, including a total parotidectomy with sacrifice of the inferior division of the facial nerve. At the 2-year follow-up, he remained free of disease. Familiarity with the risk factors associated with the development of cutaneous mucormycosis is critical to determining the need for early tissue biopsy to confirm the diagnosis. Generally favorable clinical outcomes are associated with prompt and aggressive medical and surgical therapy.