Surgical management of Warthin tumor: long-term follow-up of 224 patients from 2002 to 2018.

PURPOSE: Warthin tumors (WT) are the second most common benign parotid gland neoplasms. They can occur as synchronous or metachronous lesions in 6-10% of cases. This study aims to compare the complication rate in 224 patients who underwent extracapsular dissection (ECD) or superficial parotidectomy (SP) for the treatment of a WT. METHODS: This retrospective study was conducted at the Department of Maxillo-Facial Surgery at the University of Naples "Federico II" from February 2002 to December 2018 on a group of patients who underwent surgical treatment for WT. The type of surgical technique was chosen based on Quer's classification. The complications evaluated were facial nerve palsy, hematoma, Frey's syndrome, and bleeding. RESULTS: A total of 224 patients treated from 2002 to 2018 for Warthin tumor were included in the study. Two hundred elven had solitary tumors (94.1%) and 13 had multicentric lesions (5.8%), of which 9 cases presented synchronous lesions and 4 cases presented metachronous lesions. Extracapsular dissection (ECD) was performed in 130 patients (58.3% of cases) and superficial parotidectomy (SP) in the other 94 (41.7% of cases). CONCLUSIONS: We consider both surgical techniques as valid. In our opinion, it is essential to study each case based on Quer's Classification to obtain the best surgical outcome. Based on a lower observed rate of complications such as facial nerve palsy, Frey's syndrome, and bleeding, ECD seems to be the best option for the surgical treatment of Quer Class I lesions.

Uncommon Coexistence of Pleomorphic Adenoma and Warthin's Tumor in a Painfully Swollen Left Parotid Gland: A Surgical Case Report.

BACKGROUND Benign pleomorphic adenoma is the most common primary tumor of the salivary glands and mainly arises in the parotid gland. Warthin's tumor, or papillary cystadenoma lymphomatosum, represents <30% of benign parotid tumors. The simultaneous occurrence of multiple parotid tumors is rarely described - depending on the corresponding histology (different/identical), the time of their occurrence (synchronous/metachronous), as well as their location (unilateral/bilateral), multiple parotid tumors can be further sub-classified. CASE REPORT We describe the case of a 54-year-old female patient with progressive and painful swelling of the left parotid gland for the last 6 months. During extra-oral examination, a bulging, displaceable mass of approximately 3 cm was determined. A subsequent MRI (magnetic resonance imaging) examination revealed a multifocal lesion but failed to provide a decisive clue as to the tumor entity of the lesion, and a lateral (superficial) parotidectomy was performed. Postoperative histomorphological interpretation allowed the final pathological diagnosis of synchronous, unilateral occurrence of a pleomorphic adenoma as well as a Warthin's tumor. CONCLUSIONS This report presents a rare case of synchronous unilateral parotid tumors and supports that benign pleomorphic adenoma and Warthin's tumor are the most common associations. Since clinical examination, MRI imaging, and even cytological assessment could be misleading in the detection of synchronous ipsilateral multiple parotid gland tumors, our report also highlights the importance of timely and accurate diagnosis with histopathology to plan surgery and to exclude malignant transformation, which is a rare but important association with both types of primary salivary gland tumor.

Warthin tumor complicated with T-lymphoblastic lymphoma: a case report.

Warthin tumor is a benign salivary gland tumor comprising ductal epithelium and lymphoid stroma. To date, reports about the malignant transformations of intraepithelial and lymphoid components in Warthin tumor are extremely rare; lymphoid malignant transformation into B-cell lymphoma is particularly rare in combination with T-cell lymphoma. The case of Warthin tumor complicated with T-lymphoblastic lymphoma is reported to emphasize the importance of a careful light microscopic evaluation of lymphoid tissue in Warthin tumor for identifying occult lymphoma presence, reducing misdiagnosis and missed diagnosis, and determining a timely treatment.

Xanthogranulomatous Sialadenitis, an Uncommon Reactive Change is Often Associated with Warthin's Tumor.

Xanthogranulomatous sialadenitis (XGS) is rare in salivary glands and only reported in the literature as single cases. Here we report a cohort of four cases with XGS and summarize the clinicopathologic features of these cases. All four patients had persistent mass lesions concerning for neoplasm. In two patients (patient 1 and 3), the initial fine needle aspirations (FNAs) contained oncocytic cells consistent with or suspicious for Warthin's tumor, but follow-up FNAs showed only inflammation and/or debris indicating tumor infarction after FNA. All patients eventually had surgical resection. Histologically, all cases contained abundant macrophages with necrosis and fibroblastic proliferation. Warthin's tumor with a grossly identifiable tumor nodule (0.7 cm) was noted in patient 1 and a microscopic focus (0.2 cm) of Warthin's tumor was identified in patient 3. No identifiable tumor was observed in patient 2 and 4. There are a total of 10 XGS cases in the literature (including four from this series) and Warthin tumor was identified in 50% of reported cases of XGS, suggesting that XGS is an uncommon reactive process to spontaneous or procedure-induced infarction of Warthin tumor. As a diagnostic mimicker for malignancy, a thorough examination and generous sampling of surgical resection specimen is warranted, although a benign salivary gland neoplasm, commonly Warthin's tumor, is often identified.

Clinical and sonographic characteristics of Warthin-like variant papillary thyroid carcinomas.

AIM: To summarize the clinical, ultrasonographic (US) and pathological characteristics of Warthin-like variant papillary thyroid carcinomas (WVPTC). MATERIAL AND METHODS: Medical records and US images of 32 cases of WVPTCs diagnosed between December, 2006 and September, 2018 were reviewed. Clinical, pathological and US characteristics of these cases were collected and summarized. ACR TI-RADS was followed during the analysis of the US features of the lesions. Results: Totally, 32 patients with 33 WVPTC nodules were reviewed. WVPTC was more often seen in female patients (27/32,84.4%) with a relatively high age (mean age, 51.0±10.8 years old). Hyperthyroidism was observed in 14 patients; 2 patients were diagnosed as subclinical hyperthyroidism and 1 patient as subclinical hypothyroidism. Abnormal thyroglobulin antibody was detected in 22 patients. Mean size of the nodule was 1.2±0.5 cm (range, 0.5~2.99 cm) on US. Pathologically, tumor margin of 63.6% carcinomas were infiltrative but most (72.9%) of the enrolled carcinomas were intra-thyroidal. Lymphocytic thyroiditis was detected in 87.5% (28/32) patients. On US, most WVPTCs were solid or almost complete solid (32/33, 97.0%) and very hypoechoic (26/33, 78.8%). Taller-than-wide shape (6/33, 18.2%) and punctate echogenic foci (9/33, 27.3%) were not popular. All the nodules were scored higher than 5 points according to the ACR TI-RADS, including 9 nodules that were classified into TR4 and 24 nodules as TR5. Follow-up information was available in 31 patients and no recurrence or distal metastasis was detected. CONCLUSIONS: WVPTC is a rare variant of PTCs with favorable outcomes. Very hypoechoic echogenicity, solid or almost complete solid composition are the vital indicators for biopsy, even though the nodule may be wider-than-tall and have a lack of punctuate echoic foci.

Warthin-like papillary thyroid carcinoma with immunoglobulin G4-positive plasma cells possibly related to Hashimoto's thyroiditis.

Hashimoto's thyroiditis with heavy lymphoplasmacytic infiltration is a common comorbidity of immunoglobulin G4 (IgG4)-related thyroiditis and Warthin-like papillary thyroid carcinoma (WL-PTC). We hypothesized that WL-PTC may have a strong association with IgG4-related thyroiditis. To validate this hypothesis, we clinically and immunohistochemically studied 17 WL-PTC cases. Fourteen patients (82.4%) had anti-thyroglobulin antibody and were confirmed to have Hashimoto's thyroiditis through microscopic analysis. Among them, five (29.4%) had disease consistent with IgG4-related thyroiditis but did not exhibit a "storiform" pattern or obliterative phlebitis. IgG4-related diseases were not found in other organs. No cases with serum IgG4 level of >135 mg/dL were noted. A total of 94.1% of WL-PTC cases had IgG4-positive plasma cells (+PCs) in the stroma, and cases with rich IgG4+PCs were more frequently associated with Hashimoto's thyroiditis than those with poor IgG4+PCs. In this study, all three cases without Hashimoto's thyroiditis had poor IgG4+PCs, and one of them did not exhibit IgG4+PCs in the stroma of WL-PTC and Hashimoto's thyroiditis. Nodal metastatic lesions were seen in eight cases, all of which were not WL-PTC. As such, we should consider that the Hashimoto's disease with rich IgG4+PCs seen in our cases is representative of non-IgG4-related disease and not IgG4-related disease involving multiple organs. This study is the first to demonstrate the presence of IgG4+PCs in the stroma of WL-PTC. We concluded that the appearance of IgG4+PCs in the stroma of WL-PTC may be related to Hashimoto's thyroiditis with rich IgG4+PC.

Carcinoma papilar de tiroides variante Warthin-like / Warthin-like variant of thyroid papillary carcinoma

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Cutaneous Lymphadenoma: A Trichoblastoma with Regressive Inflammatory Changes.

The authors address the entity of cutaneous lymphadenoma. Although considered benign, cutaneous lymphadenoma can be easily misdiagnosed as basal cell carcinoma because of its close clinical and histological resemblance. This entity is rare and controversial both in terms of its histogenesis and the various diagnostic terms assigned to it throughout the literature. While rare, cutaneous lymphadenoma should be considered in the differential of any facial nodule or papule in addition to the more common basal cell carcinoma, nevi, cysts, and appendiceal tumors.

The incidence of Warthin tumours and pleomorphic adenomas in the parotid gland over a 25-year period.

OBJECTIVES: Pleomorphic adenoma (PA) is reported to be the most common benign parotid tumour followed by Warthin tumour (WT), but the proportion of these two entities might have changed. DESIGN: Retrospective file analysis. SETTING: Tertiary referral head and neck centre. PARTICIPANTS: Patients who underwent a parotidectomy within a period of 25 years (1990-2014). MAIN OUTCOME MEASURES: Rate of occurrence of PA and WT as well as the development of the PA/WT ratio over the years. RESULTS: Overall, 1818 patients with WT (707, 38.9%) and PA (1111, 61.1%) were identified. There was a dominance of PA over WT in all years. An increase in percentage of WT, from 24% in 1990 to 48% in 2014, in comparison with PA was evident. CONCLUSIONS: In our single-institution hospital-based material of parotidectomies, the percentage of WT in comparison with PA has significantly increased over the last 25 years.

Ultrasonographic features and clinical characteristics of Warthin-like variant of papillary thyroid carcinoma.

Warthin-like variant of papillary thyroid carcinoma (WVPTC) is a rare entity recently characterized. We evaluated ultrasonographic (US) features and clinical characteristics of WVPTC. Nine patients were diagnosed with WVPTC through surgery in our institution from May 2005 to January 2015. Eight of nine patients had available preoperative US images. A retrospective review of the US and clinical characteristics was performed. WVPTC compromised of 0.06% of 14,071 PTCs surgically confirmed. A mean age of nine patients was 53.2 years (range, 32-75 years). The mean nodule size of nine WVPTCs was 0.9 cm (range, 0.5-1.5 cm). Two patients showed central nodal metastasis and one patient with conventional PTC as an index tumor underwent central and lateral neck dissection. No one showed recurrence or distant metastasis during the follow-up period (mean, 4.6 years; range, 0.6-10 years). The most common US features of WVPTCs were solid composition (62.5%), hypoechogenicity (75%), and wider-than-tall shape (100%), respectively. Four (50%) of eight nodules showed well-defined margin and three (37.5%) of them had cystic component. One of eight resembled focal thyroiditis. Three nodules were considered as probably benign with US. All nine cases demonstrated underlying heterogeneous parenchymal echogenicity and accompanied chronic lymphocytic thyroiditis in permanent sections. Thyroid function tests in all patients were normal except for one with subclinical hypothyroidism. WVPTC is an uncommon subtype of PTC and has favorable prognosis, which can be misdiagnosed as a probably benign nodule or focal thyroiditis with US. All cases are associated with heterogeneous parenchyma in the background.

An unusual initial presentation of mantle cell lymphoma arising from the lymphoid stroma of warthin tumor.

BACKGROUND: Warthin tumors presenting concomitantly with a lymphoma is vanishingly rare with only 15 reported cases in English literature. Herein, we report an unusual initial presentation of a mantle cell lymphoma involving the lymphoid stroma of a Warthin tumor. CASE PRESENTATION: A seventy-seven year old otherwise healthy gentleman with a 50-pack year smoking history presents with a slowly enlarging left cheek mass. CT scan of the neck demonstrated a left parotid gland tumor measuring 3.4 cm in greatest dimension. He underwent a left superficial parotidectomy, with subsequent histopathologic examination revealing a Warthin tumor with extensive expansion of the lymphoid stroma. Flow cytometric, immunohistochemical, and cytogenetic studies of the stromal component of the tumor confirmed the presence of a mantle cell lymphoma. Clinical staging demonstrated stage IVa disease, and was considered to be at low to intermediate risk due to the slow growth of the parotid lesion. The patient is undergoing close follow up with repeat PET-CT scans at six months. CONCLUSION: To the best of our knowledge, this is the first well documented collision tumor between mantle cell lymphoma and a Warthin tumor. This case also brings to light the significance of thorough evaluation of the lymphoid component of Warthin tumor.

Warthin's tumors and their relationship to lung cancer.

Warthin's tumors (WT) are frequently encountered in clinical practice and can be easily mis-identified. To date, there has been no proven association between WT and lung cancer. A retrospective chart review was conducted of records from 2008 to 2013 in the University of Pennsylvania Health System. Nineteen percent of patients had a diagnosis of lung cancer, compared to the general patient population, where 7.62% of males and 6.26% of females have a lifetime risk of developing lung cancer. Patients with WT are at heightened risk of lung malignancy; detection of WT may lead to earlier diagnosis of lung cancer.

Recoverin-associated retinopathy secondary to Warthin tumor of parotid gland.

PURPOSE: To present a case of photoreceptor degeneration associated with a benign Warthin tumor of the parotid gland. CASE REPORT: A 57-year-old man visited our clinic complaining of blurred vision in both eyes. His best-corrected visual acuity was 0.07 in the right and 0.04 in the left eyes. All components of the full-field electroretinograms (ERGs) were reduced in both eyes. The focal macular ERGs were extinguished in both eyes, which was consistent with the deterioration of the outer retina in optical coherence tomographic images. Positron emission tomography showed (18)F-fluorodeoxy glucose accumulation in the left parotid gland. Parotidectomy was performed, and the histopathology of the specimen had features compatible with a Warthin tumor without malignancy. Western blot analysis of the patient's sera detected an antibody against recoverin. In addition, the tumor tissue had an aberrant expression of recoverin. CONCLUSION: The findings in this case indicate that recoverin-associated retinopathy can develop secondary to a benign Warthin tumor.

Warthin's tumor with superimposed mycobacterium tuberculosis infection.

We report a case of atypical mycobacterium infection in a Warthin's tumor which occurred in a 79-year-old man. The patient had along history of a left parotid mass that underwent rapid growth, became painful, and fistulized. The patient underwent left parotidectomy and neck dissection which showed a Warthin's tumor with areas of necrotizing granulomas. Subsequent culture showed the growth of acid fast bacilli. Consideration of an underlying mycobacterium infection, although rare, is important due to the public health concerns and other treatment needs that such a diagnosis raises.

Warthin's tumor associated with IgG4-related disease.

Immunoglobulin G4-related disease (IgG4-RD) is an inflammatory condition associated with elevated serum IgG4 levels and tissue infiltration by IgG4-expressing plasma cells. Several inflammatory conditions associated with IgG4-RD have been reported. Warthin's tumor is a benign parotid gland tumor consisting of oncocytic epithelial cells and lymphoid stroma containing lymphoid follicles with reactive germinal centers. This is the first report describing a case of Warthin's tumor with possible involvement of IgG4-RD. The patient was a 71-year-old man presenting with swollen right parotid and bilateral submandibular glands. He had a history of a Warthin's tumor of the left parotid gland, autoimmune pancreatitis, and an inflammatory abdominal aortic aneurysm. Laboratory tests revealed a high serum IgG4 level. Histological examination of the resected parotid gland showed a Warthin's tumor and a nodule showing severe lymphocytic infiltration containing many IgG4-positive plasma cells. This case shows the possible involvement of Warthin's tumor with IgG4-RD.

Low grade mucoepidermoid carcinoma in a setting of Warthin's tumor.

Warthin's tumor also known as papillary cystadenoma lymphomatosum is a common benign salivary gland neoplasm that occurs exclusively in parotid gland. Rarely, the tumor can undergo carcinomatous or lymphomatous transformation of epithelial or lymphoid component, respectively. Herein, we describe a case of 55-year-old female who had undergone parotidectomy for a rapidly growing tumor in the right parotid gland. The case was diagnosed as mucoepidermoid carcinoma developed in a setting of Warthin's tumor based on the histomorphology, special staining characters, and immunohistochemical findings. The pathogenesis and differential diagnoses of such rare malignancy has been discussed briefly.

Aneurisma intraparotídeo de vena comunicante posterior / Intraparotid aneurysm of the posterior communicating vein

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