HIV-associated parotid lymphoepithelial cysts

BACKGROUND: An outstanding feature of the diffuse infiltrative lymphocytosis syndrome, or DILS, a subset of HIV-1 disease, is asymptomatic bilateral parotid swelling. Recognition of the entity is important because people with this disease will seek routine dental care. CASE DESCRIPTION: The authors present a classic case of DILS. The patient exhibited bilateral parotid swellings caused by lymphoepithelial cysts, cervical lymphadenopathy, a CD8 circulating lymphocytosis and a CD8 lymphocytic infiltration into the labial salivary glands. A right superficial parotidectomy had been performed several years previously. However, no intervention was advised for the remaining left parotid because of its benign course. CLINICAL IMPLICATIONS: Since patients with DILS can develop lymphomas, periodic observation is mandatory. Any change in the growth pattern requires that a fine-needle aspiration biopsy be performed.     

Histopathological analysis and demonstration of EBV and HIV p-24 antigen but not CMV expression in labial minor salivary glands of HIV patients affected by diffuse infiltrative lymphocytosis syndrome

BACKGROUND: The diffuse infiltrative lymphocytosis syndrome (DILS) in HIV patients is characterized by the persistence of CD8-circulating lymphocytes and lymphocytic infiltration, predominantly in salivary glands. METHODS: We examined seven HIV-positive patients with bilateral parotid enlargement and sicca symptoms. Minor labial salivary gland biopsies were performed in all patients and submitted for histopathological analysis and immunohistochemistry for CD4, CD8, cytomegalovirus (CMV), LMP-EBV protein, and HIV p-24 protein. RESULTS: In all cases, lymphocytic infiltration of the minor salivary glands, mainly periductal, was found. Acinar atrophy, ductal ectasia, and mild to moderate fibrosis were also observed. We noticed strong immunohistochemical reaction for LMP-EBV and p-24 proteins in ductal cells in all cases, while staining for CMV was consistently negative. The lymphocytes were positive for CD8, but consistently negative for CD4. CONCLUSIONS: A role of Epstein-Barr virus (EBV) and HIV, but not CMV, in the pathogenesis of DILS, is suggested by our immunohistochemical findings.     

HIV-associated salivary gland disease: a review.

Human immunodeficiency virus-associated salivary gland disease (HIV-SGD) is defined as the presence of xerostomia and/or swelling of the major salivary glands. It is common among children but uncommon among adults. HIV-SGD includes lymphoepithelial lesions and cysts involving the salivary gland tissue and/or intraglandular lymph nodes, and Sj?gren's syndrome-like conditions, diffuse interstitial lymphocytosis syndrome, and other reported lesions of the major salivary glands. This article reviews the terminology, prevalence, symptoms, clinical features, diagnostic procedures, histopathology, serology, natural history, treatment, and pathogenesis of HIV-SGD.     

Mikulicz syndrome

Mikulicz syndrome is a term used to denote any bilateral chronic enlargement of the lacrimal and salivary glands due to a specific disease process other than Mikulicz disease (benign lymphoepithelial lesion). The purposes of this article are to describe the differential diagnosis of Mikulicz syndrome, illustrate it by a case arising from malignant lymphoma, and make an attempt to place Mikulicz, his syndrome, and his disease in historical perspective, thereby shedding light on the confusion which has surrounded these terms in recent literature.     

Parotid gland swelling in HIV diffuse infiltrative CD8 lymphocytosis syndrome

HIV can affect salivary gland tissue. Parotid swellings have been observed in a subset of patients who are HIV-positive; the swellings are the result of a condition that has been termed diffuse infiltrative CD8 lymphocytosis syndrome. Initially the glandular enlargement results from a massive CD8 cell lymphoproliferation, but with time lymphoepithelial cysts become manifest. Two case reports are presented to illustrate this progression of diffuse infiltrative CD8 lymphocytosis     

EB病毒感染与涎腺良性淋巴上皮病损恶变的关系

目的 了解Epstein-Barr病毒(EBV)感染与涎腺良性淋巴上皮病损恶变的关系。方法 采用针对EB病毒编码的小分子RNA(EBER-1)的寡核苷酸探针对2例涎腺良性淋巴上皮病损灶性恶变、14例恶性淋巴上皮病损(MLEL)、4例良性淋巴上皮病损(BLEL)、4例非特异性慢性涎腺炎石蜡包埋组织进行原位杂交检测;采用聚合酶链反应(PCR)进行EBV BamHI-W片段检测。结果 2例良性淋巴上皮病损灶性恶变形态学上良性区域或恶性区域,EBER-1及EBV BamHI-W片段均为阳性,14例MLEL的癌细胞核均可见EBER-1强阳性信号,其中12例EBV BamHI-W片段阳性,4例BLEL及4例非特异性慢性涎腺炎均为阴性。结论 少数恶性淋巴上皮病损可能在BLEL基础上发生,EB病毒感染与涎腺良性淋巴上皮病损癌变发生可能有密切关系,EBV感染可发生在BLEL癌变形态学改变之前。     

Punch-biopsy of minor salivary glands in the diagnosis of Sjögren's syndrome

Abstract— The aim of the present investigation has been to examine whether the histopathologic changes in the minor salivary glands in patients with Sjögren's syndrome are of the same nature as the changes in the major salivary glands. Punch-biopsies were taken from the lower labial mucosa in 14 patients; 12 of these had a verified Sjögren's syndrome. In 10 patients changes in accordance with those of a benign lymphoepithelial lesion were present in the minor salivary glands. The technique was found valuable in the diagnosis of general disorders with salivary gland involvement.     

涎腺淋巴上皮病变25例报告

作者对２５例涎腺淋巴上皮病变的临床资料和观察随访结果进行了分析。并对本病的名称和性质，Ｍｉｋｕｌｉｃｚ病与Ｓｊｏｅｇｒｅｒ综合征的关系作了讨论，认为涎腺淋巴上皮病变一般为良性过程，但具有肿瘤的特性。临床易误诊，治疗需按涎腺肿瘤的治疗原则。     

大涎腺淋巴上皮病的临床分析

目的:探讨淋巴上皮病(lymphoepitheliallesion,LEL)流行病学特点以提高临床诊断准确率。方法:对19例淋巴上皮病病例的年龄、性别、病理类型、病程、临床和影像学表现、实验室检查结果、诊断情况进行回顾性分析。结果:淋巴上皮病发病年龄多在40～50岁左右,无性别差异。主要表现为局限性肿块或涎腺弥漫性肿大。肿块界清,直径0.6～4.6cm不等,活动度尚可,多数位于耳垂周围,良性者质地中等,恶性者质地较硬。一般无自发痛及面神经功能障碍,少数有轻压痛。无眼干、口干及关节疼痛等症状。淋巴上皮病可累及双侧腺体,绝大多数发生于大涎腺,腮腺最常见,且常呈多病灶。该病患者血清球蛋白往往升高,白球比下降。结论:淋巴上皮病的临床表现有一定规律性,结合影像学表现和实验室检查,可提高诊断率。     

Parotid gland swelling in HIV diffuse infiltrative CD8 lymphocytosis syndrome

The signs and symptoms of diffuse infiltrative CD8 lymphocytosis syndrome (DILS), a subset of HIV, include parotid swelling, cervical lymphadenopathy and a serologic CD8 elevation. A case report is used to illustrate the condition. Patients with the syndrome will be seen in the dental office. Recognition and appropriate referral are responsibilities of the dental practitioner.     

涎腺恶性淋巴上皮病损的免疫组化研究

涎腺恶性淋巴上皮病损（ＭＬＥＬ）是涎腺罕见的恶性肿瘤之一，具有明显的种族特性，好发于南部中国人。本文对１２例ＭＬＥＬ（其中１０例有随访资料）进行Ｋｅｒａｔｉｎ，Ｓ－１００，ＧＦＡＰ，ＣＥＡ免疫组化染色。结果发现Ｋｅｒａｔｉｎ免疫组化染色反应强者预后好，染色反应弱者预后差。以上四种抗体免疫组化染色结果尚提示ＭＬＥＬ的癌细胞具有与正常腺上皮细胞、肌上皮细胞及鳞状上皮细胞相似的抗原特性，提示该瘤来自涎腺导管上皮，常有鳞状上皮化生     

Benign lymphoid hyperplasia of the palate

Seven patients with benign lymphoid hyperplasia of the palate are reported. Clinically, these lesions presented as painless non-ulcerated masses that were unilateral in 6 of the cases and bilateral in one. Microscopically, the lesions were characterized by a mixed lymphoid infiltrate, germinal centres and vascular channels with hyperplasia of endothelial cells. The patients were followed for 3-10 years subsequent to diagnosis. One patient had 2 sequential lesions on opposite sides of the palate. Another patient with lesions of both submandibular salivary glands in addition to the palate developed rheumatoid arthritis, xerophthalmia and serologic changes consistent with systemic autoimmune disease. To date, none of the patients developed signs and symptoms of lymphoma or leukemia. The etiology of these lesions is unknown. Their distinction from lymphoma of the palate, as well as their possible relationship to the benign lymphoepithelial lesion of palatal mucous glands, is discussed.     

Less common oral lesions associated with HIV infection: Prevalence and classification

This paper deals with a number of group II and III lesions, ie lesions definitely but less commonly, and lesions possibly associated with HIV infection, respectively. Salivary gland disease includes dry mouth and/or swelling of major salivary glands, often as a part of CD8-lymphocytosis syndrome. Xerostomia occurs commonly (2–10%) in HIV-infected individuals. Enlargement of the major salivary glands occurs frequently (19%) among HIV-infected children, but rarely among adults (0.8%). The major salivary glands show lymphoepithelial lesions or cysts histopathologically. Hyperpigmentation of the oral mucosa was found in 2.2% of 1710 HIV⁺ individuals in seven studies. The hyperpigmentation has been ascribed to a number of medicaments, and possibly to HIV. The prevalence of pigmentation is not significantly higher among HIV⁺ than HIV^? individuals. Thrombocytopenia frequently occurs in HIV infection. Oral petechiae were reported in 2% of 1121 HIV⁺ in five studies. Human papilloma virus (HPV) infection occurred in 1.1 % of 989 HIV⁺ in seven studies. Drug reactions (white lichenoid lesions, ulceration, toxic epidermal necrolysis) have been reported in a number of cases, not allowing prevalence figures. However certain drugs, notably Foscarnet, Interferon and 2,3-dideoxycytidine, may frequently cause oral ulcerations. Oral neurologic manifestations such as peripheral facial paralysis and sensory neuropathy have been reported in a few cases or series only.     

Benign lymphoid hyperplasia of the palate

Seven patients with benign lymphoid hyperplasia of the palate are reported. Clinically, these lesions presented as painless non-ulcerated masses that were unilateral in 6 of the cases and bilateral in one. Microscopically, the lesions were characterized by a mixed lymphoid infiltrate, germinal centres and vascular channels with hyperplasia of endothelial cells. The patients were followed for 3–10 years subsequent to diagnosis. One patient had 2 sequential lesions on opposite sides of the palate. Another patient with lesions of both submandibular salivary glands in addition to the palate developed rheumatoid arthritis, xerophthalmia and serologic changes consistent with systemic autoimmune disease. To date, none of the patients developed signs and symptoms of lymphoma or leukemia. The etiology of these lesions is unknown. Their distinction from lymphoma of the palate, as well as their possible relationship to the benign lymphoepithelial lesion of palatal mucous glands, is discussed.     

Viruses and salivary gland disease (SGD): lessons from HIV SGD

Viral infections are often associated with salivary gland pathology. Here we review the pathogenesis of HIV-associated salivary gland disease (HIV-SGD), a hallmark of diffuse infiltrative lymphocytosis syndrome. We investigate the presence and contributions of viral diseases to the pathogenesis of salivary gland diseases, particularly HIV-SGD. We have detected BK viral shedding in the saliva of HIV-SGD patients consistent with viral infection and replication, suggesting a role for oral transmission. For further investigation of BKV pathogenesis in salivary glands, an in vitro model of BKV infection is described. Submandibular (HSG) and parotid (HSY) gland salivary cell lines were capable of permissive BKV infection, as determined by BKV gene expression and replication. Analysis of these data collectively suggests the potential for a BKV oral route of transmission and salivary gland pathogenesis within HIV-SGD.     

Minor salivary gland hyalinisation and amyloidosis in low-grade lymphoma of MALT

Two patients with low-grade lymphoma of mucosa-associated lymphoid tissue (MALT) arising in primary Sjögren's syndrome developed solitary nodules in their lips. Histologically both lesions showed enlargement and hyalinisation of single minor salivary glands with acinar atrophy, loss of most ducts and conversion into almost acellular sclerotic eosinophilic masses. In one case the lesion was shown to contain an amyloid component. No amyloid was detected in the second case but deposition of collagen and basement membrane and sclerotic neoplasm were excluded.     

Multiple parotid lymphoepithelial cysts in patients with HIV-infection: report of two cases

OBJECTIVE: Bilateral and multiple lymphoepithelial cysts (LECs) of major salivary glands, in particular of parotid glands, are quite rare and have been reported in human immunodeficiency virus (HIV) infected patients with an incidence of about 3-6%. These lesions represent an early manifestation of HIV infection and are rarely found in patients with advanced acquired immunodeficiency syndrome. MATERIALS: Two cases of parotid LECs, the first occurring in a middle-age white woman and the second in a young white boy, both in advanced phases of HIV infection, are reported. RESULTS: Clinical, cytological, histological and immunohistochemical (cytokeratin AE1/AE3, CD20, CD45RA, CD8, kappa and lambda immunoglobulin light chains, S-100, MLA and Ki67) features are described. CONCLUSIONS: Fine needle aspiration (FNA), a relatively non-traumatic procedure, could represent both a diagnostic and a therapeutic tool in parotid LECs. No surgical therapy is usually required for these lesions and aspiration of cystic fluid with FNA is quite resolutive, although evidence of further relapses does exist. Surgical excision may become necessary when pain, because of persistent and progressive swelling of the parotid gland, occurs.     

Benign parotid lymphoepithelial cysts associated with HIV infection

HIV infections are often related with lymphoepithelial benign cysts of the parotid gland; these lesions are uncommon and other salivary glands are rarely involved. The diagnosis, often difficult, is based on imaging techniques, like computed tomography, and pathologic techniques, like fine needle aspiration biopsy. The therapeutic approach can be performed by simple aspiration, surgical resection, pharmacotherapy, or radiotherapy. Three cases of HIV-related lymphoepithelial benign cysts of the parotid gland observed at the Department of Maxillofacial Surgery of the "Federico II" University of Naples and at the Department of Oral and Maxillofacial Surgery of "La Paz" Hospital of the Universitad Autonoma of Madrid are described.     

涎腺恶性淋巴上皮病损的临床病理研究

涎腺恶性淋巴上皮病损是涎腺罕见的恶性肿瘤之一，具有明显的种族特性。本文对２７例恶性淋巴上皮病损进行了临床病理学分析和随访调查，探讨肿瘤生物学行为有关的病理因素，发现间质中纤维结缔组织多，玻璃样变明显者预后较差，本文还着重对ＭＬＥＬ的命名、诊断、鉴别诊断、病理组织学与预后的关系进行了讨论。