Primary intranodal Warthin-like variant of mucoepidermoid carcinoma.

This report describes a rare case of primary intranodal Warthin-like mucoepidermoid carcinoma (WL-MEC) presenting as a left level II lymph node mass in a 48-year-old man. Warthin-like mucoepidermoid carcinoma is a recently defined variant of MEC that bears a close histologic resemblance to Warthin tumor. Whereas MEC has readily identifiable key histologic features that render diagnosis relatively straightforward, WL-MEC is a challenging diagnosis due to overlapping histologic features and only limited case reports in the literature. This case was initially diagnosed as primary intranodal MEC after the exclusion of metastasis by imaging. It was not until years later, upon review of historic cases, that the diagnosis of WL-MEC was established. This diagnosis was further supported by molecular testing that was not available at the time of the original diagnosis.

Growth rates over time of unoperated parotid benign tumors.

INTRODUCTION: The treatment of parotid benign tumor is in principle surgery, but observation may be necessary in some cases. The purpose of this study was to investigate the growth rates over time of unoperated parotid benign tumors. METHODS: We retrospectively reviewed the medical records of 63 patients with unoperated parotid benign tumors diagnosed at our institution between January 2010 and December 2022. RESULTS: Forty-nine of the 63 patients had a Warthin tumor and 13 patients had a pleomorphic adenoma. On average, the unoperated parotid benign tumors grew 0.02 cm in length and 0.4 cm3 in volume per year. Compared to pleomorphic adenomas, Warthin tumors were more predominant in male patients and in those with a smoking history and a longer duration of smoking history; patients with Warthin tumors were also followed up longer (p < 0.05). However, the length and volume growth rates of unoperated Warthin tumors and pleomorphic adenomas did not significantly differ. CONCLUSION: Surgery is the standard treatment for parotid benign tumors. However, small benign parotid tumors identified during preoperative examination can be observed through close follow-up, taking into account the patient's medical and general condition.

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.

A Safety and Feasibility Trial of Ultrasound-Guided Radiofrequency Ablation of Parotid Warthin's Tumor.

OBJECTIVE: To determine if ultrasound-guided (USG) radiofrequency ablation (RFA) of Parotid Warthin's tumor under local anesthesia is a safe and effective procedure. STUDY DESIGN: Safety and feasibility study. SETTING: Tertiary academic medical center. METHODS: This is an IDEAL phase 2a trial in a tertiary referral center. Twenty patients with Parotid Warthin's tumor were recruited. RFA was done between September and December 2021 for all 20 patients using a CoATherm AK-F200 machine with a disposable, 18G × 7 mm radiofrequency electrode. Results and follow-up statistics were compared with a historic sample of patients with parotid Warthin's tumor who underwent parotidectomy between 2019 and 2021 in the same center. RESULTS: Nineteen patients were included in the analysis as 1 patient dropped out after 4 weeks of follow-up. The mean age for the RFA group was 67 years old with most of them being male smokers. At a median of 45 weeks (44-47 weeks) postprocedure there was a 7.48 mL (68.4%) volume reduction compared to baseline. Three patients had transient facial nerve (FN) paresis, 1 recovered within hours, and the other 2 by 12 weeks follow-up. Three patients had great auricular nerve numbness; 1 patient had infected hematoma treated in an out-patient manner. Compared to a historic cohort of parotidectomy patients for Warthin's tumor, there was no significant difference in FN paresis and other minor complications between the 2 treatment modalities. CONCLUSION: The current analysis suggests that USG RFA of Warthin's Tumor is a safe alternative to parotidectomy with shorter operative time and length of stay.

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-like papillary carcinoma: Case report.

Warthin-like papillary thyroid carcinoma is a rare variant of papillary carcinoma with a very good prognosis. It is often associated with lymphocytic thyroiditis. Due to its typical histological picture resembling Warthin's salivary gland tumor, the histological diagnosis is not difficult, usually does not require an accompanying immunohistochemical examination and is based on the presence of nuclear features typical of papillary carcinoma and the presence of oncocytes in a background of rich lymphocyte infiltrate. The preoperative cytologic examination is challenging, as many other lesions may have a similar picture. Women are more likely to get affected. It appears a decade earlier than the classic variant. Clinically, it presents similarly to a conventional papillary carcinoma. In our case report, we would like to present the case of a 56-year-old woman with non-toxic multinodular goiter, in whom the presence of this rare variant of papillary carcinoma was revealed by histological examination.

Validation of indications for enucleation for benign parotid gland tumors.

BACKGROUND: Enucleation has been reported as a minimally invasive surgery for Warthin's tumor (WT). However, the definite indications for enucleation have not been clarified. METHODS: Enucleation was indicated by the following findings: findings of WT, cystic fluid, or benign leukocytes by fine-needle aspiration cytology; a well-margined and homogeneous pattern on imaging; and a tumor location in the tail or preauricular area of the parotid gland. We reviewed 552 cases treated with parotid gland surgery in our hospital. RESULTS: A total of 108 tumors were treated with enucleation and included no malignant solid tumors or pleomorphic adenoma. Enucleation demonstrated low invasiveness and complication rates. Revision surgery for WT reappearance after enucleation was rare and showed minimal scarring, with a lower risk of facial weakness. CONCLUSIONS: The indication criteria for enucleation were validated. Such enucleation is useful, as it is associated with minimal invasiveness, low complication rates, and safety in revision surgery.

Concurrent Warthin tumor and Kimura disease: a case report.

BACKGROUND: Warthin tumor (WT) is a common benign salivary tumor of the parotid gland. Clinically, it occurs in men in their fifth to seventh decades who typically smoke cigarettes. WTs have been reported with different head and neck neoplasms and other salivary gland tumors within the same or another salivary gland. Kimura disease (KD) is a rare chronic inflammatory disease with unknown etiology affecting young to middle-aged Asian men. KD presents as an asymptomatic nodule in the head and neck area, with regional lymphadenopathy and salivary gland involvement. CASE PRESENTATION: A 64-year-old Arabic man presented with a 10-year history of an asymptomatic swelling of the left face. Computed tomography showed a well-defined, multicystic mass with heterogeneous enhancement. The resected mass was composed of two distinct components. There was a well-demarcated proliferation of papillary and cystic oncocytic epithelium with lymphoid stroma, consistent with WT. Some areas exhibited sclerotic fibrosis, with multiple lymphoid follicles showing folliculolysis, follicular hyperplasia, and eosinophilic infiltrate. The patient's immunoglobulin E level serum was elevated, confirming a coexisting KD. The patient underwent a left superficial parotidectomy, with no recurrence at a 30-month follow-up. CONCLUSION: This report describes the first concurrent case of WT and KD in the parotid gland.

Multiple synchronous neoplasms in the ipsilateral parotid gland: case series.

BACKGROUND: Multiple synchronous neoplasms in the ipsilateral parotid gland are very rare. We intend to analyze the clinical characteristics and treatment results of multiple synchronous neoplasms in the ipsilateral parotid gland managed at our hospital. METHODS: The study included 21 patients of multiple synchronous neoplasms in the ipsilateral parotid gland from January 2010 to December 2020. RESULTS: Twenty-one cases of multiple synchronous neoplasms in the ipsilateral parotid gland were identified among 994 cases of parotid surgery, with a frequency of 2.1%. Multiple synchronous neoplasms in the ipsilateral parotid gland occurred in all males and one female. Except for one case, all of them involved histopathologic neoplasms. Warthin tumor (n=19, 90.5%) was the most common. Among 21 lesions, there were 9 patients of right parotid gland, 4 patients of left parotid gland, and 4 patients of bilateral parotid gland. The main symptoms were slowly enlarging mass within the parotid gland (n=20), followed by incidental detection (n=1). CONCLUSIONS: Clinicians should be aware of the possibility of multiple synchronous neoplasms in the ipsilateral parotid gland. Preoperative imaging tests, clinical examinations, and careful palpation during surgery are important for accurate diagnosis and treatment.

Warthin-like mucoepidermoid carcinoma of the parotid gland: a clinicopathological analysis of two cases.

Mucoepidermoid carcinoma (MEC) is the most common malignant tumour of the salivary gland, primarily involving the parotid gland. Here, the cases of two patients, aged 47 and 67 years, respectively, who underwent surgery for pathologically confirmed Warthin-like MEC of the parotid gland between January 2019 and December 2019 in Anyang Tumour Hospital, are described. In each case, the tumour consisted of epithelial and lymphoid cell components, covered with two or more layers of epithelium, with visible scattered mucous cells, and lymphoid stroma with a large number of lymphocytes and germinal centres formed. Most importantly, the tumours lacked the well-organized, bilayered oncocytic epithelial structure that is characteristic of Warthin's tumour. Mastermind like transcriptional coactivator 2 (MAML2) gene rearrangements were identified in the tumour cells using break-apart fluorescence in situ hybridization (FISH) probes, confirming the diagnosis of Warthin-like MEC. Post-operatively, patients have remained disease free for 31 and 27 months, respectively. Warthin-like MEC of the parotid gland is rare and is often misdiagnosed as metaplastic Warthin's tumour. Diagnosis depends mainly on the unique clinicopathologic features together with FISH analyses.

Synchronous bilateral multifocal basal cell adenomas of the parotid gland-a case report.

BACKGROUND: Bilateral parotid gland tumors account for up to 3% of cases. In this group, the vast majority are Warthin's tumors. However, bilateral presentations of other parotid gland tumor entities is also possible, an example of which is a basal cell adenoma (BCA). Bilateral BCA is extremely rare, which could cause misdiagnosing it as a Warthin tumor. CASE PRESENTATION: The current study reports the unique case of a 48-year-old woman who presented with a 6-month history of slowly growing masses located bilaterally in the parotid region, surgically treated with 5-year follow-up (no recurrence, normal facial nerve function). Magnetic resonance imaging (MRI) revealed three lesions: two in the superficial and deep lobes of the right parotid gland, and one in the superficial lobe of the left parotid gland. A total parotidectomy with facial nerve preservation was performed on the right side, and superficial parotidectomy on the left side 6 months later. Histopathological examination confirmed that all three tumors were BCAs. Molecular analysis didn't show any strong, potential of unknown clinical significance in the studied sample. CONCLUSIONS: Multifocal bilateral lesions of the parotid gland are usually Warthin tumors. Detailed preoperative diagnostics including MRI and histopathological examination is essential to avoid misdiagnosing BCA and Warthin tumors. To our best knowledge, no case of synchronous bilateral multifocal basal cell adenomas of the parotid gland has been reported in English literature so far.

Nonsebaceous lymphadenoma arising as a painless subcutaneous nodule on the eyebrow.

Salivary gland tumors can rarely present in skin excision specimens and can pose a diagnostic challenge to dermatopathologists. We present an exceptional case of a salivary gland type nonsebaceous lymphadenoma presenting as a painless subcutaneous nodule on the right medial eyebrow of a 16-year-old male, mimicking a primary cutaneous adnexal neoplasm. Histologic evaluation revealed a well-circumscribed to partially encapsulated nodule with a marked lymphoid infiltrate including reactive germinal centers. Within the lymphoid component was a central epithelial cystic neoplasm with tubuloglandular and basaloid differentiation. There was no myoepithelial component to suggest a chondroid syringoma. No sebaceous differentiation was present. The overall histopathological and immunohistochemical findings were consistent with a nonsebaceous lymphadenoma. Dermatopathologists should consider salivary gland type lymphadenoma as a differential diagnosis when encountering a subcutaneous lesion with lymphoid, cystic, glandular, and basaloid components.

Function-preserving surgery via single transverse cervical incision for Warthin's tumor in the parotid gland.

BACKGROUND: This study evaluated functional and disease control outcomes after gland-preserving surgery via a single transverse cervical incision for Warthin's tumor of the parotid gland. METHODS: One-hundred eleven patients with Warthin's tumor underwent the preservation of most normal parotid tissues and the facial nerve combined with the en-bloc resection of tumors. Postoperative complications, subjective satisfaction, salivary function, and metachronous tumor were assessed in each patient. RESULTS: No patients had an extension to modified Blair or periauricular incision with the median operation time of 32 min. Temporary and permanent paralysis of the facial nerve was 5 (4.0%) and none of 125 tumor resection sides, respectively. Postoperative complications were minimal. The secretory functions between the affected and unaffected glands did not differ. One patient had metachronous tumor for a median follow-up of 72 months. CONCLUSION: Functional gland-preserving surgery via single transverse cervical incision can safely treat Warthin's tumor in the parotid gland.

Experience with follow-up strategy in selected patients with Warthin tumour diagnosed by ultrasound-guided fine-needle aspiration biopsy (FNAB).

PURPOSE: Warthin tumour (WT) management options comprise surgery or follow-up. The purpose of this study was to asses our experience with the follow-up strategy in selected patients with an ultrasound-guided fine-needle aspiration biopsy (FNAB) showing WT. METHODS: We performed a retrospective analysis of patients diagnosed with WT using FNAB between 1.1.2006 and 31.12.2019. Patients were divided into three groups according to the therapeutic approach-immediate surgery, follow-up or surgery and follow-up. RESULTS: 323 patients were diagnosed with WT and met the study's inclusion criteria (154 women, 47.7% and 169 men, 52.3%). 192 patients were operated right after the diagnosis, 109 patients were observed with their first detected tumour and 22 patients had parotid WT surgery and were in the wait-and-scan protocol with a contralateral tumour, recurrence or both. The growth rate (GR) of observed WT was highly variable (mean GR 1.0 mm/year (5%), median GR 0.8 mm (9%), range - 19.7 to +20.0 mm/year). From 131 patients in the follow-up group, 19 patients underwent surgery and definitive histology revealed 17 WTs and 2 adenocarcinomas. However, these 2 patients had changes in sonographic findings at their next control. The mean observation time was 44.7 months (range 12-138 months) in patients followed exclusively at our institution and 50.9 months (range 12-110 months) in patients observed in cooperation with an otorhinolaryngologist at the patients' place of residence. CONCLUSION: Ultrasound-guided FNAB is an accurate and simple method in WT diagnosis and based on its result a follow-up strategy can be chosen for selected patients with WT.

Primary Warthin's-like adenocarcinoma of the lung: A clinicopathological, immunohistochemical, and molecular analysis of three cases.

Three cases of primary pulmonary adenocarcinoma with prominent lymphoid stroma and papillary features mimicking Warthin's tumor are presented. The patients are two women and one man ages 52, 62, and 74 years respectively. Clinically, the patients presented with non-specific symptoms of cough, dyspnea, and chest pain. Imaging showed the presence of an intrapulmonary mass in the right lower, right upper and left lower lobe. All patients underwent lobectomy. Histologically, the tumors were characterized by the presence of and oncocytic papillary growth pattern embedded in a lymphoid rich background. Immunohistochemical stains for TTF-1, keratin 7, and beta-catenin were positive in the epithelial component, while CD20 showed strong positive staining in the lymphoid component. In addition, CD4 and CD8 also showed positive staining in a ratio of 3-4:1, EBER was negative. Kras mutations with wild type EGFR were identified in one case. Clinical follow-up ranging from 8 to 24 months was obtained showing that all patients are alive without recurrence. The cases herein presented represent an unusual histological variant of primary lung adenocarcinoma, which closely mimics Warthin's tumor.

The potential role of follicular helper T cells and helper T cells type 1 in Warthin tumour.

Warthin tumour (WT) is a benign tumour of the salivary gland that proliferates in both glandular epithelial and lymphoid tissue components, and rarely exhibits cystic changes. T follicular helper cells (Tfh) are involved in the formation and maintenance of germinal centres, the differentiation of B cells into plasma cells, and the maintenance of helper T cell type 2 (Th2)-dominant humoral immune responses. T-bet induces differentiation into helper T cell type 1 (Th1) by suppressing differentiation into Tfh and enhances cellular immune responses. The objective of this study was to enhance our understanding of the immune responses and relationship between Tfh and Th1 cells in patients with WTs. In this study, we classified WTs (n = 64) into solid-type (n = 25) and cyst-type (n = 39). We also performed immunostaining of the Tfh markers CXCR5 and CD40 L, and the Th1 marker T-bet for statistical analysis. The cyst-type exhibited significant atrophy of the germinal centre area (P = 0.0019), significantly fewer Tfh-positive lymphocytes in germinal centres (P < 0.0001), and significantly more T-bet-positive lymphocytes in the epithelium (P = 0.0017). We observed that Tfh were involved in the formation and maintenance of lymphoid follicles in WTs. In the cyst-type, Th2-dominant humoral immune responses were suppressed, and Th1-dominant cellular immune responses may have caused damage to tumour tissue.

Prevention of Frey's Syndrome with the Use of Porcine Dermal Collagen Graft: Retrospective Analysis of 76 "Formal" Parotidectomies for Benign Pathologies.

BACKGROUND: Frey's syndrome is a well-known complication of parotid surgery; its prevention may be achieved by the use of an interpositional barrier between the overlying flaps and the exposed parenchymal bed of parotid gland. The aim of this study was to retrospectively evaluate clinical outcomes with and without the interpositional placement of a porcine dermal collagen graft (PDCG) for prevention of syndrome occurrence. METHODS: We conducted a 20-year retrospective study including the patients who had undergone "formal" (superficial, total, or subtotal) parotidectomies for benign pathologies. The inclusion criteria also involved patients that were (i) regularly monitored about clinical symptoms related to syndrome, and (ii) examined with Minor starch-iodine test. The severity of the diagnosed syndrome was retrospectively evaluated according to the grading score system of Luna-Ortiz. To assess group differences in terms of the extent of dissection in operating sites, we estimated the tumor and histological specimen volumes using the available dimensions. RESULTS: We included 73 patients who had undergone 76 formal parotid surgeries. The surgical sites were divided into 2 groups: (1) Group A consisted of 44 sites that were reconstructed with a SMAS flap, and (2) Group B, comprised 32 sites where a PDCG was additionally applied as an artificial preventive barrier. At a mean follow-up of 26.3 months, a significantly lower incidence of clinically diagnosed Frey's syndrome was found after the use of dermal collagen interpositional barrier (P = .031). Specifically, subjective symptoms were reported at an incidence of 31.8% in Group A and 6.7% in Group B. Minor's test was positive at an incidence of 59.09% in Group A and 21.87% in Group B (P = .004, 95% CI). Severe Frey's syndrome was observed in 31.82% of the patients of Group A and in 3.12% of the patients of Group B (P = .002, 95% CI). Since there were no statistical significant differences between the volumes of the removed tumors and the excised histological specimens, the extent of dissection was not proved to influence the occurrence of Frey's syndrome in the compared groups. CONCLUSION: Porcine dermal collagen is a safe, practical, and useful means for parotid reconstruction, since it seems to contribute in prevention of Frey's syndrome when increased amount of glandular tissue has to be removed. Additional randomized controlled studies with bigger samples are required to better assess the PDCG use in parotid surgery.

Efficacy of modified face lift incision for the resection of benign parotid gland tumor located anteriorly or superiorly.

OBJECTIVES: The goals of resection of benign parotid gland tumor are complete resection of lesion and preservation of the facial nerve function. Traditionally, the bayonet-shaped incision (Blair incision: BI) and the modified face lift incision (mFLI) are commonly used for parotidectomy. However, concerns exist about the adequacy of exposure and identification of the facial nerve in anterior or superior parotid lesions. The aim of this study was to compare the surgical outcomes between BI and mFLI and to evaluate the adequacy, possible indications, and limitations of mFLI for the resection of benign parotid gland tumors located anteriorly or superiorly. METHODS: This retrospective study analyzed the medical records of 175 patients with various types of benign parotid tumor who underwent partial parotidectomy via BI (97 patients) or mFLI (78 patients). Tumors were divided into five categories depending on their location: anterior, superior, inferior, middle, and deep lobe tumors. The outcomes of operation were analyzed according to tumor location between the incision types. RESULTS: Tumor locations were not significantly different between the two groups. Transient facial palsy occurred in 23 out of 152 patients (15.1%); permanent palsy was not observed in either group. The incidence rates of facial palsy were higher among patients with superior and deep lobe tumors; in the mFLI and BI groups, proportions of superior tumors were 22.2% and 27.2%, respectively, and those of deep lobe tumors were 35.7% and 23.5%, respectively. With regard to superior and anterior tumors, the incidence rate of postoperative facial palsy was insignificantly lower in the mFLI group (10.5%) than in the BI group (18.2%). CONCLUSIONS: There were no differences in the incidence rates of postoperative facial palsy between mFLI and BI for any tumor location. Use of the mFLI is feasible for the resection of most benign parotid tumors located anteriorly or superiorly.

Warthin Tumor of the Caruncle: A Rare Case Report and Review of the Literature.

The authors describe a case of Warthin tumor arising in the caruncle of a 76-year-old man. Clinical examination identified a left medial canthus swelling that was treated by complete local excision. Pathologic examination revealed a benign epithelial tumor with diagnostic features of Warthin tumor. Clinical and histologic features, along with review of the literature, are discussed.