Warthin tumor concomitant with mantle cell lymphoma: a case report and review of literature

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It has a slow growth rate and most frequently occurs in parotid gland. Most patients present with incidental finding of painless masses inferior/anterior to the ear. Besides the epithelial component of the tumor, WT is characteristic of associated lymphoid stroma that is considered benign. While there have been few reports of malignant transformation of lymphoid components in WT, cases of WT concomitant with mantle cell lymphoma (MCL) are extremely rare. To the best of our knowledge two cases have described in English literature. Herein, we report a case of WT concomitant with MCL in a 70-year-old female patient, and emphasize the importance of careful examination of lymphoid stroma in WT so that the possibility of concurrent lymphoma would not be missed. Patient concerns: A 70-year-old Chinese woman with a 40-year history of cigarette smoking presented with a right submaxillary mass for the past year with recent enlargement. Diagnosis: Cervical ultrasound (US) and computed tomography (CT) scans of the neck revealed a well circumscribed mass lesion in the right parotid with a maximum diameter of 3.1 cm. Surgical resection of the mass was performed. Histopathological examination revealed a characteristic double-layers of neoplastic epithelium with prominent lymphoid stroma, suggesting WT. In addition, morphology and immunohistochemistry study confirmed the coexistance of MCL. Thereafter, the final diagnosis of this case was WT concomitant with MCL. Interventions: The patient was staged as stage IVa after clinical assessment. Due to the slow growth of parotid lesions, close observation was decided with periodic clinical and radiological monitoring. Outcomes: Currently, the patient demonstrates a stable disease by clinical evaluation Lessons: To the best of our knowledge, reported cases of WT concomitant with MCL are very rare. This case highlights the importance of a comprehensive assessment of the lymphoid stroma of WT to avoid missed diagnosis of lymphoma component in a collision tumor. Parotid gland Lymph node Warthin tumor Mantle cell lymphoma Hodgkin lymphoma Non-Hodgkin lymphoma Figures Figure 1 Figure 2 1. Introduction Warthin tumor (WT), also known as adenomatoma or lymphopapillary cystadenoma, is the second most common benign tumor of the salivary gland, 1,2 accounting for about 17% of total salivary gland benign tumors, second only to pleomorphic adenoma of the parotid gland. 3 The tumor was first reported by Hildebrad in 1895, and was first described by Warthin in 1929 and named after lymphomatous papillary cystadenoma, hence the name. In 1991, the World Health Organization (WHO) proposed the name Warthin tumor in the new classification of salivary gland tumors, which is still used today. 4 The main feature of WT is that its epithelium is mainly composed of bilayer eosinophilic columnar cells embedded in the lymphoid matrix or lymphoid stroma. The incidence of malignancy in WT is exceedingly low, and in recent years there have been reports of malignant transformation of epithelial components, but lymphomas involving stromal components are extremely rare. Of the cases with lymphomatous involvement reported in the literature, most cases demonstrate a widely disseminated disease. WT with localized mantle cell lymphoma (MCL) is limited to sporadic case reports. Herein, we report a rare collision tumor of WT with MCL. To the best of our knowledge, there have been only two cases of WT with MCL reported in English literature. 5 Since lymphoma cells could potentially be confused with benign stromal components within the tumor, the case highlights the importance of careful examination the lymphoid stroma within WT to avoid diagnostic error. 2. Case presentation A 70-year-old Chinese woman with a 40-year history of cigarette smoking presented to our hospital with one year history of the right submaxillary swelling mass. The patient reported significant enlargement of the mass lesion after tooth extraction. Physical examination showed a painless mass in the area of right parotid without change of skin color or palpable enlargement of regional lymph nodes. The patient denied fever, night sweat and unintentional weight loss. The ultrasonography showed scattered lymph node echoes in bilateral cervical areas, about 13×4mm on the left and 18×5mm on the right. The hypoechoic mass in the right neck, about 25×12mm, showed strong cord-like echo, clear boundary, regular shape, and enhanced blood flow signal. Neck enhanced CT scan showed that the size of the right parotid gland tumor was 2.5cm (anterior and posterior) ×1.2cm(transverse)×3.1cm(cranial); there was no pathological cervical lymphadenopathy; facial nerve function was intact, and the House-Brackmann score was 1 (out of 6). The patient subsequently underwent right superficial parotid gland resection and right sternocleidomastoid myocutaneous flap reconstruction. Facial nerve function returned to normal after surgery, and the complete blood count was within normal range. Gross examination of the right superficial parotid gland specimen showed that brown red oval specimen with lobed surface, sectioning demonstrated a well-defined tan mass with uniform "fish" appearance, measuring 3.5×3.0× 2.5 cm. No tissue necrosis was identified. Microscopical examination showed an intraparenchymal lesion at low magnification that was composed of epithelial components and lymphoid stromal tissue. The epithelioid tissue had a cystic adenoid structure and formed irregular large glandular tubes and sacs that protrude into the lumen in a papillary growth pattern. Lymphocytes in the stroma were diffusely distributed and markedly expanded stromal cores (Fig. 1 A). At a high magnification, the epithelium is composed of bilayers of the cells, with eosinophilic columnar cells in the inner layer and basal cells in the outer layer, both of which were compressed by significantly expanded lymphoid stroma. The lymphoid stroma consisted of small to medium-sized lymphocytes with clumped chromatin, irregular nuclear contours, indistinct nucleolus, and scant cytoplasm. Hyaline vascular wall was noted, and scattered increase in epithelioid histiocytes was appreciable among the stromal lymphoid proliferation (Fig. 1 B). Immunohistochemical analysis showed that the epithelial component was positive for CK and stromal lymphoid component was positive for, CD20, CD43, Cyclin D1, PAX5, SOX11and CD5 (weakly positive) (Fig. 2 ), negative for CD3, CD23. Ki67 proliferation index was estimated to be 70% within stromal lymphoid component and was very low within epithelial component (Fig. 2 H). The histopathologic findings supported the diagnosis of WT concomitant with stromal MCL. 3. Discussion WT is the second most common benign tumor of the salivary glands. 1,2 It occurs almost exclusively in parotid gland, with occasional involvement of regional lymph nodes. It is characterized by multifocal intraglandular and bilateral parotid involvements. 1,6 Its occurrence in other salivary glands is rare. It is relatively common in men aged 50 to 60. 7 Both epithelial and lymphoid stromal components of WT are considered benign, but can undergo malignant transformation, with rate of transformation lower than 1% of the cases. The underlying mechanism of malignant transformation has not been fully understood. 8 The concurrent detection of malignancy in WT was initially documented in 1954. 9 In recent years, there have been reports of WT complicated with cancer, however, WT concomitant with lymphoma is rare. An extensive review of the literature revealed 35 reports of lymphoma concomitant with WT (Table 1). These include totally thirty-nine cases (including the present case) of concurrent WT and lymphoma. Of these, 33 (84.6%) are Non-Hodgkin lymphoma (NHL) and 6 (15.4%) are Hodgkin lymphoma (HL) The cases of WT concomitant with MCL appear extremely rare. Including the present case, MCL comprises only 3 (9%) of 33 total reported NHL cases. 10 Table 1 : Summary of malignant lymphoma concomitant with Warthin tumor. (cHL,classical Hodgkin lymphoma; NLPHL, nodular lymphocyte predominant Hodgkin lymphoma; DLBCL, diffuse large B cell lymphoma; SLL/CLL, small lymphocytic lymphoma/chronic lymphocytic leukemia) Hodgkin lymphoma (n = 6, 15.8%) Frequency (n) CHL, mixed cellularity 2 11,12 CHL, lymphocyte rich 1 13 CHL (Not distinguished) 2 14,15 NLPHL 1 16 Non-Hodgkin lymphoma (n = 32, 84.2%) Follicular lymphoma 14 9,17–26 In situ follicular neoplasia 1 10 DLBCL 6 22,27–31 SLL/CLL 4 8,10,32,33 Mantle cell lymphoma 2 17,34 MALT-type lymphoma 1 35 Peripheral T cell lymphoma 1 36 T cell-lymphoblastic lymphoma 2 37,38 Unclassified 1 20 Of two reported cases with concurrent WT and MCL, one described MCL occurring within the parotid gland in patients with chronic myoepithelial mumps, secondary to an adjacent small WT, but this lymphoma did not occur within the lymphoid stroma of the tumor as seen in the present case. [ 17 ] Furthermore, the diagnosis of that case was based on morphology alone without immunophenotypic support or evidence of t(11; 14) (q13; q32) to confirm it. 34 There is a strong correlation between cigarette smoking and WT according to the literature. 1 Most scholars believe WT arises from intraparotid lymph nodes or ectopic salivary gland tissue remnant in regional lymph nodes. It has been proposed that benzene and/or other substances in tobacco tar be dissolved in saliva being carried retrograde to the glandular parenchyma via parotid duct, thus acting on the parotid duct epithelium and stromal lymphoid tissue, causing metaplasia and neoplastic transformation of both epithelial and stromal lymphoid components. According to the report, 89.1% of the patients with WT had a history of cigarette smoking and almost 90% were found in males. 39 In addition, autoimmune diseases and radiation exposure increase the risk of WT. 1,2,6 All these factors have also been associated with lymphomagenesis. Summary of literature demonstrates history of rapid growth of parotid mass lesions, so was seen in the present case. Therefore, in case of sudden increase in parotid lesion, the possibility of malignant tumor should be raised, and diagnostic procedures, such as radiological evaluation and/or surgical resection with histopathologic examination, should be considered to confirm or rule out the diagnosis. WT itself is a slow-growing benign tumor. Due to its unique morphological features, the histopathologic diagnosis is not difficult, and surgical resection is generally required for therapeutic purpose. WT concomitant with MCL, however, is quite different in terms of clinical presentation, treatment and prognosis. Thereafter, it is critical to carefully evaluate lymphoid stroma, particularly in cases with stromal expansion, and thus rule out the possibility of lymphomatous involvement. Theoretically, because the lymphoid stroma may be part of lymphoid tissue throughout the body, existing lymphomas may involve in WT via homing mechanism and systemic dissemination. Of the reported cases of lymphoma within WT, most patients already had a diagnosis of lymphoma or demonstrated significant lymphadenopathy or systemic involvement by clinical staging subsequently to the diagnosis of WT. While MCL in our case appeared to confine to the stroma of WT without involving adjacent normal parotid glandular parenchyma tissue, clinical staging showed the evidence of lymphomatous involvement in multiple lymph nodes, in keeping with majority of the reported cases. Mantle cell lymphoma (MCL) is mature B-cell lymphoma, with overexpression of cyclin D1 driven by the underlying genomic change, CCND1::IGH fusion. 5 Microscopically, tumor cells were small to medium-sized lymphocytes with scant cytoplasm, irregular nuclear contours, condensed chromatin and inconspicuous nucleolus. In general, MCL is considered a highly invasive and incurable disease. The median survival time of patients is 3–5 years. 34 A high proportion of Ki67-positive cells has been reported as a poor prognostic indicator. In the current case, Ki67 proliferation index was estimated at 70%, predicting aggressiveness of the lymphoma. 34 Patients with MCL require a comprehensive clinical staging, as most patients have developed the tumor with advanced clinical stages (Ann Arbor III/IV) at the time of diagnosis, accompanied by enlargement of multiple lymph nodes, often involving the spleen, liver, and bone marrow. 5 The patient in this case was assigned to stage Iva, in keeping with the high proliferation rate of the tumor by histopathologic evaluation. Nonetheless, given the slow growth of parotid lesions, the case was judged as low to intermediate risk, and close observation was decided for the initial management, with next PET-CT examination scheduled in 6 months. In the examination and diagnosis of patients with WT concomitant with MCL, CT has been preferred as the initial method because it is easily accessible and provides the location, size, and structures involved in the tumor. 31 Magnetic resonance imaging (MRI) may also be useful in the assessment of parotid tumors. 1 A definitive diagnosis relies primarily on histopathological assessment of tissue biopsy, including fine needle aspiration (FNA), core needle biopsy (CNB), and excisional biopsy. FNA plays an important role in the assessment of salivary gland tumors. 40 However, since the diagnostic value of FNA may be affected by insufficient cells and a high false negative rate, CNB is considered to be more superior to FNA in the detection of malignant tumors. However, it remain risky to overlook lymphomatous component within the stroma of WT . 41 We believe that surgical excision with histopathological examination is necessary for a definitive diagnosis of WT and exclusion of lymphomatous involvement. In conclusion, collision tumors characterized by WT concomitant with MCL are very rare, with only 2 cases reported in the English literature to date. WT is a slow-growing benign tumor that can be surgically removed. WT concomitant with MCL, however, is quite different in terms of treatment and prognosis. Since most parotid tumors are benign, malignancy is not usually considered as differential diagnosis and is thus often overlooked, once diagnosis of WT is made. Due to distinct treatment approach, overlooking diagnosis of mantle cell lymphoma would lead to delayed treatment and dismal clinical outcome. Pathologists should carefully and thoroughly examine the stromal lymphoid component of WT to identify potential lymphoma hidden in WT so that adequate management can be delivered timely. Abbreviations WT Warthin tumor MCL Mantle cell lymphoma MRI magnetic resonance imaging US ultrasound CT computed tomography PET/CT positron emission tomography/computed tomography HL Hodgkin lymphoma NHL Non-Hodgkin lymphoma WHO World Health Organization MRI Magnetic resonance imaging Declarations Ethical approval and consent to participate The ethical approval and documentation for a case report was waived with approval of the Institutional Review Board at China Medical University. Consent for publication Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review on request by the Editor-in-Chief of this journal. Acknowledgement: None Authors’ contributions Conceptualization: Hai-Chao Tong，Wan-Lin Zhang, Data curation: Ying-Chun Li， Formal analysis: Hai-Chao Tong，Shuang Ma, Funding acquisition: Lian-He Yang, Investigation: Ying-Chun Li，Shuang Ma, Methodology: Hong-Tao Xu, Project administration: Lian-He Yang, Resources: Le-Yao Li， Software: Le-Yao Li， Supervision: Endi Wang, Validation: Hong-Tao Xu, Visualization: Wan-Lin Zhang, Writing-original draft: Hai-Chao Tong， Writing-review and editing: Lian-He Yang, Tyler Wildes. Funding This study was supported by 1.National Natural Science Foundation of China (Grant No. 81301930 to L.-H. Yang). 2.General project of Education Department of Liaoning Province（Grant No. L2015595 to L.-H. Yang). 3.Key R&D Program Projects of Liaoning Province (Grant No. 2018225085 to L.-H. Yang). 4.Natural Science Fund of Liaoning Province (Grant No. 2019JH3/10300420 and 2019-MS-374 to L.-H. Yang). 5.Natural Science Fund of Liaoning Province (Grant No. 2020-MS-142 to S. Ma). 6.Natural Fund Guidance Plan of Liaoning Provincial Science and Technology Department (Grant No.2019-ZD-0735 to S. Ma). 7. 345 Talent Project of Shengjing Hospital of China Medical University (Grant No. M0364 to S. Ma). 8. Supporting the high-quality development of science and technology funding projects in China Medical University (2023020778-JH2/202, to L.-H. Yang) Declaration of conflict of interest: None. References Gökçe E, Beyhan M. Advanced magnetic resonance imaging findings in salivary gland tumors. World J Radiol Aug. 2022;28(8):256–71. Ginat DT. Imaging of Benign Neoplastic and Nonneoplastic Salivary Gland Tumors. Neuroimaging Clin North Am May. 2018;28(2):159–69. Alsanie I, Rajab S, Cottom H, et al. Distribution and Frequency of Salivary Gland Tumours: An International Multicenter Study. Head neck Pathol Dec. 2022;16(4):1043–54. Teymoortash A. Back to the roots of Warthin’s tumor of the parotid gland. 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Yang","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAAyElEQVRIie3QsQqCUBSA4SOCLmdoPGH4BsENoUn0Va4ENgWNjklwHaIH6C2KwFm5g4vujd43cG2JnIPwtjXcbz4/h3MADONPVTwjjJs8V4POOAJYfd+GC2jlMSDNxF4pkYbw2IoZ6iSxe0qJOxKtixJAEPnLw9QW7EriKNH2EtHvYROsq6mEdmNCEh0vKRhBlZR6CZOI81oQaiZ3xnmKRJZu0na3nlchMkzGJzONW9zifK2fL4qZ2yg1ZJE/mXxgv40bhmEYX7wBtL5BV6nUMKwAAAAASUVORK5CYII=","orcid":"","institution":"the First Hospital and College of Basic Medical Sciences, China Medical University","correspondingAuthor":true,"submittingAuthor":false,"prefix":"","firstName":"Lian-He","middleName":"","lastName":"Yang","suffix":""},{"id":330798261,"identity":"17445c93-9121-42e7-8d90-5a697bd33ec8","order_by":10,"name":"Endi Wang","email":"","orcid":"","institution":"University of Southern California","correspondingAuthor":false,"submittingAuthor":false,"prefix":"","firstName":"Endi","middleName":"","lastName":"Wang","suffix":""}],"badges":[],"createdAt":"2024-07-03 03:39:41","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4677348/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4677348/v1","draftVersion":[],"editorialEvents":[{"content":"https://doi.org/10.1186/s13000-024-01538-6","type":"published","date":"2024-08-24T15:57:26+00:00"}],"editorialNote":"","failedWorkflow":false,"files":[{"id":61182703,"identity":"eb7d6ff8-7b24-47ab-92ac-2f04a73b05a8","added_by":"auto","created_at":"2024-07-26 16:58:32","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":3598160,"visible":true,"origin":"","legend":"\u003cp\u003eA: The lesion is composed of epithelial components and lymphoid stroma. Note the cystic adenoid structure of epithelial tissue that protrudes into the lumen in a papillary pattern and marked expansion of stromal component by diffuse proliferation of lymphocytes (H\u0026amp;E stain, 100×). B: A high magnification demonstrates the epithelial component consists of two layers of cells, with eosinophilic columnar cells in the inner layer and basal cells in the outer layer. The stroma is composed of small to medium-sized lymphocytes without appreciable lymphoid follicles. Note the hyaline vascular change in blood vessels and scattered epithelioid histiocytes in lymphoid stroma, the two histologic features characteristic of mantle cell lymphoma (400×).\u003c/p\u003e","description":"","filename":"Figure1.png","url":"https://assets-eu.researchsquare.com/files/rs-4677348/v1/b914d8f0050411424f531926.png"},{"id":61182704,"identity":"fe8777fa-98bb-4b46-8ecf-fd7b6a14a215","added_by":"auto","created_at":"2024-07-26 16:58:32","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":13028924,"visible":true,"origin":"","legend":"\u003cp\u003eA: CK stain highlights the epithelial component in WT (100×). CK=Cytokeratin. B: CD20 stain is weakly positive in stromal lymphocytes (100×). C-G: CD43 stain, Pax5 stain, Cyclin D1 stain, SOX11stain, and Mum1 stain show positive expression of each antigen in stromal lymphocytes (100×). H: Ki-67 proliferation index is about 70% within stromal lymphocytes (100×).\u003c/p\u003e","description":"","filename":"FIgure2.png","url":"https://assets-eu.researchsquare.com/files/rs-4677348/v1/3f45887ac209376f73229b63.png"},{"id":63300177,"identity":"340c83f5-78cb-4183-88f6-1da5f3dda601","added_by":"auto","created_at":"2024-08-26 16:12:16","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":24149172,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4677348/v1/33125e4c-f2ba-427e-b2f9-986d9c245687.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003eWarthin tumor concomitant with mantle cell lymphoma: a case report and review of literature\u003c/p\u003e","fulltext":[{"header":"1. Introduction","content":"\u003cp\u003eWarthin tumor (WT), also known as adenomatoma or lymphopapillary cystadenoma, is the second most common benign tumor of the salivary gland,\u003csup\u003e1,2\u003c/sup\u003e accounting for about 17% of total salivary gland benign tumors, second only to pleomorphic adenoma of the parotid gland.\u003csup\u003e3\u003c/sup\u003e The tumor was first reported by Hildebrad in 1895, and was first described by Warthin in 1929 and named after lymphomatous papillary cystadenoma, hence the name. In 1991, the World Health Organization (WHO) proposed the name Warthin tumor in the new classification of salivary gland tumors, which is still used today. \u003csup\u003e4\u003c/sup\u003eThe main feature of WT is that its epithelium is mainly composed of bilayer eosinophilic columnar cells embedded in the lymphoid matrix or lymphoid stroma. The incidence of malignancy in WT is exceedingly low, and in recent years there have been reports of malignant transformation of epithelial components, but lymphomas involving stromal components are extremely rare. Of the cases with lymphomatous involvement reported in the literature, most cases demonstrate a widely disseminated disease. WT with localized mantle cell lymphoma (MCL) is limited to sporadic case reports. Herein, we report a rare collision tumor of WT with MCL. To the best of our knowledge, there have been only two cases of WT with MCL reported in English literature.\u003csup\u003e5\u003c/sup\u003e Since lymphoma cells could potentially be confused with benign stromal components within the tumor, the case highlights the importance of careful examination the lymphoid stroma within WT to avoid diagnostic error.\u003c/p\u003e"},{"header":"2. Case presentation","content":"\u003cp\u003eA 70-year-old Chinese woman with a 40-year history of cigarette smoking presented to our hospital with one year history of the right submaxillary swelling mass. The patient reported significant enlargement of the mass lesion after tooth extraction. Physical examination showed a painless mass in the area of right parotid without change of skin color or palpable enlargement of regional lymph nodes. The patient denied fever, night sweat and unintentional weight loss. The ultrasonography showed scattered lymph node echoes in bilateral cervical areas, about 13\u0026times;4mm on the left and 18\u0026times;5mm on the right. The hypoechoic mass in the right neck, about 25\u0026times;12mm, showed strong cord-like echo, clear boundary, regular shape, and enhanced blood flow signal. Neck enhanced CT scan showed that the size of the right parotid gland tumor was 2.5cm (anterior and posterior) \u0026times;1.2cm(transverse)\u0026times;3.1cm(cranial); there was no pathological cervical lymphadenopathy; facial nerve function was intact, and the House-Brackmann score was 1 (out of 6). The patient subsequently underwent right superficial parotid gland resection and right sternocleidomastoid myocutaneous flap reconstruction. Facial nerve function returned to normal after surgery, and the complete blood count was within normal range.\u003c/p\u003e \u003cp\u003eGross examination of the right superficial parotid gland specimen showed that brown red oval specimen with lobed surface, sectioning demonstrated a well-defined tan mass with uniform \"fish\" appearance, measuring 3.5\u0026times;3.0\u0026times; 2.5 cm. No tissue necrosis was identified.\u003c/p\u003e \u003cp\u003eMicroscopical examination showed an intraparenchymal lesion at low magnification that was composed of epithelial components and lymphoid stromal tissue. The epithelioid tissue had a cystic adenoid structure and formed irregular large glandular tubes and sacs that protrude into the lumen in a papillary growth pattern. Lymphocytes in the stroma were diffusely distributed and markedly expanded stromal cores (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eA). At a high magnification, the epithelium is composed of bilayers of the cells, with eosinophilic columnar cells in the inner layer and basal cells in the outer layer, both of which were compressed by significantly expanded lymphoid stroma. The lymphoid stroma consisted of small to medium-sized lymphocytes with clumped chromatin, irregular nuclear contours, indistinct nucleolus, and scant cytoplasm. Hyaline vascular wall was noted, and scattered increase in epithelioid histiocytes was appreciable among the stromal lymphoid proliferation (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eB). Immunohistochemical analysis showed that the epithelial component was positive for CK and stromal lymphoid component was positive for, CD20, CD43, Cyclin D1, PAX5, SOX11and CD5 (weakly positive) (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e), negative for CD3, CD23. Ki67 proliferation index was estimated to be 70% within stromal lymphoid component and was very low within epithelial component (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003eH). The histopathologic findings supported the diagnosis of WT concomitant with stromal MCL.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"3. Discussion","content":"\u003cp\u003eWT is the second most common benign tumor of the salivary glands.\u003csup\u003e1,2\u003c/sup\u003e It occurs almost exclusively in parotid gland, with occasional involvement of regional lymph nodes. It is characterized by multifocal intraglandular and bilateral parotid involvements.\u003csup\u003e1,6\u003c/sup\u003e Its occurrence in other salivary glands is rare. It is relatively common in men aged 50 to 60.\u003csup\u003e7\u003c/sup\u003e Both epithelial and lymphoid stromal components of WT are considered benign, but can undergo malignant transformation, with rate of transformation lower than 1% of the cases. The underlying mechanism of malignant transformation has not been fully understood.\u003csup\u003e8\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eThe concurrent detection of malignancy in WT was initially documented in 1954.\u003csup\u003e9\u003c/sup\u003e In recent years, there have been reports of WT complicated with cancer, however, WT concomitant with lymphoma is rare. An extensive review of the literature revealed 35 reports of lymphoma concomitant with WT (Table\u0026nbsp;1). These include totally thirty-nine cases (including the present case) of concurrent WT and lymphoma. Of these, 33 (84.6%) are Non-Hodgkin lymphoma (NHL) and 6 (15.4%) are Hodgkin lymphoma (HL) The cases of WT concomitant with MCL appear extremely rare. Including the present case, MCL comprises only 3 (9%) of 33 total reported NHL cases.\u003csup\u003e10\u003c/sup\u003e\u003c/p\u003e \u003cp\u003e\u003cstrong\u003eTable 1\u003c/strong\u003e:\u003c/p\u003e\n\u003cp\u003eSummary of malignant lymphoma concomitant with Warthin tumor. (cHL,classical Hodgkin lymphoma; NLPHL, nodular lymphocyte predominant Hodgkin lymphoma; DLBCL, diffuse large B cell lymphoma; SLL/CLL, small lymphocytic lymphoma/chronic lymphocytic leukemia)\u003c/p\u003e\u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"No\" id=\"Taba\" border=\"1\"\u003e \u003ccolgroup cols=\"2\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eHodgkin lymphoma (n\u0026thinsp;=\u0026thinsp;6, 15.8%)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eFrequency (n)\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCHL, mixed cellularity\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003csup\u003e11,12\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCHL, lymphocyte rich\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e13\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCHL (Not distinguished)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003csup\u003e14,15\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNLPHL\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e16\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eNon-Hodgkin lymphoma (n\u0026thinsp;=\u0026thinsp;32, 84.2%)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eFollicular lymphoma\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e14\u003csup\u003e9,17\u0026ndash;26\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eIn situ follicular neoplasia\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e10\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDLBCL\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e6\u003csup\u003e22,27\u0026ndash;31\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSLL/CLL\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e4\u003csup\u003e8,10,32,33\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMantle cell lymphoma\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003csup\u003e17,34\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMALT-type lymphoma\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e35\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePeripheral T cell lymphoma\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e36\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eT cell-lymphoblastic lymphoma\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e2\u003csup\u003e37,38\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eUnclassified\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e1\u003csup\u003e20\u003c/sup\u003e\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003eOf two reported cases with concurrent WT and MCL, one described MCL occurring within the parotid gland in patients with chronic myoepithelial mumps, secondary to an adjacent small WT, but this lymphoma did not occur within the lymphoid stroma of the tumor as seen in the present case.\u003csup\u003e[\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e]\u003c/sup\u003e Furthermore, the diagnosis of that case was based on morphology alone without immunophenotypic support or evidence of t(11; 14) (q13; q32) to confirm it.\u003csup\u003e34\u003c/sup\u003e\u003c/p\u003e \u003cp\u003eThere is a strong correlation between cigarette smoking and WT according to the literature.\u003csup\u003e1\u003c/sup\u003e Most scholars believe WT arises from intraparotid lymph nodes or ectopic salivary gland tissue remnant in regional lymph nodes. It has been proposed that benzene and/or other substances in tobacco tar be dissolved in saliva being carried retrograde to the glandular parenchyma via parotid duct, thus acting on the parotid duct epithelium and stromal lymphoid tissue, causing metaplasia and neoplastic transformation of both epithelial and stromal lymphoid components. According to the report, 89.1% of the patients with WT had a history of cigarette smoking and almost 90% were found in males.\u003csup\u003e39\u003c/sup\u003e In addition, autoimmune diseases and radiation exposure increase the risk of WT.\u003csup\u003e1,2,6\u003c/sup\u003e All these factors have also been associated with lymphomagenesis.\u003c/p\u003e \u003cp\u003eSummary of literature demonstrates history of rapid growth of parotid mass lesions, so was seen in the present case. Therefore, in case of sudden increase in parotid lesion, the possibility of malignant tumor should be raised, and diagnostic procedures, such as radiological evaluation and/or surgical resection with histopathologic examination, should be considered to confirm or rule out the diagnosis.\u003c/p\u003e \u003cp\u003eWT itself is a slow-growing benign tumor. Due to its unique morphological features, the histopathologic diagnosis is not difficult, and surgical resection is generally required for therapeutic purpose. WT concomitant with MCL, however, is quite different in terms of clinical presentation, treatment and prognosis. Thereafter, it is critical to carefully evaluate lymphoid stroma, particularly in cases with stromal expansion, and thus rule out the possibility of lymphomatous involvement.\u003c/p\u003e \u003cp\u003eTheoretically, because the lymphoid stroma may be part of lymphoid tissue throughout the body, existing lymphomas may involve in WT via homing mechanism and systemic dissemination. Of the reported cases of lymphoma within WT, most patients already had a diagnosis of lymphoma or demonstrated significant lymphadenopathy or systemic involvement by clinical staging subsequently to the diagnosis of WT. While MCL in our case appeared to confine to the stroma of WT without involving adjacent normal parotid glandular parenchyma tissue, clinical staging showed the evidence of lymphomatous involvement in multiple lymph nodes, in keeping with majority of the reported cases.\u003c/p\u003e \u003cp\u003eMantle cell lymphoma (MCL) is mature B-cell lymphoma, with overexpression of cyclin D1 driven by the underlying genomic change, \u003cem\u003eCCND1::IGH\u003c/em\u003e fusion.\u003csup\u003e5\u003c/sup\u003e Microscopically, tumor cells were small to medium-sized lymphocytes with scant cytoplasm, irregular nuclear contours, condensed chromatin and inconspicuous nucleolus. In general, MCL is considered a highly invasive and incurable disease. The median survival time of patients is 3\u0026ndash;5 years. \u003csup\u003e34\u003c/sup\u003e A high proportion of Ki67-positive cells has been reported as a poor prognostic indicator. In the current case, Ki67 proliferation index was estimated at 70%, predicting aggressiveness of the lymphoma.\u003csup\u003e34\u003c/sup\u003e\u003c/p\u003e \u003cp\u003ePatients with MCL require a comprehensive clinical staging, as most patients have developed the tumor with advanced clinical stages (Ann Arbor III/IV) at the time of diagnosis, accompanied by enlargement of multiple lymph nodes, often involving the spleen, liver, and bone marrow.\u003csup\u003e5\u003c/sup\u003e The patient in this case was assigned to stage Iva, in keeping with the high proliferation rate of the tumor by histopathologic evaluation. Nonetheless, given the slow growth of parotid lesions, the case was judged as low to intermediate risk, and close observation was decided for the initial management, with next PET-CT examination scheduled in 6 months.\u003c/p\u003e \u003cp\u003eIn the examination and diagnosis of patients with WT concomitant with MCL, CT has been preferred as the initial method because it is easily accessible and provides the location, size, and structures involved in the tumor.\u003csup\u003e31\u003c/sup\u003e Magnetic resonance imaging (MRI) may also be useful in the assessment of parotid tumors. \u003csup\u003e1\u003c/sup\u003e A definitive diagnosis relies primarily on histopathological assessment of tissue biopsy, including fine needle aspiration (FNA), core needle biopsy (CNB), and excisional biopsy. FNA plays an important role in the assessment of salivary gland tumors.\u003csup\u003e40\u003c/sup\u003e However, since the diagnostic value of FNA may be affected by insufficient cells and a high false negative rate, CNB is considered to be more superior to FNA in the detection of malignant tumors. However, it remain risky to overlook lymphomatous component within the stroma of WT .\u003csup\u003e41\u003c/sup\u003e We believe that surgical excision with histopathological examination is necessary for a definitive diagnosis of WT and exclusion of lymphomatous involvement.\u003c/p\u003e \u003cp\u003eIn conclusion, collision tumors characterized by WT concomitant with MCL are very rare, with only 2 cases reported in the English literature to date. WT is a slow-growing benign tumor that can be surgically removed. WT concomitant with MCL, however, is quite different in terms of treatment and prognosis. Since most parotid tumors are benign, malignancy is not usually considered as differential diagnosis and is thus often overlooked, once diagnosis of WT is made. Due to distinct treatment approach, overlooking diagnosis of mantle cell lymphoma would lead to delayed treatment and dismal clinical outcome. Pathologists should carefully and thoroughly examine the stromal lymphoid component of WT to identify potential lymphoma hidden in WT so that adequate management can be delivered timely.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eWT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eWarthin tumor\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMCL\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eMantle cell lymphoma\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMRI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003emagnetic resonance imaging\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eUS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eultrasound\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ecomputed tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003ePET/CT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003epositron emission tomography/computed tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eHL\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eHodgkin lymphoma\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eNHL\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eNon-Hodgkin lymphoma\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eWHO\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eWorld Health Organization\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMRI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eMagnetic resonance imaging\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthical approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe ethical approval and documentation for a case report was waived with approval of the Institutional Review Board at China Medical University.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review on request by the Editor-in-Chief of this journal.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgement: None\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors’ contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eConceptualization: Hai-Chao Tong，Wan-Lin Zhang,\u003c/p\u003e\n\u003cp\u003eData curation: Ying-Chun Li，\u003c/p\u003e\n\u003cp\u003eFormal analysis: Hai-Chao Tong，Shuang Ma,\u003c/p\u003e\n\u003cp\u003eFunding acquisition: Lian-He Yang,\u003c/p\u003e\n\u003cp\u003eInvestigation: Ying-Chun Li，Shuang Ma,\u003c/p\u003e\n\u003cp\u003eMethodology: Hong-Tao Xu,\u003c/p\u003e\n\u003cp\u003eProject administration: Lian-He Yang,\u003c/p\u003e\n\u003cp\u003eResources: Le-Yao Li，\u003c/p\u003e\n\u003cp\u003eSoftware: Le-Yao Li，\u003c/p\u003e\n\u003cp\u003eSupervision: Endi Wang,\u003c/p\u003e\n\u003cp\u003eValidation: Hong-Tao Xu,\u003c/p\u003e\n\u003cp\u003eVisualization: Wan-Lin Zhang,\u003c/p\u003e\n\u003cp\u003eWriting-original draft: Hai-Chao Tong，\u003c/p\u003e\n\u003cp\u003eWriting-review and editing: Lian-He Yang, Tyler Wildes.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study was supported by\u003c/p\u003e\n\u003cp\u003e1.National Natural Science Foundation of China (Grant No. 81301930 to L.-H. Yang).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e2.General project of Education Department of Liaoning Province（Grant No. L2015595 to L.-H. Yang).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e3.Key R\u0026amp;D Program Projects of Liaoning Province (Grant No. 2018225085 to L.-H. Yang).\u003c/p\u003e\n\u003cp\u003e4.Natural Science Fund of Liaoning Province (Grant No. 2019JH3/10300420 and 2019-MS-374 to L.-H. Yang).\u003c/p\u003e\n\u003cp\u003e5.Natural Science Fund of Liaoning Province (Grant No. 2020-MS-142 to S. Ma).\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e6.Natural Fund Guidance Plan of Liaoning Provincial Science and Technology Department (Grant No.2019-ZD-0735 to S. Ma).\u003c/p\u003e\n\u003cp\u003e7. 345 Talent Project of Shengjing Hospital of China Medical University (Grant No. M0364 to S. Ma).\u003c/p\u003e\n\u003cp\u003e8. Supporting the high-quality development of science and technology funding projects in China Medical University (2023020778-JH2/202, to L.-H. Yang)\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDeclaration of conflict of interest:\u003c/strong\u003e None.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eG\u0026ouml;k\u0026ccedil;e E, Beyhan M. Advanced magnetic resonance imaging findings in salivary gland tumors. World J Radiol Aug. 2022;28(8):256\u0026ndash;71.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGinat DT. Imaging of Benign Neoplastic and Nonneoplastic Salivary Gland Tumors. Neuroimaging Clin North Am May. 2018;28(2):159\u0026ndash;69.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAlsanie I, Rajab S, Cottom H, et al. Distribution and Frequency of Salivary Gland Tumours: An International Multicenter Study. Head neck Pathol Dec. 2022;16(4):1043\u0026ndash;54.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTeymoortash A. Back to the roots of Warthin\u0026rsquo;s tumor of the parotid gland. 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Oncol Lett Jul. 2018;16(1):619\u0026ndash;22.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMelato M, Falconieri G, Fanin R, Baccarani M. Hodgkin's disease occurring in a Warthin's tumor: first case report. Pathol Res Pract Oct. 1986;181(5):615\u0026ndash;20.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLiu YQ, Tang QL, Wang LL, Liu QY, Fan S, Li HG. Aug. Concomitant lymphocyte-rich classical Hodgkin's lymphoma and Warthin's tumor. \u003cem\u003eOral surgery, oral medicine, oral pathology and oral radiology.\u003c/em\u003e 2013;116(2):e117-120.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eCozzolino I, Zeppa P, Cuccuru A, Picardi M, Vetrani A, Palombini L. Collision Hodgkin lymphoma and Warthin tumour. Report of a case and review of the literature. Oral Surg. 2009;2:188\u0026ndash;92.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBadve S, Evans G, Mady S, Coppen M, Sloane J. 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Am J Clin Pathol Jan. 2000;113(1):113\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBanik S, Howell JS, Wright DH. Non-Hodgkin's lymphoma arising in adenolymphoma\u0026ndash;a report of two cases. J Pathol Jul. 1985;146(3):167\u0026ndash;77.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eColby TV, Dorfman RF. Malignant lymphomas involving the salivary glands. Pathol Annu. 1979;14:307\u0026ndash;24.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGiardini R, Mastore M. Follicular non Hodgkin's lymphoma in adenolymphoma: report of a case. Tumori Apr. 1990;30(2):212\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGriesser GH, Hansmann ML, Bogman MJ, Pielsticker K, Lennert K. Germinal center derived malignant lymphoma in cystadenolymphoma. Virchows Archiv Pathological Anat histopathology. 1986;408(5):491\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHall G, Tesluk H, Baron S. Lymphoma arising in an adenolymphoma. Hum Pathol Apr. 1985;16(4):424\u0026ndash;7.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMedeiros LJ, Rizzi R, Lardelli P, Jaffe ES. Malignant lymphoma involving a Warthin's tumor: a case with immunophenotypic and gene rearrangement analysis. Hum Pathol Sep. 1990;21(9):974\u0026ndash;7.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMiller R, Yanagihara ET, Dubrow AA, Lukes RJ. Malignant lymphoma in the Warthin's tumor. Report of a case. Cancer Dec. 1982;15(12):2948\u0026ndash;50.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRomero M, Gonz\u0026aacute;lez-Fontal GR, Duarte M, Saavedra C, Henao-Mart\u0026iacute;nez AF. Small clonal B-cell population in the bone marrow as a possible tool in the diagnosis of occult primary parotid lymphoma. Colombia Med (Cali Colombia) Mar. 2016;30(1):59\u0026ndash;62.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eChu CY, Pan SC, Chang KC. EBV-positive diffuse large B-cell lymphoma of the elderly involving Warthin tumor. Pathol Int Dec. 2015;65(12):677\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGorai S, Numata T, Kawada S, Nakano M, Tamaru J, Kobayashi T. Malignant lymphoma arising from heterotopic Warthin's tumor in the neck: case report and review of the literature. Tohoku J experimental Med Jun. 2007;212(2):199\u0026ndash;205.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eOzk\u0026ouml;k G, Taşlı F, Ozsan N, Ozt\u0026uuml;rk R, Postacı H. Diffuse Large B-Cell Lymphoma Arising in Warthin's Tumor: Case Study and Review of the Literature. Korean J Pathol Dec. 2013;47(6):579\u0026ndash;82.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eReiner M, Goldhirsch A, Luscieti PR, Pedrinis E, Kaplan E, Cavalli F. Aug. Warthin's tumor with Sj\u0026ouml;gren's syndrome and non-Hodgkin's lymphoma. \u003cem\u003eEar, nose, \u0026amp; throat journal.\u003c/em\u003e 1979;58(8):345\u0026ndash;350.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGutierrez-Alvarez M, Martinez C, Campollo Lopez AP, Fuentes K, Robles Avi\u0026ntilde;a JA. Warthin's Tumor of the Parotid Gland With Degeneration to Diffuse Large B-cell Lymphoma: A Case Report and Review of Literature. Cureus Mar. 2023;15(3):e36802.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBunker ML, Locker J. Warthin's tumor with malignant lymphoma. DNA analysis of paraffin-embedded tissue. Am J Clin Pathol Mar. 1989;91(3):341\u0026ndash;4.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSaxena A, Memauri B, Hasegawa W. Initial diagnosis of small lymphocytic lymphoma in parotidectomy for Warthin tumour, a rare collision tumour. J Clin Pathol Mar. 2005;58(3):331\u0026ndash;3.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eArcega RS, Feinstein AJ, Bhuta S, Blackwell KE, Rao NP, Pullarkat ST. An unusual initial presentation of mantle cell lymphoma arising from the lymphoid stroma of warthin tumor. Diagn Pathol Dec. 2015;3:10:209.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMarioni G, Marchese-Ragona R, Marino F, et al. MALT-type lymphoma and Warthin's tumour presenting in the same parotid gland. Acta Otolaryngol Apr. 2004;124(3):318\u0026ndash;23.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003ePescarmona E, Perez M, Faraggiana T, Granati L, Baroni CD. Nodal peripheral T-cell lymphoma associated with Warthin's tumour. Histopathology Aug. 2005;47(2):221\u0026ndash;2.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGiaslakiotis K, Androulaki A, Panagoulias G, et al. T cell lymphoblastic lymphoma in parotidectomy for Warthin's tumor: case report and review of the literature. Int J Hematol Apr. 2009;89(3):359\u0026ndash;64.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eXi Y, Ma Y, Yao Z, Qin Y, Zhao M. Warthin tumor complicated with T-lymphoblastic lymphoma: a case report. \u003cem\u003eHua xi kou qiang yi xue za zhi\u0026thinsp;=\u0026thinsp;Huaxi kouqiang yixue zazhi\u0026thinsp;=\u0026thinsp;West\u003c/em\u003e. China J stomatology Dec. 2022;1(6):727\u0026ndash;30.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLee DH, Yoon TM, Lee JK, Lim SC. Surgical treatment strategy in Warthin tumor of the parotid gland. Brazilian J Otorhinolaryngol Sep-Oct. 2019;85(5):546\u0026ndash;50.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKrishnan V, Victor AR, Bose S, Bakkar R. Lymphoid cell rich fine-needle aspirations of the salivary gland: What is the risk of malignancy? CytoJournal. 2023;20:11.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eRyoo HJ, Lim JS, Kim MC. Primary non-Hodgkin lymphoma of the parotid gland: a case report. Archives Craniofac Surg Jun. 2022;23(3):125\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"diagnostic-pathology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"dpat","sideBox":"Learn more about [Diagnostic Pathology](http://diagnosticpathology.biomedcentral.com)","snPcode":"13000","submissionUrl":"https://submission.nature.com/new-submission/13000/3","title":"Diagnostic Pathology","twitterHandle":"@OncoBioMed","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"Parotid gland, Lymph node, Warthin tumor, Mantle cell lymphoma, Hodgkin lymphoma, Non-Hodgkin lymphoma","lastPublishedDoi":"10.21203/rs.3.rs-4677348/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4677348/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eRationale:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWarthin tumor (WT) is the second most common benign tumor in salivary gland. It has a slow growth rate and most frequently occurs in parotid gland. Most patients present with incidental finding of painless masses inferior/anterior to the ear. Besides the epithelial component of the tumor, WT is characteristic of associated lymphoid stroma that is considered benign. While there have been few reports of malignant transformation of lymphoid components in WT, cases of WT concomitant with mantle cell lymphoma (MCL) are extremely rare. To the best of our knowledge two cases have described in English literature. Herein, we report a case of WT concomitant with MCL in a 70-year-old female patient, and emphasize the importance of careful examination of lymphoid stroma in WT so that the possibility of concurrent lymphoma would not be missed.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003ePatient concerns:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eA 70-year-old Chinese woman with a 40-year history of cigarette smoking presented with a right submaxillary mass for the past year with recent enlargement.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDiagnosis:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eCervical ultrasound (US) and computed tomography (CT) scans of the neck revealed a well circumscribed mass lesion in the right parotid with a maximum diameter of 3.1 cm. Surgical resection of the mass was performed. Histopathological examination revealed a characteristic double-layers of neoplastic epithelium with prominent lymphoid stroma, suggesting WT. In addition, morphology and immunohistochemistry study confirmed the coexistance of MCL. Thereafter, the final diagnosis of this case was WT concomitant with MCL.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eInterventions:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe patient was staged as stage IVa after clinical assessment. Due to the slow growth of parotid lesions, close observation was decided with periodic clinical and radiological monitoring.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eOutcomes:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eCurrently, the patient demonstrates a stable disease by clinical evaluation\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eLessons:\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eTo the best of our knowledge, reported cases of WT concomitant with MCL are very rare. This case highlights the importance of a comprehensive assessment of the lymphoid stroma of WT to avoid missed diagnosis of lymphoma component in a collision tumor.\u003c/p\u003e","manuscriptTitle":"Warthin tumor concomitant with mantle cell lymphoma: a case report and review of literature","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-07-26 16:58:27","doi":"10.21203/rs.3.rs-4677348/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2024-08-01T16:39:31+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-07-29T10:07:54+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-07-29T06:04:38+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"287745316219142517718201273155038337363","date":"2024-07-29T04:16:18+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-07-25T11:24:42+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-07-22T11:38:44+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-07-22T02:53:34+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"14046037327531020622559493120292177337","date":"2024-07-15T14:54:50+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"123563004216483015538618297366635957310","date":"2024-07-15T14:51:38+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"44623986450087325679547048918530455665","date":"2024-07-15T14:14:06+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"23613932346841567991523123351754432765","date":"2024-07-14T06:45:00+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"265227282485675731831247265080380585040","date":"2024-07-13T16:28:51+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"162401796177498309784936563928679432165","date":"2024-07-13T14:47:21+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"129607329523500595332786022415284552224","date":"2024-07-13T14:47:17+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"67962348828940094275959072735018154949","date":"2024-07-13T14:03:47+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-07-13T14:02:12+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-07-03T05:36:45+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2024-07-03T04:26:24+00:00","index":"","fulltext":""},{"type":"submitted","content":"Diagnostic Pathology","date":"2024-07-03T03:38:21+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"diagnostic-pathology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"dpat","sideBox":"Learn more about [Diagnostic Pathology](http://diagnosticpathology.biomedcentral.com)","snPcode":"13000","submissionUrl":"https://submission.nature.com/new-submission/13000/3","title":"Diagnostic Pathology","twitterHandle":"@OncoBioMed","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"01a91bc0-0aea-4f7d-9617-b0350b602921","owner":[],"postedDate":"July 26th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2024-08-26T16:02:03+00:00","versionOfRecord":{"articleIdentity":"rs-4677348","link":"https://doi.org/10.1186/s13000-024-01538-6","journal":{"identity":"diagnostic-pathology","isVorOnly":false,"title":"Diagnostic Pathology"},"publishedOn":"2024-08-24 15:57:26","publishedOnDateReadable":"August 24th, 2024"},"versionCreatedAt":"2024-07-26 16:58:27","video":"","vorDoi":"10.1186/s13000-024-01538-6","vorDoiUrl":"https://doi.org/10.1186/s13000-024-01538-6","workflowStages":[]},"version":"v1","identity":"rs-4677348","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4677348","identity":"rs-4677348","version":["v1"]},"buildId":"cTy_lsJlmDsVRNrSptgXS","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}