Benign Lymphoepithelial Lesion Research Articles

Utility of fine needle aspiration in HIV-positive patients with corresponding CD4 counts. Four years' experience in a large inner city hospital.

To assess the utility of fine needle aspiration (FNA) in human immunodeficiency virus (HIV)-positive patients with corresponding CD4 count analysis. The study group consisted of 108 FNA specimens from 80 patients performed from January 1991 to December 1994. FNAs were sub-typed into four categories: lymph nodes (59 specimens), masses (26 specimens), salivary glands (19 specimens) and breast (4 specimens). Thirty-three were diagnosed as reactive lymph nodes, 15% as benign lymphoepithelial lesions and 9% as granulomas; 9% revealed acute inflammation, 5% were positive for malignant lymphoma, and 3% were suspicious for Kaposi's sarcoma. Differences in CD4 counts were statistically significant for specific subsets of HIV-related illnesses. General trends were noted in mean CD4 counts for specific sub-groups of HIV-related illnesses. Material adequate for culture can be obtained with the FNA procedure. FNA in skilled hands is a very useful simple and cost-effective procedure for the diagnosis of HIV-related lesions and in the management of these patients. This study suggested that mean CD4 counts are statistically significant in specific subsets of HIV related illnesses.

Epstein‐Barr virus‐associated undifferentiated carcinoma with lymphoid stroma of the salivary gland in Japanese patients: Comparison with benign lymphoepithelial lesion

Epstein‐Barr virus‐associated undifferentiated carcinoma with lymphoid stroma of the salivary gland in Japanese patients: Comparison with benign lymphoepithelial lesion

Lymphoid infiltrates of the salivary glands

Lymphoid infiltrates of the salivary glands are common to a variety of pathologic conditions including autoimmune disorders, malignant lymphomas, and immunoregulatory responses to parenchymal neoplasms. Clearly, the correct identification of these salivary gland lymphoid infiltrates has important implications regarding patient prognosis and management. Immunophenotypic and molecular analyses have demonstrated that many lesions formerly regarded as myoepithelial sialadentis or benign lymphoepithelial lesion in fact represent neoplastic lymphoid proliferations with the potential for extrasalivary dissemination. In the most recent classification scheme of non-Hodgkin's lymphomas, these neoplasms fall within the spectrum of low-grade B-cell lymphoma of mucosa-associated lymphoid tissue. In the early stages of HIV infection, patients may develop salivary gland enlargement resulting from cystic lymphoepithelial lesions. These lesions are thought to reflect a localized manifestation of persistent generalized lymphadenopathy. Although HIV-associated salivary gland disease is regarded as a benign condition, malignant lymphoma has been described in association with some of these lesions, and further work is required to define more precisely the risk of salivary gland lymphoma in HIV-infected patients. Tumor-associated lymphoid proliferation refers to a prominent lymphoid reaction accompanying certain epithelial tumors of the salivary glands. Although tumor-associated lymphoid proliferation has not received as much attention as other types of salivary lymphoid infiltrates, it is a common phenomenon that is sometimes mistaken for an intraparotid lymph node harboring metastatic carcinoma.

耳下腺の悪性リンパ上皮性病変例

A 78-year-old female was admitted because of a painless mass in the right parotid gland of 3 month's duration. On September 14, 1994, the mass was excised, and she was additionally treated with irradiation. There was no evidence of recurrence until November, 1995.Malignant lymphoepithelial lesion (MLEL) is a rare tumor with a relatively high incidence in Eskimos. Epstein-Barr virus (EBV) may play a role in the etiology of MLEL. Our patient's serum contained EBY antigen (EBNA). There is controversy about the origin of MLEL of the salivary glands and its relation to benign lymphoepithelial lesions.Microscopically, the tumor was seen to be growing in diffuse sheets with many areas of lymphocytic infiltration and considerable fibrous stroma. Mitoses were numerous, and the nucleoli were prominent, so it appeared to be MLEL-an undifferentiated or poorly differentiated squamous cell carcinoma associated with abundant lymphocytes. Benign lymphoepithelial lesions have been reported in the literature as preceding or co-existing with malignant lesions in some patients. Our patient also had a benign lymphoepithelial lesion with smaller nuclei and fine chromatin. The pathological findings may support the above-mentioned theory of the etiology of MLEL. Persistent EBV infection might have induced a pronounced lymphoid reaction against parotid duct tissue leading to a BLEL. Finally malignant changes of the ducts might have arisen from the hyperplastic, squamous metaplastic and dysplastic epithelium.

2171 Low dose radiation in the treatment of HIV related benign lymphoepithelial lesions of the parotid gland

2171 Low dose radiation in the treatment of HIV related benign lymphoepithelial lesions of the parotid gland

Benign Lymphoepithelial Parotid Lesions in Hiv-Positive Patients: Spectrum of Findings at Grayscale and Doppler Sonography

Benign Lymphoepithelial Parotid Lesions in Hiv-Positive Patients: Spectrum of Findings at Grayscale and Doppler Sonography

Lymphoepithelial carcinoma of the salivary gland: in situ detection of Epstein-Barr virus.

To examine the role of Epstein-Barr virus (EBV) in lymphoepithelial carcinoma of the salivary gland in Hong Kong Chinese. Ten cases of lymphoepithelial carcinoma of the salivary gland (eight parotid and two submandibular) were examined. In situ hybridisation was used to localise EBER RNA, immunohistochemical methods to detect expression of latent membrane protein 1 (LMP-1) in EBV positive tumours, and Southern blot analysis to examine the clonality of EBV in the two cases where frozen tissue was available. None of the cases had a history of Sjögren's syndrome or histological evidence of a benign lymphoepithelial lesion. The IgA antibody titre against EBV viral capsid antigen was elevated in four cases. All cases were EBV positive by in situ hybridisation, with a strong uniform positive signal in the epithelial cells, and all cases expressed LMP-1. Southern blot analysis revealed that the clonal episomal form of the virus was present. Two of the three female patients in this series also developed carcinoma of cervix. One of these carcinomas had histological features of a lymphoepithelioma-like carcinoma but was EBV negative. A consistent association between EBV and lymphoepithelial carcinoma of the salivary gland was found. The presence of the virus in a clonal episomal form, and the expression of LMP-1 viral oncoprotein is further evidence of the role of EBV in the oncogenesis of this tumour.

Benign lymphoepithelial parotid lesions in HIV-positive patients: spectrum of findings at gray-scale and Doppler sonography.

Benign lymphoepithelial lesions are intraparotid nodules that develop as an early manifestation of HIV infection. The purpose of this study was to describe the spectrum of sonographic and Doppler findings of benign lymphoepithelial lesions in HIV-positive patients and to assess whether these findings are specific for the diagnosis of such lesions. Nine HIV-positive patients with swelling of the parotid gland caused by single or multiple parotid nodules were evaluated. Fourteen nodules were identified sonographically and proved to be benign lymphoepithelial lesions at cytologic examination. They were assessed with gray-scale, duplex, color Doppler, and power Doppler sonography. For each nodule, the echogenicity, grade and pattern of vascularity, and spectral tracings were evaluated. Benign lymphoepithelial lesions manifested as parotid nodules with a wide spectrum of sonographic appearances, ranging from simple cysts to mixed masses with predominantly solid components. Cystic lesions (10/14) were not purely anechoic; they had an internal network of thin septa supplied by vessel pedicles and, in four of 10 cases, a solid mural nodule. On the other hand, mixed nodules (4/14) had a higher conspicuity of solid tissue and thicker stromal septa, thus resembling a parotid neoplasm more than a cyst. The overall grade of vascularity of benign lymphoepithelial lesions varied from avascular to intensely hypervascular. Generally, mixed nodules were more hypervascular than were the cystlike ones. Spectral analysis showed slow velocity (peak systolic velocity range, 8-28 cm/sec; mean, 20 20 cm/sec) and low impedance (resistive index range, 0.36-0.70; mean, 0.57) arterial flows. The spectrum of sonographic and Doppler features of benign lymphoepithelial lesions in HIV-positive patients is broad. Sonographic findings are non-specific for the diagnosis of such lesions, especially in cases of mixed solid-cystic isolated nodules.

Nodal inclusion cysts of the parotid gland and parapharyngeal space: A discussion of lymphoepithelial, aids‐related parotid, and branchial cysts, cystic warthin's tumors, and cysts in sjogren's syndrome

The purpose of this report is to examine the computed tomography scans, magnetic resonance images, and pathologic findings in 44 patients, 42 of whom had inclusion-type cysts of the parotid and parapharyngeal space of varying etiologies. Two additional cases of cystic changes in the benign lymphoepithelial lesion (BLEL) of Sjögren's syndrome are highlighted here, since they had unusually large cystic components mimicking acquired immunodeficiency syndrome-related parotid cysts (ARPCs). A retrospective examination identified 18 ARPCs, 3 lymphoepithelial cysts (LECs), 13 cystic Warthin's tumors, 8 branchial cysts, and 2 cases of cysts in patients with Sjögren's syndrome (BLEL), all of whom had imaging studies and pathologic confirmation. There were 30 men and 14 women with an age range of 25 to 72 years (median, 46.82 years). Any similarities in the imaging appearances were noted, as were any differences in pathologic detail. On imaging, only the cystic Warthin's tumors had any focal wall nodularity; the other cysts had smooth walls. When multiple parotid cysts were present, the distinguishing feature between ARPCs and cysts in BLEL (and some cystic Warthin's tumors) was the presence of diffuse cervical adenopathy in patients with ARPCs. Imaging usually could not differentiate between a solitary parotid LEC, a branchial cyst, and some cystic Warthin's tumors. Extraparotid lesions were either branchial cysts or cystic Warthin's tumors. Physicians should be aware of the variety of different inclusion-type cysts that may occur in the parotid gland and parapharyngeal space, all of which may have similar imaging appearances. Although imaging clearly identifies these cysts and may suggest a specific diagnosis, it must always be remembered that the precise diagnosis remains in the province of the pathologist.

Ultrastructure and three-dimensional imaging of epimyoepithelial islands in benign lymphoepithelial lesions.

Benign lymphoepithelial lesions of the salivary glands associated with Sjögren's syndrome are characterized by extensive infiltration of lymphoid cells, atrophy of acini and the presence of so-called epimyoepithelial islands. This report describes ultrastructural and three-dimensional reconstructive studies of epimyoepithelial islands performed at Tokyo Women's Medical College. Ultrastructural examination showed that these islands are composed mainly of epithelial cells containing intermediate filaments and/or tonofilament bundles, scattered lymphocytes and plasma cells. Myoepithelium-like cells containing myofilaments were sometimes found in the peripheral portion of the myoepithelial islands. Also, mitotic figures were rarely found in these islands. Three-dimensional reconstructive study revealed that the epimyoepithelial islands are not isolated cell clusters but are continuous hypertrophic duct-like structures. These results suggest that the epimyoepithelial islands are derived from proliferating duct epithelial cells, especially those of large peripheral ducts.

両側耳下腺ＭＡＬＴ型リンパ腫の異時性発生例

It has been reported that lymphoepithelial lesions (LEL), including Sjögren syndrome and benign lymphoepithelial lesions (BLEL), often change to low-grade malignant B-cell mucosa associated lymphoid tissue (MALT) lymphomas. In the patient described here BLEL changed to malignant lymphoma in both parotid glands over a 2-year interval. Surgical removal was performed because this malignant lymphoma generally resists to both chemotherapy and radiation therapies, and the prognosis is poor when the lymphoma extends outside the gland.

良性リンパ上皮性病変の２症例 腺上皮におけるサイトケラチン発現の検討

Two cases of benign lymphoepithelial lesions (BLEL) are reported along with a discussion on the expression of cytokeratin.Case 1: A 46-year-old man was referred to our clinic with a chief complaint of swelling at the right submandibular gland. Computed tomography showed the submandibular gland as a homogeneous high density area. Gallium scintigraphy demonstrated a clear hot spot at the same region. The submandibular mass was extirpated extraorally. Pathologically, there were atrophic changes of the ducts and acini, acompanied by severe lymphoid cell infiltration.Case 2: A 55-year-old man complained of swelling of the right buccal region. Magnetic resonance imaging revealed a homogeneous mass showing low intensity in T1 weighted SE and slightly high intensity in T2 weighted SE at the right lower parotid region. Examination of a specimen of the extraorally excised mass showed severe lymphoid tissue infiltration interspersed with myoepithelial islands in the atrophic parotid gland.Immunohistochemical staining was performed to check for the expression of the cytokeratin in the atrophic salivary gland. All duct-like structures (DLSs) reacted positively against K8.13 and cytokeratin #7 and #18. On the other hand, # 8 was negative especially in small DLSs.

Low-dose radiotherapy for multicystic benign lymphoepithelial lesions of the parotid gland in HIV-positive patients: long-term results.

Multicystic benign lymphoepithelial lesions of the parotid gland (BLL) seen in patients with human immunodeficiency virus (HIV) can produce considerable cosmetic deformity as well as physical discomfort. We previously reported our preliminary results with low-dose radiotherapy in this disease, and all 8 patients were satisfied with the initial improvement in their appearance. We now report the long-term follow-up of those patients and additional patients. Twelve HIV-positive patients with BLL were treated with 8-10 Gy of external radiation using 2-Gy daily fractions. Objective responses and subjective duration of patient-defined cosmetic control were recorded. All 12 patients (100%) had at least a 50% decrease in the size of their parotid masses. Five of 12 (42%) had a complete response and 7 (58%) had a partial response. Persisting complete response was achieved in only 1 patient, however, with relapse in the other 11 patients. Cosmetic palliation, as judged by the patients, was achieved for a median of 9.5 months. Eight patients were subsequently retreated with doses of 6-16 Gy (median and mode: 10 Gy). None (0%) of the 8 patients retreated achieved local control. Very low-dose radiation (8-10 Gy) provides reliable but temporary cosmetic palliation for BLL. Retreatment was unsatisfactory, and we are now investigating higher initial doses of radiation to prolong palliation and eliminate recurrences.

Atypical Periorbital Xanthogranulomas Associated with Systemic Benign Lymphoepithelial Lesions

The report reviews the clinical, laboratory, and pathologic features of a 47-year-old woman with systemic benign lymphoepithelial lesions in whom atypical periorbital xanthogranulomas with rare central necrosis subsequently developed.

Benign lymphoepithelial lesion (Mikulicz's syndrome) of the submandibular glands in a four-year-old boy

Benign lymphoepithelial lesion (Mikulicz's syndrome) of the submandibular glands in a four-year-old boy

Benign Lymphoepithelial Lesion (Mikulicz's Syndrome) of the Submandibular Glands in a Four‐Year‐Old Boy

Benign Lymphoepithelial Lesion (Mikulicz's Syndrome) of the Submandibular Glands in a Four‐Year‐Old Boy

悪性腫ようが疑われた耳下腺リンパ上皮性病変

Two atypical benign lymphoepithelial lesions in the parotid gland and cervical lymphadenopathy are reported. The nonhomogeneity of the tumor echogram and the swollen neck lymph nodes strongly suggested malignancy of the parotid gland. Pathological findings of the resected parotid lesions revealed atrophy of the glandular parenchyma and epimyoepithelial islands, diagnosed as “benign lymphoepithelial lesion”. The neck lymph nodes in case 1 were reactive hyperplastic lesions and in case 2 granulomatous and/or tuberculous lymphadenitis. No lymphoepithelial lesion was found in the neck lymph nodes. The relationship between the original parotid lesion and neck lymphadenopathy was not clear. Benign lymphoepithelial lesion could be approvable entity in some parotid masses even when accompanied by cervical lymphadenopathy.

耳下腺良性リンパ上皮性疾患例

Benign lymphoepithelioma is a comparatively rare disease which shows sialaden tissue findings which are pathohistologically similar to those seen in Sjögren's syndrome. We recently treated 27-year-old female with a benign lymphoepithelioma in the parotid gland. She had no signs or symptoms other than a tumor in the right parotid gland. There was no xerostomia. A definite diagnosis was reached from the pathohistological appearance of resected tissue. Since benign lymphoepithelial lesions may become malignant lymphomas, this patient must be followed carefully.

良性リンパ上皮性疾患の２例

We report two cases of benign lymphoepithelial lesions (BLL) of the salivary gland. BLL arose in the sublingual gland in case 1 and in the palatine gland in case 2. Neither patient complainted of xerostomia or xerophthalmia, and other salivary glands were normal.In both cases histopathologic findings of lymphocytic infiltration, ectatic ducts, myoepithelial islands and acinar atrophy were observed. Immunohistological examination revealed that the predominat population of lymphocytes around the ducts and myoepithelial islands consisted of B-cells rather than T-cells.

Benign lymphoepithelial lesions of the parotid

Persistent generalized lymphadenopathy has been well described in patients with seropositivity to the human immunodeficiency virus (HIV). Moreover, isolated enlargement of the parotid gland and parotid lymphadenopathy have been noted much more frequently over the past few years. Histologically, these lesions demonstrate follicular hyperplasia, cystic dilatation of the ducts lined by pseudo-stratified squamous epithelium, and lymphocytic infiltrates. They are generally considered to be benign lymphoepithelial lesions of the parotid or hyperplastic periparotid lymph nodes. The relationship of this entity to the AIDS-related complex (ARC) and the subsequent development of AIDS is not clear. Over the past 7 years, we have seen 50 patients with parotid enlargement in whom the diagnosis of benign lymphoepithelial lesion was made. Fine-needle aspiration was performed in 32 patients. Although not conclusively diagnostic, needle aspirates ruled out primary salivary glandular pathology. Most patients gave a history of intravenous drug abuse. HIV tests have been performed on a routine basis only in the last 2 years, and these were positive in the majority of the patients. Thirty-five patients underwent surgical excision. In the initial 20 patients, we routinely performed parotid exploration, identification of the facial nerve, and superficial parotidectomy. In the last 15 patients, we changed our surgical approach to parotid exploration and excision of the mass in the tail of the parotid. The exposure of the posterior belly of the digastric muscle, with identification and removal of the deep jugular node, has become routine. In each case, we found an enlarged lymph node in the deep jugular region, which was not clinically palpable preoperatively. The rate of surgical complications was minimal, and, after resection of the mass, patients improved symptomatically. If the patient shows obvious signs of AIDS, a nonsurgical approach with repeated aspirations should be considered, and treatment with zidovudine offered.