Warthin's tumor with necrotizing tuberculous granulomatous inflammation causing severe facial nerve adhesion in parotid gland: A case report and literature review.

RATIONALE: Warthin's tumor is the second most common tumor arising from the parotid gland, but it rarely occurs concomitantly with tuberculous granulomatous inflammation with only 13 documented case reports in the English literature. PATIENT CONCERNS: An 82-year-old woman had a left infraauricular mass for approximately 3 years that had significantly increased in size over the previous 1 month. DIAGNOSES: A diagnosis of Warthin's tumor was made by ultrasonography (US)-guided core needle biopsy. Pathological examinations of the specimen obtained by total extirpation confirmed that the tumor was superimposed with tuberculous granuloma. INTERVENTIONS: The core biopsy wound did not heal and there was formation of a skin fistula tract with persistent discharge. During the operation with en bloc resection of the necrotic parotid tumor, adhesion between the branches of the facial nerve was too tight to allow preservation. OUTCOMES: A diagnosis of necrotic Warthin's tumor superimposed with tuberculous granuloma was made. Due to the high-clinical suspicion of tuberculosis (TB) due to Mycobacterium tuberculosis infection, anti-TB chemotherapy was given. LESSONS: Poor wound healing from a core biopsy and formation of a skin fistulous tract with persistent discharge should raise concern regarding potential extrapulmonary tuberculous infection. Although very rare, tuberculous granuloma concomitant with Warthin's tumor should be considered in the differential diagnosis of a parotid mass lesion. Adhesion of branches of the facial nerve should be expected, and sacrifice of the nerve may be planned. This consideration can be explained to the patient in preoperative counseling and planning. Anti-TB chemotherapy should be given in cases with a definite pathological report associated with speculative clinical presentation.

Surgical treatment strategy in Warthin tumor of the parotid gland / Estratégia de tratamento cirúrgico no tumor de Warthin de glândula parótida

Abstract Introduction: Warthin tumors are the second most common benign tumors of the parotid gland. We examined the clinical features of Warthin tumors in our hospital, and analyzed the consistency within the literatures. Objective: The aim of this study is to analyze the clinical features of Warthin tumors in our 10-year experience of 118 Warthin tumors undergoing surgery at a single institute. Methods: From December 2006 to December 2016, 110 patients who underwent surgical treatment for Warthin tumors were identified based on their medical records. Results: A total of 118 parotid gland operations were performed in 110 patients. Almost 90% of Warthin tumors were found in males, and average patient age was 66.1 ± 6.1 years. The prevalence of smoking history was 89.1% (98/110). Eight patients (7.3%) had bilateral Warthin tumors. Seventy-seven lesions (65.3%) were located in the parotid tail portion, followed by 34 lesions in the superficial lobe (28.8%) and 7 lesions in the deep lobe (5.9%). Conclusion: We determined the appropriate extent of surgery depending on the fine needle aspiration cytology and tumor location by computed tomography scans. Partial facial dysfunction after the operation was detected in 12 cases, and facial nerve function recovered within 3 months. Only one patient experienced a recurrence, and was disease free after the re-operation. We suggest that our treatment algorithm, depending on the location of tumors and the result of fine needle aspiration cytology, can be useful to determine the appropriate extent of surgery for Warthin tumors.

Resumo Introdução: Os tumores de Warthin são os segundos tumores benignos mais comuns da glândula parótida. Avaliamos as características clínicas dos tumores de Warthin em nosso hospital e analisamos a consistência com a literatura. Objetivo: Analisar as características clínicas dos tumores de Warthin em nossa experiência de 10 anos de 118 tumores de Warthin submetidos a tratamento cirúrgico em um único instituto. Método: De dezembro de 2006 a dezembro de 2016, 110 pacientes que receberam tratamento cirúrgico para tumores de Warthin foram identificados com base em seus prontuários médicos. Resultados: Foram feitas 118 cirurgias na glândula parótida em 110 pacientes. Quase 90% dos tumores de Warthin foram encontrados em homens e a média da idade dos pacientes foi de 66,1 ± 6,1 anos. A prevalência de tabagismo foi de 89,1% (98/110). Oito pacientes (7,3%) tinham tumores de Warthin bilaterais na glândula parótida. Das lesões, 77 (65,3%) localizavam-se na porção da cauda da parótida, seguidas por 34 no lobo superficial (28,8%) e 7 no lobo profundo (5,9%). Conclusão: Determinamos a extensão apropriada da cirurgia de acordo com a punção aspirativa com agulha fina e localização do tumor por tomografia computadorizada. Disfunção facial parcial após a cirurgia foi detectada em 12 casos e a função do nervo facial foi recuperada em 3 meses. Apenas um paciente apresentou recidiva e ficou livre da doença após reoperação. Sugerimos que nosso algoritmo de tratamento, a depender da localização dos tumores e do resultado da PAAF, pode ser útil para determinar a extensão apropriada da cirurgia para os tumores de Warthin.

Surgical treatment strategy in Warthin tumor of the parotid gland.

INTRODUCTION: Warthin tumors are the second most common benign tumors of the parotid gland. We examined the clinical features of Warthin tumors in our hospital, and analyzed the consistency within the literatures. OBJECTIVE: The aim of this study is to analyze the clinical features of Warthin tumors in our 10-year experience of 118 Warthin tumors undergoing surgery at a single institute. METHODS: From December 2006 to December 2016, 110 patients who underwent surgical treatment for Warthin tumors were identified based on their medical records. RESULTS: A total of 118 parotid gland operations were performed in 110 patients. Almost 90% of Warthin tumors were found in males, and average patient age was 66.1±6.1 years. The prevalence of smoking history was 89.1% (98/110). Eight patients (7.3%) had bilateral Warthin tumors. Seventy-seven lesions (65.3%) were located in the parotid tail portion, followed by 34 lesions in the superficial lobe (28.8%) and 7 lesions in the deep lobe (5.9%). CONCLUSION: We determined the appropriate extent of surgery depending on the fine needle aspiration cytology and tumor location by computed tomography scans. Partial facial dysfunction after the operation was detected in 12 cases, and facial nerve function recovered within 3 months. Only one patient experienced a recurrence, and was disease free after the re-operation. We suggest that our treatment algorithm, depending on the location of tumors and the result of fine needle aspiration cytology, can be useful to determine the appropriate extent of surgery for Warthin tumors.

Back to the roots of Warthin's tumor of the parotid gland.

The exact etiopathogenesis of the Warthin's tumor of the parotid gland is still unclear. Since the time of the first histological description of this lesion by Hildebrand and later Albrecht and Arzt many different partly confusing hypothesis about pathogenesis of Warthin's tumor were introduced. However, the review of published data about the pathogenesis of this lesion revealed that recent literature is in conformity with the supposed hypothesis about pathogenesis of Warthin's tumor of those authors about the origin of this lesion from lymph nodes almost a century ago.

Galectin fingerprinting in Warthin's tumors: lectin-based approach to trace its origin?

Warthin's tumor of the parotid gland is assumed to originate from the proliferation of epithelial inclusions within parotid lymph nodes. In that case, these cells are supposed to retain characteristics similar to common salivary gland ductal cells. Using immunohistochemical fingerprinting with four members of the family of adhesion/growth-regulatory galectins and comparison to intra- and interlobular ducts, marked similarities were noted for presence of galectins-3, -7 and -8. Notably, profiles of lectin binding, determined by applying human lectins as probes, were also similar when testing biotinylated galectins-3 and -8. Besides defining the galectin histochemical parameters in Warthin's tumors this study adds support to the hypothesis of heterotopia.

Using time-dependent covariate analysis to elucidate the relation of smoking history to Warthin's tumor risk.

The authors aimed to elucidate the relation of the time-dependent smoking history parameters--age at smoking initiation and smoking intensity, duration, and latency--to the risk of Warthin's tumor, a benign tumor of the salivary gland for which cigarette smoking is a strong risk factor. They studied 117 cases of Warthin's tumor and 336 matched controls included in an Israeli nationwide case-control study of parotid gland tumors conducted from 2002 to 2003 by using the Cox regression model with time-dependent covariates, with age as the time axis. When current age and smoking duration were included in the statistical model, the authors show that the coefficient of a latency variable does not represent latency as such, but a balancing of the effects of age at initiation and time since cessation. They found a strong positive linear effect of duration of smoking, together with a positive nonlinear effect of intensity that levels off at higher intensities, and a negative effect of latency from 25 years onward. The latter finding implies that the effect of time since cessation dominates the effect of age at initiation, with risk decreasing sharply after smoking cessation. The relation of smoking variables to Warthin's tumor agrees with the patterns reported for lung cancer.

Tumor solido pseudopapilar: presentacion de caso, manejo y tratamiento quirurgico curativo / Solid pseudopapilar tumor: case presentation, handling and surgical healing treatment

Descrito por primera vez por Frantz en 19591,2 lostumores sólidos pseudopapilares (TSS) son neoplasiasde origen epitelial, de etiología incierta que afectangeneralmente a mujeres jóvenes.2 Dentro la sinonimiapara referir dicha lesión se los conoce también comoneoplasia papilar epitelial, tumor acinar sólido y quístico,neoplasia papilar quística, carcinoma papilar quístico, tumor papilar de bajo grado y finalmente tumor de Frantzpor su primera descripción. Si bien es cierto que tienenun potencial maligno, este es solo del 1 a 2 3 y no fuesino hasta 1996 cuando la OMS4 lo reconoce con suactual nombre, tumor sólido pseudopapilar.Debido a que la neoplasia es muy rara, el objetivode este reporte de casos es presentar el caso clínicoy discutir sobre algunos hechos de su diagnóstico y manejo quirúrgico.

High risk for bilateral Warthin tumor in heavy smokers--review of 185 cases.

CONCLUSIONS: We identified smoking as a significant risk factor for multilocular Warthin tumor development. Therefore, we recommend taking history of smoking into account when making the decisions for surgical strategy. OBJECTIVES: Warthin tumor is a common benign neoplasm of the parotid gland. Risk factors for multilocular development have not been defined. PATIENTS AND METHODS: A total of 185 consecutive patients treated for Warthin tumor were included. Charts were reviewed for symptoms, risk factors, and diagnostic and surgical procedures. Patients were followed for facial function and recurrence. Risk factors were evaluated. RESULTS: Overall, in 203 parotid operations, a lateral parotidectomy (77%) was performed in most cases; 94% were primary surgery and 6% were revision surgery. In 89% of patients swelling was the only symptom. Bilateral Warthin tumor was seen in 17% of patients. Of these cases synchronous and metachronous bilateral tumors were observed in 61% and 39%, respectively. The median time period for second contralateral tumor development was 7 years. Postoperative transient facial dysfunction was observed in 31%, which recovered within 3 months in all cases. Evaluation of risk factors revealed that 89% of the subjects were smokers and 66% were heavy smokers. The risk for bilateral Warthin tumors correlated significantly with the amount of nicotine intake (p=0.003).

Warthin's tumor with multiple granulomas: a clinicopathologic study of six cases.

Granulomatous inflammation occurring in Warthin's tumor is a rare phenomenon. It has been suggested that prior fine-needle aspiration (FNA) might be a triggering factor. We studied cases of Warthin's tumor with granulomatous inflammation to analyze the possible relationship with prior FNA. Granulomatous inflammation was noted in 6 cases (1.6%) of 382 cases of Warthin's tumor in a medical center. Clinical history, histology, and cytology slides were reviewed. Special stains for detection of infective agents were performed. All 6 cases showed typical features of Warthin's tumor accompanied by multiple granulomas. No specific microbiologic infective agents were revealed in these cases. There were 2 cases with history of preoperative FNA; both already had granulomas in Warthin's tumor in cytologic specimens taken by aspiration. The pathogenesis of granulomatous inflammation in Warthin's tumor remains speculative. Our observation of granulomas in the cytology specimens did not support the hypothesis of prior FNA being the cause.

Clinical features of cystadenolymphoma (Warthin's tumor) of the parotid gland: a retrospective comparative study of 96 cases.

The details of the etiopathogenesis of cystadenolymphoma (Warthin's tumor) are still unclear. To explore the possible risk factors for the development of this tumor, medical records of 81 patients with 96 Warthin's tumors of the parotid glands were compared retrospectively with those of 91 patients with pleomorphic adenoma. The medical history and clinical tumor characteristics of all patients were similar. There were no significant differences between these two patient groups with respect to concomitant diseases, regular medications, and preoperative laboratory findings. However, a significant male predominance of patients with Warthin's tumor could be noted (P<0.05). The male to female ratio was 3.3:1 in patients with Warthin's tumor. Multifocal Warthin's tumor were detected in five cases (6.2%), and 10 patients (12.3%) had bilateral lesions. The odds ratio for the incidence of Warthin's tumor among current smokers compared with never smokers was 8.3 (P<0.0001). Compared with never smokers, clearly higher odds of Warthin's tumor was observed in heavy smokers (more than 30 pack-years) (odds ratio=24.1, P<0.0001) than patients who smoked less than 30 pack-years (odds ratio=4.9, P<0.0001).

Warthin's tumour and smoking.

OBJECTIVE: In an evaluation of our patients with parotid gland neoplasms, we noticed that patients with a Warthin's tumour were heavy smokers. The aim of this study was to confirm earlier findings in the literature concerning a possible association between smoking and the development of a Warthin's tumour. METHODS: A case control study was performed using the clinical records and discharge letters of all consecutive patients with a Warthin's tumour in the pathology database of our hospital covering the last 15 years. Patients with a pleomorphic adenoma and a group of patients visiting our audiology department were used as controls. RESULTS: A smoking history was found in 97.5% of the patients with a Warthin's tumour. Of the patients with a pleomorphic adenoma, 59% had a smoking history; 56.5% of the audiology group had a smoking history. Mean age at the time of the operation was 60.1 years of age in the Warthin's tumour group and 48.6 for the pleomorphic adenoma group. CONCLUSION: The mean age for the development of a Warthin's tumour is ten years older than for a pleomorphic adenoma. Furthermore, the development seems to be closely related to smoking habits.

Mitochondrial DNA mutations in the parotid gland of cigarette smokers and non-smokers.

It has previously been demonstrated that mitochondrial DNA (mtDNA) mutations accumulate in the lung and increase in frequency with age. It has also been shown that the level of mtDNA mutations including deletions and base substitutions are elevated in lung tissue of smokers relative to non-smokers. We have previously shown that the 'common' 4977 bp mtDNA deletion is present in the parotid (salivary) gland of smokers and non-smokers and that there is a significant increase in the level of this deletion in Warthins tumour, an oncocytoma of the parotid gland. In this study we used semi-quantitative PCR to confirm the presence of 4977 bp mtDNA deletion in the parotid gland of non-smokers and smokers. Importantly, we show that the deletion accumulates with age regardless of smoking status and that there was no significant difference in the level of the 4977 bp deletion in parotid tissue of smokers and non-smokers. Using strand conformational polymorphism (SSCP) and direct sequencing we also found 5/23 smokers had parotid tissue specific base substitutions: either an A/T to G/C transition at A4767 or a G/C to A/T transition at G4853. These results are evidence of age related increase in the 4977 bp deletion and a higher level of mutations, probably due to oxidative damage, in the parotid gland of smokers.

Role of Epstein-Barr virus and cytomegalovirus in the etiology of benign parotid tumors.

BACKGROUND: Pleomorphic adenomas and Warthin's tumors are the two most common benign parotid tumors. Previous studies investigating the role of viruses in tumorigenesis of these neoplasms have been conflicting. The aim of this study was to determine whether Epstein-Barr virus (EBV) or cytomegalovirus (CMV) might play a role in the pathogenesis of pleomorphic adenomas and Warthin's tumors. METHODS: Paraffin-embedded surgical specimens of 24 pleomorphic adenomas, 10 Warthin's tumors, and 13 normal parotid tissues were obtained from the University of California-San Francisco Pathology Department. Genomic DNA was extracted from the specimens, and primers for connexin 26, a gap junction protein, were used to confirm the integrity of this DNA. The presence or absence of EBV and CMV DNA within the samples was determined with PCR-based assays, in which radiolabeled primers were used for maximal sensitivity of detection. RESULTS: PCR analysis of serially diluted control DNA revealed that using radiolabeled primers, five copies of viral DNA could be detected. By use of this method, we showed that none of the 24 pleomorphic adenomas, 10 Warthin's tumors, or 13 normal parotid samples contained EBV DNA or CMV DNA. CONCLUSIONS: These results do not support CMV or EBV as etiologic factors in pleomorphic adenomas or Warthin's tumors. In addition, normal parotid seems not to harbor either of these viruses. Future studies with larger numbers of specimens are needed to confirm these findings.

Metaplastic (infarcted) Warthin's tumour of the parotid gland: a possible consequence of fine needle aspiration biopsy.

AIMS: The metaplastic (or infarcted) variant of Warthin's tumour is characterized by replacement of much of the original oncocytic epithelium by metaplastic squamous cells, along with areas of extensive necrosis, fibrosis and inflammatory change. The pathogenesis is unknown, but it is most likely to be vascular in origin. An association with a previous fine needle aspiration (FNA) has been suggested, and this is explored further. METHODS AND RESULTS: Nine metaplastic Warthin's tumours were collected from several centres: all arose in the parotid gland, and all showed the characteristic histological features. Eight had previously undergone FNA some 1-4 months before surgery; the other case had had an incisional biopsy. CONCLUSIONS: It is important to recognize metaplastic Warthin's tumour, because the differential diagnoses of this benign neoplasm include mucoepidermoid and squamous carcinoma, both primary and metastatic. The tumours in this study followed FNA or biopsy, and we believe this association is unlikely to be coincidental. Although many metaplastic Warthin's tumours clearly arise spontaneously, we conclude that the balance of probabilities favours the view that FNA is capable of causing metaplastic change in a Warthin's tumour, and may have done so in these cases. If so, this previously unusual subtype will become increasingly common, as FNA becomes more widely used (and its value appreciated) in the investigation of patients with a mass in the neck.

Malignant oncocytoma of major salivary glands. Report of a post-irradiation case.

The fourth case of malignant oncocytoma arising in the submandibular gland is here reported. This tumor arose in a 48-year-old man after radiation exposure, a finding never described before for malignant oncocytoma. In addition, several regional metastatic lymph nodes were found. The diagnosis was confirmed by histochemical and ultrastructural findings. The tumor cells showed easily recognizable mucus production and, ultrastructurally, abundant mitochondria, intracytoplasmic lumina lined by microvilli and lipid droplets. These last features have only seldom been described in malignant oncocytoma. Furthermore, the neoplastic cells were alpha-1-antitrypsin positive and S100, thyroglobulin, carcinoembryonic antigen, and smooth muscle actin negative. A thorough review of the literature is also presented.

Smoking and the development of Warthin's tumour of the parotid gland.

The smoking history was surveyed in 160 consecutive patients with Warthin's tumour of the parotid gland in order to investigate the relationship between smoking and the development of this tumour. Two hundred cases of middle-aged or elderly normal persons and 163 consecutive patients with pleomorphic adenoma in the parotid gland served as controls. The percentage of smoking in patients with Warthin's tumour (96.3%) was much higher than that of middle-aged or elderly normal persons (25.5%) and in patients with pleomorphic adenoma (26.4%). Moreover, the amount of smoking was greater and the smoking history was longer in patients with Warthin's tumour. When the male and female patients were analysed respectively, the same result was shown. This case-control study suggests that smoking may be one of the aetiologic factors associated with the development of Warthin's tumour.

Warthin's tumor and cigarette smoking.

Warthin's tumor is a relatively uncommon salivary gland neoplasm, traditionally considered a disease of men. Recent reports have brought this assumption into question by identifying a substantial percentage of patients who are women. Furthermore, several reports have attempted to correlate cigarette smoking with development of Warthin's tumor. We retrospectively studied all cases of parotid tumor entered in the Brooke Army Medical Center Tumor Registry from 1973 to 1993. Relative percentages of benign and malignant disease, as well as frequency of individual tumor types, in general mirrored those in earlier reports. A high proportion of Warthin's tumor-23% of all tumors-was identified. More than one third of patients with Warthin's tumor were women. Also, 94% of patients with Warthin's tumor had a history of tobacco use; for all other tumor types combined, only 60% of patients had a history of tobacco use. This statistical difference supports the correlation between cigarette smoking and Warthin's tumor.

[Warthin tumor of the palate: an unusual location]. / Il tumore di Warthin del palato: un'insolita localizzazione.

UNLABELLED: Warthin's tumor (papillary cystadenoma lymphomatosum) primarily involves the parotid gland, whereas localization in the palate has been reported by a few authors. A new case of true Warthin's tumor arising from the hard palate is here presented and histogenesis and differential diagnosis are briefly discussed. CASE REPORT: A 50 year-old woman who was referred to us because of the presence of multiple contiguous bluish cyst-like lesions involving both sides of the hard palate mucosa. The lesion was removed with a wide excision involving almost all the soft tissues of the hard palate. Histologic diagnosis (E.E stain) was: papillary cystadenoma lymphomatosum. DISCUSSION AND CONCLUSION: The histogenesis of Warthin's tumor is still controversial. The most widely accepted theory is that the tumor represents a neoplastic proliferation of salivary gland ducts entrapped in pre-existing lymph nodes. In fact, immunohistochemical analysis and cell-surface markers studies have shown that the lymphoid component is predominantly formed by T-lymphocytes, with a relatively small number of polyclonal B-lymphocytes. On the contrary, other authors found opposite relationship between T and B lymphocytes. These findings supported the concept that lymphoid tissue in Warthin's tumor represented a reactive cellular infiltrate in a pattern similar to that seen in reactive lymph nodes. Similar results have been found in this report; moreover, the hard palate does not usually contain lymphatic tissue. This could support the idea that the lymphoid tissue associated with this case is reactive and a direct origin from the ductal epithelium with secondary lymphocytic infiltration is more likely to occur in this area.