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Vu KV, Trong NV, Khuyen NT, Huyen Nga D, Anh H, Tien Trung N, Trung Thong P, Minh Duc N. Synchronous rectal adenocarcinoma and intestinal mantle cell lymphoma: A case report. World J Clin Cases 2023; 11:5772-5779. [PMID: 37727717 PMCID: PMC10506007 DOI: 10.12998/wjcc.v11.i24.5772] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2023] [Revised: 06/18/2023] [Accepted: 07/11/2023] [Indexed: 08/24/2023] Open
Abstract
BACKGROUND Mantle cell lymphoma (MCL) of the gastrointestinal tract is a rare malignancy, accounting for about 0.2% of malignant colorectal tumors. MCL synchronous with rectal adenocarcinoma is extremely rare. We know of only a few cases reported in the literature. We describe the case of a patient with synchronous rectal adenocarcinoma and intestinal MCL. CASE SUMMARY A 63-year-old man was admitted to our hospital due to abdominal pain and hematochezia over the past month. The patient was diagnosed with middle rectal cancer cT2N0M0 and underwent surgery. However, we found a large tumor in the small intestine during surgery. The patient underwent total mesorectal excision for rectal cancer and resectioning of the ileal segment containing the large mass. Pathology and immunohistochemistry revealed the presence of both rectal adenocarcinoma and pathognomonic MCL stage IIE presenting as multiple lymphomatous polyposis. The patient subsequently underwent RDHAP/RCHOP chemotherapy and was maintained with rituximab. A Positron Emission Tomography and Computed Tomography (PET/CT) scan showed that the disease responded well to treatment without tumor-increased metabolism in the gastrointestinal tract. CONCLUSION Synchronous rectal adenocarcinoma and intestinal MCL presenting as multiple lymphomatous polyposis are extremely rare. MCL is often discovered fortuitously when rectal cancer is diagnosed. The coexistence of these tumors poses treatment challenges.
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Affiliation(s)
- Kim-Van Vu
- Department of General Surgery, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
- Department of Practical Surgery, Hanoi Medical University, Ha Noi 100000, Viet Nam
| | - Nguyen-Van Trong
- Department of Oncology, Hanoi Medical University, Ha Noi 100000, Viet Nam
| | - Nguyen-Thi Khuyen
- Pathology and Molecular Biology Center, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
| | - Do Huyen Nga
- Department of Hematologic Oncology, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
| | - Hoang Anh
- Department of General Surgery, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
- Department of Practical Surgery, Hanoi Medical University, Ha Noi 100000, Viet Nam
| | - Nguyen Tien Trung
- Department of General Surgery, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
| | - Pham Trung Thong
- Department of General Surgery, Vietnam National Cancer Hospital, Ha Noi 100000, Viet Nam
| | - Nguyen Minh Duc
- Department of Radiology, Pham Ngoc Thach University of Medicine, Ho Chi Minh City 700000, Viet Nam
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Ali K, Habib MB, Alloush F, Yassin MA. Mantle cell lymphoma presenting with severe upper gastrointestinal bleeding: A case report. Medicine (Baltimore) 2022; 101:e29236. [PMID: 35687772 PMCID: PMC9276165 DOI: 10.1097/md.0000000000029236] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/14/2022] [Accepted: 04/29/2022] [Indexed: 01/04/2023] Open
Abstract
INTRODUCTION Although it usually involves extranodal sites such as the gastrointestinal tract in more than 80% of cases, mantle cell lymphoma is considered a rare cause of gastrointestinal bleeding, especially severe and life-threatening bleeding. PATIENT CONCERN A 60-year-old man with peptic ulcer disease, who presented with severe upper gastrointestinal (GI) bleeding and large gastric ulcer. DIAGNOSIS Primary gastric mantle cell lymphoma. INTERVENTIONS He was treated conservatively with blood transfusion and started on Traneximic acid for 3 days. Then, the patient underwent urgent hemostatic radiotherapy. OUTCOMES The patient became stable and kept in the hospital for monitoring with a definite diagnosis of stage IV Mantle cell lymphoma is made. CONCLUSION Mantle cell lymphoma should be kept in mind when assessing massive upper GI bleeding, as an unusual cause of bleeding gastric ulcer, given that bleeding is an uncommon presenting feature of GI lymphoma.
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Affiliation(s)
- Khaled Ali
- Community Medicine Department, Hamad Medical Corporation, Doha, Qatar
| | - Mhd Baraa Habib
- Department of Internal Medicine, Hamad Medical Corporation, Doha, Qatar
| | - Ferial Alloush
- Pathology Department, Hamad Medical Corporation, Doha, Qatar
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3
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Sheng JQ, Liu L, Liu C, Li PY. Mantle cell lymphoma with late onset gyrus-like colorectal infiltration: A rare case and literature review. Shijie Huaren Xiaohua Zazhi 2018; 26:137-142. [DOI: 10.11569/wcjd.v26.i2.137] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Mantle cell lymphoma (MCL), characterized by t (11; 14) (q13; q32) translocation and cyclin D1 overexpression, is still a challenging subtype of non-Hodgkin's lymphoma. The majority of MCL cases are discovered in male elderly people and in advanced stages with extranodal infiltration especially the gastrointestinal tract. Multiple lymphomatous polyposis is a typical manifestation of MCL infiltrating the gastrointestinal tract, and the colon is the most commonly involved site. In this paper, we report a case of MCL with late onset colorectal infiltration in a 53-year-old female patient presenting with gyrus-like thickened mucosal folds instead of multiple polyps as revealed by colonoscopic examination. Remarkably, the patient developed colorectal infiltration presenting with intermittent bloody stool after being diagnosed with classical MCL for two years and treated with several cycles of combined chemotherapy. Immunohistochemical staining showed that the lymphoid infiltrate in the colorectal mucosa was positive for cyclin D1, CD5, CD19, CD20, CD21, CD43, CD79a, Bcl-2, PAX-5, and SOX11 (scattered), but negative for CD2, CD3, CD7, CD10, CD23, Bcl-6, c-myc, and Mum-1. The Ki-67 proliferation index was around 15%.
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Affiliation(s)
- Jia-Qi Sheng
- Department of Gastroenterology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei Province, China
| | - Lian Liu
- Department of Gastroenterology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei Province, China
| | - Cong Liu
- Department of Gastroenterology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei Province, China
| | - Pei-Yuan Li
- Department of Gastroenterology, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei Province, China
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Medlicott SAC, Brown HA, Roland B, Beck PL, Auer I, Mansoor A. Multiple Lymphomatous Diverticulosis and Comorbid Chronic Lymphocytic Leukemia: Novel Manifestations of Ileocolic Mantle Cell Lymphoma. Int J Surg Pathol 2016; 15:408-13. [PMID: 17913952 DOI: 10.1177/1066896907302372] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
Mantle cell lymphoma (MCL) has tropism for the gastrointestinal tract (GIT) identifiable as multiple polyps and mass lesions throughout the GIT. We describe 2 novel manifestations of MCL. A 60-year-old woman with known chronic lymphocytic leukemia (CLL) had an exophytic mass of the appendiceal orifice. Multiple polypoid masses of the distal ileum were identified in the right hemicolectomy specimen (multiple lymphomatous polyposis). Ancillary studies confirmed the coexistence of the 2 independent lymphoproliferative disorders. A 69-year-old man had recurrent urinary tract infections and pneumatouria caused by a colovesicular fistula complicating diverticulosis coli. Segmental resections of the sigmoid and ileocecum confirmed diverticulosis of the left and right colon. Histology identified infiltrates of MCL confined to the penetrating aspects of colonic diverticula. MCL has not been documented to coexist with CLL. An invaginating morphology of lymphoma, multiple lymphomatous diverticulosis is also a novel presentation. These 2 scenarios expand MCL's known manifestations within the GIT.
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MESH Headings
- Aged
- Biomarkers, Tumor/metabolism
- Bone Marrow Cells/pathology
- Colonic Neoplasms/metabolism
- Colonic Neoplasms/pathology
- Colonic Neoplasms/surgery
- Combined Modality Therapy
- Diverticulum/complications
- Diverticulum/metabolism
- Diverticulum/pathology
- Female
- Humans
- Ileal Neoplasms/metabolism
- Ileal Neoplasms/pathology
- Ileal Neoplasms/surgery
- In Situ Hybridization, Fluorescence
- Intestinal Fistula/complications
- Intestinal Fistula/pathology
- Leukemia, Lymphocytic, Chronic, B-Cell/metabolism
- Leukemia, Lymphocytic, Chronic, B-Cell/pathology
- Leukemia, Lymphocytic, Chronic, B-Cell/therapy
- Lymphoma, Mantle-Cell/metabolism
- Lymphoma, Mantle-Cell/pathology
- Lymphoma, Mantle-Cell/therapy
- Male
- Middle Aged
- Neoplasms, Multiple Primary/metabolism
- Neoplasms, Multiple Primary/pathology
- Neoplasms, Multiple Primary/therapy
- Treatment Outcome
- Urinary Bladder Fistula/complications
- Urinary Bladder Fistula/pathology
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Affiliation(s)
- Shaun A C Medlicott
- Department of Laboratory Medicine, University of Calgary, Calgary, Alberta, Canada.
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5
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Aiman S, Chakrapani A, Sawaimoon S, Sen S, Chandy M, Chatterjee S. Multiple lymphomatous polyposis: Characteristic endoscopic features. Indian J Gastroenterol 2015; 34:87. [PMID: 25200696 DOI: 10.1007/s12664-014-0497-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Affiliation(s)
- Shah Aiman
- Department of Gastroenterology, Tata Medical Center, Kolkata, 700 156, India
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6
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A colonization of a different kind. Indian J Hematol Blood Transfus 2014; 30:250-4. [PMID: 25332591 DOI: 10.1007/s12288-014-0347-7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/08/2013] [Accepted: 01/27/2014] [Indexed: 10/25/2022] Open
Abstract
An unusual morphological pattern in the lymph node can at times, pose a diagnostic problem. We report a case of a 55 year old male whose cervical lymph node biopsy showed an unusual pattern of follicular colonization by T-lymphoblasts. The interfollicular area showed a diffuse infiltrate of Langerhans cells. A diagnosis of a T lymphoblastic lymphoma coexisting with Langerhans cell histiocytosis like lesion was made, keeping in mind lack of clinical evidence for the latter.
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7
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Xu XJ, Wu SM. Multiple lymphomatous polyposis of the gastrointestinal tract: report of three cases and literature review. J Dig Dis 2012; 13:649-53. [PMID: 23134448 DOI: 10.1111/j.1751-2980.2012.00635.x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Affiliation(s)
- Xiao Jing Xu
- Division of Gastroenterology and Hepatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai Institute of Digestive Disease, Shanghai, China
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8
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Kim ES, Goh PG, Kim YJ, Lee SY, Moon HS, Sung JK, Lee BS, Jeong HY. [A case of mantle cell lymphoma with meningioma]. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2012; 60:56-60. [PMID: 22832802 DOI: 10.4166/kjg.2012.60.1.56] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/03/2022]
Abstract
Mantle cell lymphoma (MCL) is an uncommon type of gastrointestinal lymphoma. MCL is a distinct subtype of B-cell non-Hodgkin lymphomas. The major subtype of MCL is characterized by the presence of multiple lymphomatous polyposis (MLP), in which multiple polyps are observed along the gastrointestinal tract. The malignant cells express pan B-cell marker and the T-cell marker cluster of differentiation 5. The chromosomal translocation t(11;14)(q13;q32) that causes cyclin D1 overexpression is commonly observed on the cytogenetic analysis of MCL. Survival improvement has recently been achieved for patient with MCL by the successful introduction of monoclonal antibodies and dose-intensified approaches for treatment, including autologous stem cell transplantation strategies. Some reports suggest that there is an increased incidence of second malignancies in patients with MCL or lymphoma. We report a case of MCL involving the colon; the patient was a 60-year-old man who complained of low abdominal discomfort during defecation. During the workup, a meningioma was unexpectedly discovered. On analysis, the tumor was found to be a t(11;14)-negative and non-MLP-type MCL.
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Affiliation(s)
- Eui Sik Kim
- Department of Internal Medicine, Chungnam National University College of Medicine, Daejeon, Korea
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9
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Burke JS. Lymphoproliferative disorders of the gastrointestinal tract: a review and pragmatic guide to diagnosis. Arch Pathol Lab Med 2011; 135:1283-97. [PMID: 21970484 DOI: 10.5858/arpa.2011-0145-ra] [Citation(s) in RCA: 45] [Impact Index Per Article: 3.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
CONTEXT The gastrointestinal tract is the most common site of extranodal lymphomas. Although all histologic categories of malignant lymphoma develop in the gastrointestinal tract, large B-cell lymphomas predominate, followed by extranodal marginal zone lymphomas of mucosa-associated lymphoid tissue (MALT) type; the latter is especially prevalent in stomach. The acceptance of extranodal marginal zone lymphoma of MALT type as a clinicopathologic entity has reduced the number of cases that formerly were interpreted as florid lymphoid hyperplasia ("pseudolymphoma"). Nonetheless, the distinction of lymphoid hyperplasia from a lymphoma of MALT type in small biopsy specimens remains problematic. OBJECTIVE To assess the relevant morphologic, immunologic, molecular, and genetic properties of gastrointestinal lymphomas and to present a feasible tactic for diagnosis, expressly for small biopsy specimens. DATA SOURCES Case-derived material and literature review using PubMed (National Library of Medicine). CONCLUSIONS Most gastrointestinal lymphomas are readily amenable to an unqualified diagnosis, primarily those cases consisting of monomorphic large cells whether of B- or T-cell lineage, including cases associated with enteropathy. Diagnosis for infiltrates dominated by small lymphocytes remains taxing, as the differential diagnosis embraces not only MALT lymphoma and lymphoid hyperplasia but also mantle cell lymphoma, follicular lymphoma, and chronic lymphocytic leukemia/small lymphocytic lymphoma. Adherence to strict morphologic criteria is the standard for diagnosis, but these criteria should be augmented by immunologic studies together with judicious use of molecular techniques to determine clonality. In establishing a diagnosis of gastric marginal zone lymphoma of MALT type, determination of t(11;18)(q21;q21) status may be required since this translocation has clinical ramifications.
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Affiliation(s)
- Jerome S Burke
- Department of Pathology, Alta Bates Summit Medical Center, Berkeley, California 94705, USA.
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10
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Sander B. Mantle cell lymphoma: recent insights into pathogenesis, clinical variability, and new diagnostic markers. Semin Diagn Pathol 2011; 28:245-55. [DOI: 10.1053/j.semdp.2011.02.010] [Citation(s) in RCA: 27] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
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11
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Yamamoto S, Nakase H, Yamashita K, Matsuura M, Takada M, Kawanami C, Chiba T. Gastrointestinal follicular lymphoma: review of the literature. J Gastroenterol 2010; 45:370-88. [PMID: 20084529 DOI: 10.1007/s00535-009-0182-z] [Citation(s) in RCA: 77] [Impact Index Per Article: 5.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/09/2009] [Accepted: 11/23/2009] [Indexed: 02/04/2023]
Abstract
Gastrointestinal follicular lymphoma (GI-FL) is a relatively rare disease, accounting for only 1%-3.6% of gastrointestinal non-Hodgkin's lymphoma. Although the duodenum and terminal ileum are considered to be the most common sites of origin, the development of wireless capsule endoscopy and double-balloon enteroscopy has increased the detection of GI-FL in every part of the small intestine. Approximately 70% of patients with GI-FL are estimated to have multiple lesions throughout the entire gastrointestinal tract. FL is a low-grade lymphoma that usually develops very slowly. If the lymphoma causes no symptoms, immediate treatment may not be necessary. Standard therapy has not yet been established for GI-FL, but chemotherapy, radiotherapy, monoclonal antibody therapy, or a combination of these therapies, is sometimes performed based on the therapeutic regimens for nodal FL. Regimens including conventional chemotherapy with rituximab, which achieve high response rates in nodal FL, are commonly used for GI-FL. The long-term clinical outcome of GI-FL is unclear. The results of a few series on the long-term outcomes of patients with GI-FL treated with conventional therapy indicate a median relapse-free time ranging from 31 to 45 months. On the other hand, in patients with GI-FL who were followed without treatment, the median time to disease progression was 37.5 months. Thus, whether to initiate aggressive therapy or whether to continue watchful waiting in patients with GI-FL is a critically important decision. Ongoing research on biomarkers to guide individualized GI-FL therapy may provide invaluable information that will lead to the establishment of a standard therapeutic regimen.
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Affiliation(s)
- Shuji Yamamoto
- Department of Gastroenterology and Hepatology, Graduate School of Medicine, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto, 606-8507, Japan
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12
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Ruskoné-Fourmestraux A, Audouin J. Primary gastrointestinal tract mantle cell lymphoma as multiple lymphomatous polyposis. Best Pract Res Clin Gastroenterol 2010; 24:35-42. [PMID: 20206107 DOI: 10.1016/j.bpg.2009.12.001] [Citation(s) in RCA: 52] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/14/2009] [Revised: 11/27/2009] [Accepted: 12/21/2009] [Indexed: 01/31/2023]
Abstract
Primary gastrointestinal involvement of mantle cell lymphoma (MCL) is rare with a frequency reported between 4 and 9% of all gastrointestinal B-cell non-Hodgkin lymphomas. It was first described and so-called as multiple lymphomatous polyposis (MLP). Its clinical presentation is usually characteristic, with multiple lymphomatous polyps involving several digestive tract segments and a marked tendency towards extra-intestinal spread. The constant and typical phenotypic features of the small cleaved tumour cells, characterised as CD20+, CD5+ CD23- with a t(11;14) (q13;q32) and cyclin D1 overexpression on immunochemistry, allow MLP to be considered as the gastrointestinal counterpart of peripheral nodal MCL. They both share a very poor outcome. Response to intensive chemotherapy regimens usually results in regression of macroscopic and sometimes microscopic lesions but remissions are short and median survival from 3 to 4 years. Prognosis has been significantly improved since in younger patients, intensive front-line immunochemotherapy with autologous stem cell transplantation has been proposed. Earlier diagnosis with further studies integrating novel agents are still required to determine the optimal treatment with less toxicity.
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13
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Yang SF, Liao YL, Kuo SY, Ye H, Lin SF, Chen FM, Chen CY, Chuang SS. Primary intestinal diffuse large B-cell lymphoma presenting as multiple lymphomatous polyposis. Leuk Lymphoma 2009; 50:1219-22. [PMID: 19557644 DOI: 10.1080/10428190902974247] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
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14
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Kella VKN, Constantine R, Parikh NS, Reed M, Cosgrove JM, Abo SM, King S. Mantle cell lymphoma of the gastrointestinal tract presenting with multiple intussusceptions--case report and review of literature. World J Surg Oncol 2009; 7:60. [PMID: 19646237 PMCID: PMC2732623 DOI: 10.1186/1477-7819-7-60] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/21/2009] [Accepted: 07/31/2009] [Indexed: 01/16/2023] Open
Abstract
Background Mantle cell lymphoma (MCL) is an aggressive type of B-cell non-Hodgkin's lymphoma that originates from small to medium sized lymphocytes located in the mantle zone of the lymph node. Extra nodal involvement is present in the majority of cases, with a peculiar tendency to invade the gastro-intestinal tract in the form of multiple lymphomatous polyposis. MCL can be accurately diagnosed with the use of the highly specific marker Cyclin D1. Few cases of mantle cell lymphoma presenting with intussuception have been reported. Here we present a rare case of multiple intussusceptions caused by mantle cell lymphoma and review the literature of this disease. Case presentation A 68-year-old male presented with pain, tenderness in the right lower abdomen, associated with nausea and non-bilious vomiting. CT scan of abdomen revealed ileo-colic intussusception. Laparoscopy confirmed multiple intussusceptions involving ileo-colic and ileo-ileal segments of gastrointestinal tract. A laparoscopically assisted right hemicolectomy and extended ileal resection was performed. Postoperative recovery was uneventful. The histology and immuno-histochemistry of the excised small and large bowel revealed mantle cell lymphoma with multiple lymphomatous polyposis and positivity to Cyclin D1 marker. The patient was successfully treated with Rituximab-CHOP chemotherapy and remains in complete remission at one-year follow-up. Conclusion This is a rare case of intestinal lymphomatous polyposis due to mantle cell lymphoma presenting with multiple small bowel intussusceptions. Our case highlights laparoscopic-assisted bowel resection as a potential and feasible option in the multi-disciplinary treatment of mantle cell lymphoma.
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Affiliation(s)
- Venkata K N Kella
- Department of Surgery and Oncology, Bronx-Lebanon Hospital Center, Bronx, New York, USA.
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15
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Hsieh Y, Lin C, Tsao C, Hsieh P, Tzeng C, Chuang S. Aberrant expression of CD19 and CD43 in a patient with therapy-related acute myeloid leukemia and a history of mantle cell lymphoma. Kaohsiung J Med Sci 2009; 25:389-94. [PMID: 19605331 PMCID: PMC11917950 DOI: 10.1016/s1607-551x(09)70532-7] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/06/2009] [Accepted: 04/06/2009] [Indexed: 11/28/2022] Open
Abstract
Mantle cell lymphoma (MCL) is an aggressive B cell lymphoma with frequent involvement of the gastrointestinal tract and peripheral blood (PB). In addition to the B cell markers, the neoplastic cells express CD5 and CD43. In patients with a prior history of MCL with PB involvement, the appearance of leukemic cells after chemotherapy usually heralds a relapse, particularly if the leukemic cells express B cell markers and CD43. We report a patient with MCL who presented with multiple lymphomatous polyposis of the intestine. The staging procedures revealed the involvement of lymph nodes, bone marrow and PB. Three years after chemotherapy, thrombocytopenia with the appearance of rare leukemic cells in the PB was noted. Leukemic cells obtained from bone marrow aspirate expressed CD19 and CD43, suggesting a relapse. Detailed cytomorphological and immunophenotypic studies unveiled the myeloid nature of these leukemic cells, and a diagnosis of therapy-related acute myeloid leukemia was made. This case illustrates the importance of morphologic examination and performing a complete antibody panel in the diagnosis of a suspected relapse in patients with a prior history of lymphoma.
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MESH Headings
- Antigens, CD19/immunology
- Flow Cytometry
- Gene Expression Regulation, Neoplastic
- Humans
- Leukemia, Myeloid, Acute/etiology
- Leukemia, Myeloid, Acute/immunology
- Leukemia, Myeloid, Acute/pathology
- Leukosialin/immunology
- Lymphoma, Mantle-Cell/immunology
- Lymphoma, Mantle-Cell/pathology
- Male
- Middle Aged
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Affiliation(s)
- Yen‐Chuan Hsieh
- Department of Pathology, Chi‐Mei Medical Center, Tainan, Taipei, Taiwan
| | - Chien‐Liang Lin
- Division of Hemato‐Oncology, Department of Internal Medicine, Lioying Chi‐Mei Hospital, Tainan, Taipei, Taiwan
| | - Chao‐Jung Tsao
- Division of Hemato‐Oncology, Department of Internal Medicine, Lioying Chi‐Mei Hospital, Tainan, Taipei, Taiwan
| | - Pin‐Pen Hsieh
- Department of Pathology, Veterans General Hospital, Kaohsiung, Taipei, Taiwan
- Center for General Education, Yuh‐Ing Junior College of Health Care and Management, Kaohsiung, Taipei, Taiwan
| | | | - Shih‐Sung Chuang
- Department of Pathology, Chi‐Mei Medical Center, Tainan, Taipei, Taiwan
- Department of Pathology, Taipei Medical University, Taipei, Taiwan
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17
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Liao MT, Cheng MF, Chang WC, Wu YC, Lee HS, Tsai SH. Duodenal Mantle Cell Lymphoma in a Patient with Advanced Sigmoid Adenocarcinoma. South Med J 2009; 102:429-31. [DOI: 10.1097/smj.0b013e31819bc4b0] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
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18
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Sztarkier I, Levy I, Walfisch S, Delgado J, Benharroch D. Mantle cell lymphoma in a tubular adenoma: unusual presentation with synchronous colonic carcinoma. Ann Diagn Pathol 2009; 13:47-49. [PMID: 19118782 DOI: 10.1016/j.anndiagpath.2007.05.017] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
An 80-year-old man underwent sigmoidectomy for adenocarcinoma. Six months later, after a near-syncope incident, pancytopenia was detected in the absence of occult blood in the stools. A bone marrow biopsy showed malignant lymphoma, suggestive of mantle cell lymphoma (MCL). Colonoscopy at this time revealed 3 colonic tubular adenomas. Reassessment of the histology of the colonic polyps and appropriate immunohistochemical stains showed that the lamina propria of one of the tubular adenomas was infiltrated by MCL. Reexamination of the sections taken at the time of the original sigmoidectomy showed MCL in 2 of the regional lymph nodes removed at that time, but no evidence of lymphoma in the colon was found. To our knowledge, this is the fifth reported case of synchronous occurrence of intestinal MCL and colonic carcinoma and the first report of MCL presenting in a tubular adenoma of the colon.
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Affiliation(s)
- Ignacio Sztarkier
- Institute of Pathology, Soroka University Medical Center, Beer-Sheva, Israel
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19
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Hokama A, Tomoyose T, Yamamoto YI, Watanabe T, Hirata T, Kinjo F, Kato S, Ohshima K, Uezato H, Takasu N, Fujita J. Adult T-cell leukemia/lymphoma presenting multiple lymphomatous polyposis. World J Gastroenterol 2008; 14:6584-8. [PMID: 19030219 PMCID: PMC2773353 DOI: 10.3748/wjg.14.6584] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Multiple lymphomatous polyposis (MLP) is an unusual form of non-Hodgkin’s lymphoma characterized by polyps throughout the gastrointestinal tract. It has been reported that most MLP are observed in cases with mantle cell lymphoma of B-cell type. We herein present a case of a 66-year-old man with adult T-cell leukemia/lymphoma (ATLL). Colonoscopy revealed MLP throughout the colon and histopathological findings of ATLL cell infiltration. The patient died despite combination of chemotherapy. The literature of manifestations of colonic involvement of ATLL is reviewed and the importance of endoscopic evaluation to differentiate ATLL intestinal lesions from opportunistic infectious enterocolitis is discussed.
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Nonpolypoid Intestinal Mantle Cell Lymphoma Resembling MALT Lymphoma with Successful Response to Rituximab. South Med J 2008; 101:1168-9. [DOI: 10.1097/smj.0b013e3181800afd] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
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21
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Lee JA, Taxier MS, Nicely CJ. Electronic clinical challenges and images in GI. Lymphomatous polyposis. Gastroenterology 2008; 135:e1-3. [PMID: 18619969 DOI: 10.1053/j.gastro.2008.06.041] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Affiliation(s)
- Joo Ahn Lee
- Departments of Internal Medicine, Gastroenterology, and Pathology, Riverside Methodist Hospital, Columbus, Ohio, USA
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22
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Hirata N, Tominaga K, Ohta K, Kadouchi K, Okazaki H, Tanigawa T, Shiba M, Watanabe T, Fujiwara Y, Nakamura S, Oshitani N, Higuchi K, Arakawa T. A case of mucosa-associated lymphoid tissue lymphoma forming multiple lymphomatous polyposis in the small intestine. World J Gastroenterol 2007; 13:1453-7. [PMID: 17457982 PMCID: PMC4146935 DOI: 10.3748/wjg.v13.i9.1453] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
A 50-year old woman suffering from diabetes had a CT scan that revealed a diffuse thickening of small intestinal wall and swollen paraaortic lymph nodes. An esophagogastroduodenoscopy (EGD) confirmed multiple polypoid lesions in the duodenum and small intestine, and conventional histological testing revealed non-specific inflammatory changes. Further examinations including the immunohistochemical profiles of the biopsied specimens led us to diagnose the lesion as a marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue type, forming multiple lymphomatous polyposis sequentially spreading from duodenal bulb to terminal ileum. According to Lugano’s classification, its staging was clinically diagnosed as stage II. Two courses of a standard CHOP (cyclophosphamide, doxorubicin hydrochloride, vincristine sulfate, and predonisolone) regimen with rituximab reduced the lesion and the patient had a almost complete response. A 5-year follow-up EGD and histological examinations detected no recurrence of the disease.
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MESH Headings
- Antibodies, Monoclonal/administration & dosage
- Antibodies, Monoclonal, Murine-Derived
- Antineoplastic Combined Chemotherapy Protocols/administration & dosage
- Antineoplastic Combined Chemotherapy Protocols/therapeutic use
- Biopsy
- Cyclophosphamide/administration & dosage
- Doxorubicin/administration & dosage
- Endoscopy, Digestive System
- Female
- Humans
- Intestinal Polyposis/diagnosis
- Intestinal Polyposis/etiology
- Intestine, Small/pathology
- Lymphoma, B-Cell, Marginal Zone/complications
- Lymphoma, B-Cell, Marginal Zone/diagnosis
- Lymphoma, B-Cell, Marginal Zone/drug therapy
- Middle Aged
- Prednisolone/administration & dosage
- Rituximab
- Vincristine/administration & dosage
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Affiliation(s)
- Naoto Hirata
- Department of Gastroenterology, Osaka City University Graduate School of Medicine, 1-4-3 Asahimachi, Abeno-ku, Osaka 545-8585, Japan
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23
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Jeong DH, Do SH, Hong IH, Yang HJ, Yuan DW, Choi DH, Jeong KS. Multiple intestinal lymphomatous polyposis in a Jindo dog. J Vet Sci 2006; 7:401-3. [PMID: 17106235 PMCID: PMC3242152 DOI: 10.4142/jvs.2006.7.4.401] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022] Open
Abstract
A male, 5-year-old Jindo dog underwent enterectomy and enteroanastomosis due to ileus of the intestine at a local veterinary hospital. Grossly, the excised intestine showed markedly thickened multinodular masses in the serosal layer of the upper part, and soft-to-firm, cream-colored neoplastic masses that displayed extensive nodular mucosal protuberances into the lumen. The neoplastic masses were filled with large round cells that were ovoid in shape and they had pale and/or hyperchromatic nuclei. The neoplastic cells had mainly infiltrated into the mucosal and submucosal layers, and they had diffusely invaded the muscular and serosal layers. Therefore, the diagnosis of canine multiple intestinal malignant lymphomatous polyposis was made based on the gross and histopathological findings. The origin of these tumor cells was determined to be B-cells since they were positive for anti-CD20.
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Affiliation(s)
- Da-Hee Jeong
- Department of Pathology, College of Veterinary Medicine, Kyungpook National University, Daegu 702-701, Korea
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24
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Salar A, Juanpere N, Bellosillo B, Domingo-Domenech E, Espinet B, Seoane A, Romagosa V, Gonzalez-Barca E, Panades A, Pedro C, Nieto M, Abella E, Solé F, Ariza A, Fernández-Sevilla A, Besses C, Serrano S. Gastrointestinal involvement in mantle cell lymphoma: a prospective clinic, endoscopic, and pathologic study. Am J Surg Pathol 2006; 30:1274-80. [PMID: 17001159 DOI: 10.1097/01.pas.0000208899.15859.cb] [Citation(s) in RCA: 89] [Impact Index Per Article: 4.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/02/2023]
Abstract
The frequency of gastrointestinal (GI) tract involvement in mantle cell lymphoma (MCL) at diagnosis is reported to be below 30%. To investigate the actual frequency of GI involvement by MCL, upper and lower endoscopy was prospectively performed on 13 untreated MCL patients at diagnosis. Multiple biopsies from endoscopically normal and abnormal gastric and colonic mucosa were studied with immunohistochemistry (IHC) for CD20, CD5, and cyclin D1, as well as fluorescence in situ hybridization (FISH) for t(11;14) and polymerase chain reaction (PCR) for immunoglobulin heavy chain gene. Abnormal mucosa was identified in 38% of cases by upper endoscopy (mainly mild nonspecific gastritis) and in 54% of cases by lower endoscopy (mostly micropolyps). Histologically, infiltration by MCL was demonstrated in the stomach in 77% of cases and in the colon in 77% of cases. As a whole, 92% of patients showed upper or lower GI tract infiltration by MCL. Histologic evidence of MCL involvement was present in all cases with endoscopically abnormal mucosa, but it was also observed in two-thirds of cases with endoscopically unremarkable mucosa. Positive cyclin D1 IHC was seen in all instances displaying CD20 and CD5-positive lymphoid infiltrates, whereas t(11;14) was demonstrated by FISH in 63.5% and PCR was clonal in 64% of those instances. In conclusion, the great majority of MCL patients showed GI tract involvement at the time of diagnosis, not uncommonly in the form of minute lymphoid infiltrates. IHC for cyclin D1 was significantly more sensitive than FISH t(11;14) or PCR for immunoglobulin heavy chain gene to confirm MCL in this setting.
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Affiliation(s)
- Antonio Salar
- Department of Clinical Hematology, Hospital del Mar, Barcelona, Spain.
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25
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Kodama T, Ohshima K, Nomura K, Taniwaki M, Nakamura N, Nakamura S, Kohno S, Yamamoto J, Karube K, Yamasita Y, Shirakusa T, Kikuchi M. Lymphomatous polyposis of the gastrointestinal tract, including mantle cell lymphoma, follicular lymphoma and mucosa-associated lymphoid tissue lymphoma. Histopathology 2005; 47:467-78. [PMID: 16241994 DOI: 10.1111/j.1365-2559.2005.02225.x] [Citation(s) in RCA: 84] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
AIMS Lymphomatous polyposis (LP) is considered to represent mantle cell lymphoma (MCL) of the gastrointestinal (GI) tract. However, a few reports have suggested that some are follicular lymphoma (FL) or mucosa-associated lymphoid tissue (MALT) lymphomas. In this study, we analysed 35 patients and clarified the clinicopathological features of LP. METHODS AND RESULTS Paraffin-embedded tissue samples were stained immunohistochemically and analysed by tissue-fluorescence in situ hybridization (T-FISH) for IGH/CCND1 (cyclin D1) and IGH/BCL2. The average age of the patients was 58.3 years. Over half of the cases showed gastric, duodenal, small intestinal, ileocaecal and sigmoid colonic lesions (15, 19, 15, 16 and 16 cases, respectively). Phenotypically, cases were classified into three types of MCL (cyclin D1+ CD5+ CD10-) (n=12), FL (cyclin D1- CD5- CD10+) (n=14) and MALT (cyclin D1- CD5- CD10-) (n=9). T-FISH identified 11 of the 11 examined cases with MCLs to have IGH/CCND1, while seven of 10 cases with FL had IGH/BCL2, and none of the MALT cases were positive for IGH/CCND1 or IGH/BCL2. At the study endpoint, five of 12 patients with MCL were dead, two of 14 with FL and one of nine with MALT were dead of other disease. Event-free survival analysis showed significantly poorest outcome in MCL, followed by FL, while MALT was associated with a favourable outcome (P=0.0040). CONCLUSIONS Our study emphasizes the importance of differentiating MCL, FL and MALT of LP in evaluating prognosis and hence the most suitable therapeutic regimen.
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MESH Headings
- Aged
- Aged, 80 and over
- Diagnosis, Differential
- Female
- Gastrointestinal Neoplasms/diagnosis
- Gastrointestinal Neoplasms/pathology
- Humans
- Immunohistochemistry
- In Situ Hybridization
- Lymphoma, B-Cell, Marginal Zone/diagnosis
- Lymphoma, B-Cell, Marginal Zone/pathology
- Lymphoma, Follicular/diagnosis
- Lymphoma, Follicular/pathology
- Lymphoma, Mantle-Cell/diagnosis
- Lymphoma, Mantle-Cell/pathology
- Male
- Middle Aged
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Affiliation(s)
- T Kodama
- Department of Pathology, School of Medicine, Fukuoka University, Fukuoka, Japan.
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26
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Isomoto H, Maeda T, Akashi T, Tsuchiya T, Kawaguchi Y, Sawayama Y, Koida S, Ohnita K, Kohno S, Tomonaga M. Multiple lymphomatous polyposis of the colon originating from T-cells: a case report. Dig Liver Dis 2004; 36:218-21. [PMID: 15046193 DOI: 10.1016/j.dld.2003.09.019] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Multiple lymphomatous polyposis is an unusual form of non-Hodgkin's lymphoma characterised by myriad polyps throughout the alimentary tract. Most multiple lymphomatous polyposis cases are derived from B-cell, and there has been little information on multiple lymphomatous polyposis of T-cell origin. A 67-year-old Japanese man presented with lower abdominal pain and diarrhoea of 4-week duration. Colonoscopy revealed numerous small umbilicated polyps and several raised erosions in the colorectum. Biopsy specimens showed diffuse proliferation of lymphoma cells negative for B-cell markers but positive for T-cell markers. Polymerase chain reaction using extracted chromosomal deoxyribonucleic acid from paraffin-embedded samples identified T-cell receptor gamma and delta gene recombination. The patient was treated with combined chemotherapy, leading to complete resolution of the lesions.
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Affiliation(s)
- H Isomoto
- Department of Endoscopy, Nagasaki University School of Medicine, 1-7-1 Sakamoto, Nagasaki 852-8102, Japan.
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27
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Corcione A, Arduino N, Ferretti E, Raffaghello L, Roncella S, Rossi D, Fedeli F, Ottonello L, Trentin L, Dallegri F, Semenzato G, Pistoia V. CCL19 and CXCL12 Trigger in Vitro Chemotaxis of Human Mantle Cell Lymphoma B Cells. Clin Cancer Res 2004; 10:964-71. [PMID: 14871974 DOI: 10.1158/1078-0432.ccr-1182-3] [Citation(s) in RCA: 51] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
PURPOSE Few data are available in the literature on chemokine receptor expression and migratory capability of mantle cell lymphoma (MCL) B cells. Information on these issues may allow us to identify novel mechanisms of chemokine-driven tumor cell migration. EXPERIMENTAL DESIGN The research was designed to investigate: (a) expression of CCR1 to CCR7 and CXCR1 to CXCR5 chemokine receptors; and (b) chemotaxis to the respective ligands in MCL B cells and in their normal counterparts, i.e., CD5+ B cells. RESULTS Malignant B cells from MCL patients and normal counterparts displayed similar chemokine receptor profiles. MCL B cells were induced to migrate by CXCL12 and CCL19, whereas normal CD5+ B cells migrated to the former, but not the latter chemokine. Overnight culture of MCL B cells and their normal counterparts with CXCL12 cross-sensitized other chemokine receptors to their ligands in some tumor samples but not in CD5+ B cells. CONCLUSIONS CCR7 and CXCR4 ligands may play a key role in tumor cell migration and spreading in vivo. CXCL12 may additionally contribute by sensitizing MCL B cells to respond to the ligands of other chemokine receptors.
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Affiliation(s)
- Anna Corcione
- Laboratory of Oncology, G. Gaslini Institute, Largo G. Gaslini 5, 16148 Genoa, Italy.
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28
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Al-Marzooq YM, Chopra R, Younis M, Al-Mulhim AS, Al-Mommatten MI, Al-Omran SH. Thyroid low-grade B-cell lymphoma (MALT type) with extreme plasmacytic differentiation: Report of a case diagnosed by fine-needle aspiration and flow cytometric study. Diagn Cytopathol 2004; 31:52-6. [PMID: 15236266 DOI: 10.1002/dc.20086] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/10/2022]
Abstract
We describe a case of thyroid marginal-zone B-cell lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma) with extreme plasmacytic differentiation in an 80-year-old woman who had presented with a rapidly growing thyroid swelling. The diagnosis was initially suspected on fine-needle aspiration (FNA) and subsequently confirmed by flow cytometric analysis of the aspirated material. The smears revealed features of Hashimoto's thyroiditis admixed with large numbers of atypical large plasmacytoid lymphoid cells accompanied by variable numbers of small lymphocytes and mature plasma cells. The differential diagnosis of a predominantly plasmacytic smears on FNA of the thyroid is discussed herein, with emphasis on the role of immunophenotypic studies to exclude or confirm the diagnosis of lymphoma.
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Affiliation(s)
- Yusef M Al-Marzooq
- Department of Pathology, King Fahad Hospital Hofuf, Al-Hassa, Saudi Arabia.
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29
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Remes-Troche JM, De-Anda J, Ochoa V, Barreto-Zuñiga R, Arista-Nasr J, Valdovinos MA. A rare case of multiple lymphomatous polyposis with widespread involvement of the gastrointestinal tract. Arch Pathol Lab Med 2003; 127:1028-30. [PMID: 12873180 DOI: 10.5858/2003-127-1028-arcoml] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Multiple lymphomatous polyposis (MLP) is an uncommon type of primary non-Hodgkin gastrointestinal (GI) B-cell lymphoma characterized by the presence of multiple polyps along the GI tract. Malignant cells of MLP have mantle cell characteristics and thus are considered to be the counterpart of the mantle cell lymphoma (MCL) in the GI tract. Since 1961, no more than 70 well-documented cases have been published. We report the case of 53-year-old man diagnosed as having MLP. The patient presented with diffuse abdominal pain, chronic lower GI bleeding, peripheral lymphadenopathy, and weight loss. The lymphomatous polyps extended from the esophagus to the rectum, with bone marrow infiltration. Immunohistologic findings were characteristic of MCL. The patient was treated with a combined cyclophosphamide, vincristine, and prednisone chemotherapy regimen, resulting in a partial response.
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Affiliation(s)
- José M Remes-Troche
- Department of Gastroenterology, Instituto Nacional de Ciencias Médicas y Nutrición Salvador Zubirán, México City, México.
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30
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KOYAMA NOBUATSU, YAMASHITA NAOYUKI, SAKURAZAWA NOBUYUKI, WACHI EIKO. Endoscopic removal of primary B‐cell mucosa‐associated lymphoid tissue lymphoma of the cecum. Dig Endosc 2003. [DOI: 10.1046/j.1443-1661.2003.00232.x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/23/2023]
Affiliation(s)
- NOBUATSU KOYAMA
- Departments of * Surgery and † Pathology, Jisankai Medical Foundation, Tsuboi Hospital, Fukushima, Japan
| | - NAOYUKI YAMASHITA
- Departments of * Surgery and † Pathology, Jisankai Medical Foundation, Tsuboi Hospital, Fukushima, Japan
| | - NOBUYUKI SAKURAZAWA
- Departments of * Surgery and † Pathology, Jisankai Medical Foundation, Tsuboi Hospital, Fukushima, Japan
| | - EIKO WACHI
- Departments of * Surgery and † Pathology, Jisankai Medical Foundation, Tsuboi Hospital, Fukushima, Japan
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31
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Lam KC, Yeo W, Lee JFY, Chow J, Mok TSK. Unusual abdominal tumors: case 4. Multiple lymphomatous polyposis in mantle cell lymphoma. J Clin Oncol 2003; 21:955-956. [PMID: 12610203 DOI: 10.1200/jco.2003.11.124] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022] Open
Affiliation(s)
- Kwok C Lam
- Department of Clinical Oncology, Chinese University of Hong Kong, Prince of Wales Hospital, Hong Kong, People's Republic of China
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32
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Abstract
Synchronous occurrence of malignant lymphoma and carcinoma, both located in the intestinal tract, is unusual. We report a unique case of an adenocarcinoma of the cecum and a simultaneous mantle cell lymphoma of the colon, terminal ileum, and regional lymph nodes in an 85-year-old man. Grossly, the adenocarcinoma was identified as a cecal mass. Lymphomatous involvement of the gastrointestinal tract was evident only on microscopic examination. The terminal ileum and colon showed microscopic disseminated multiple mucosal nodules, with involvement of the regional lymph nodes. There was no involvement of distant organs, suggesting that the mantle cell lymphoma was early in its evolution without formation of polyps or a mass lesion. To our knowledge, this is the fourth reported case with this association and the second case that showed early involvement of the gastrointestinal tract with mantle cell lymphoma without polyp formation.
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33
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Vignote ML, Chicano M, Rodríguez FJ, Acosta A, Gómez F, Poyato A, Miño G. Multiple lymphomatous polyposis of the GI tract: report of a case and review. Gastrointest Endosc 2002; 56:579-82. [PMID: 12297783 DOI: 10.1016/s0016-5107(02)70453-7] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Affiliation(s)
- Maria Luisa Vignote
- Department of Gastroenterology and Hepatology, Reina Sofia University Hospital, Córdoba, Spain
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34
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Viana HL, Henrique RM, Ferreira ES, Correia AM, Silva RA, Dias LM, Viana RL. Endoscopic ultrasonography in multiple lymphomatous polyposis. J Clin Gastroenterol 2002; 34:150-4. [PMID: 11782610 DOI: 10.1097/00004836-200202000-00010] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/09/2022]
Abstract
Two cases of multiple lymphomatous polyposis (MLP) are presented, involving different segments of the gastrointestinal tract. Both cases display the characteristic clinical and pathologic features of MLP. In addition, we were able to document, for the first time, the endoscopic ultrasonographic findings in this disease. This new ancillary diagnostic technique was found to be very helpful in the evaluation of the structural changes of the wall of the gastrointestinal tract and in the detection of affected lymph nodes.
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Affiliation(s)
- Helena Lomba Viana
- Department of Gastroenterology, The Portuguese Cancer Institute, Oporto, Portugal
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35
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Antonio Álvarez M, Carbó S, Espinosa Í. Dolor abdominal y pérdida de peso en un varón de 18 años. Med Clin (Barc) 2002. [DOI: 10.1016/s0025-7753(02)72395-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
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36
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Isomoto H, Ohnita K, Mizuta Y, Maeda T, Onizuka Y, Miyazaki M, Omagari K, Takeshima F, Murase K, Haraguchi M, Murata I, Kohno S. Clinical and endoscopic features of adult T-cell leukemia/lymphoma with duodenal involvement. J Clin Gastroenterol 2001; 33:241-6. [PMID: 11500618 DOI: 10.1097/00004836-200109000-00017] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/09/2022]
Abstract
We describe three cases of adult T-cell leukemia/lymphoma (ATLL) with duodenal involvement and provide a review of the literature. The first case, a 74-year-old woman with acute subtype of ATLL, had multiple polypoid lesions from the bulbus extending into the descending portion of the duodenum. The second case, a 70-year-old man with lymphoma subtype of ATLL, had a polypoid tumor in the descending portion of the duodenum and multiple protruded lesions in the small and large intestines. The third case, a 67-year-old man with lymphoma subtype of ATLL, had a flat-elevated lesion in the descending portion of the duodenum, as well as a gastric ulcerated lesion. Biopsies from these lesions showed mucosal invasion of ATLL cells in each case. All patients received combination chemotherapy, which was successful in the first and third cases, accompanied by the disappearance of gastroduodenal lesions.
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Affiliation(s)
- H Isomoto
- Second Department of Internal Medicine, Molecular Medicine Unit, Atomic Bomb Disease Institute, Nagasaki University School of Medicine, Nagasaki, Japan.
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37
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Itoh T, Shimizu M, Kitami K, Kamata K, Mitsumori K, Fujita M, Ohnishi A, Nagashima K. Primary extranodal marginal zone B-cell lymphoma of the mucosa-associated lymphoid tissue type in the CNS. Neuropathology 2001; 21:174-80. [PMID: 11666014 DOI: 10.1046/j.1440-1789.2001.00392.x] [Citation(s) in RCA: 50] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
Extranodal marginal zone B-cell lymphoma of the mucosa-associated lymphoid tissue (MALT) type has been reported in various internal organs. Here a case is reported of MALT lymphoma developing in the cerebellopontine (CP) angle in a patient with Sjogren syndrome, and the concept of MALT lymphoma of the CNS is introduced. Pathologically, the tumor showed inflammatory features of reactive lymphocytic infiltration with follicle formation, but there were slightly atypical lymphocytes and plasmacytes with B-cell markers. These cells invaded reactive follicles, showing follicular colonization, and showed aberrant expression indicating their neoplastic nature. A review of the literature revealed eight cases of MALT lymphoma originating from the dura mater and one from the CP angle. The average age of patients was 50 years (range 28-66 years), and all patients were female. The tumors were slow to develop and the patients were cured after surgical removal and/or additional therapies. It is proposed that MALT lymphoma should be considered as a differential diagnosis of inflammatory pseudotumor of the CNS.
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Affiliation(s)
- T Itoh
- Department of Surgical Pathology, Hokkaido University Hospital, Sapporo, Japan.
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38
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Kanehira K, Braylan RC, Lauwers GY. Early phase of intestinal mantle cell lymphoma: a report of two cases associated with advanced colonic adenocarcinoma. Mod Pathol 2001; 14:811-7. [PMID: 11504842 DOI: 10.1038/modpathol.3880395] [Citation(s) in RCA: 26] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/14/2022]
Abstract
Intestinal mantle cell lymphoma characteristically produces multiple polyps, a finding reported as multiple lymphomatous polyposis. The early stages of intestinal mantle cell lymphoma before polyp formation and the pattern of initial lymph node invasion, however, have not been described. We recently encountered two cases of intestinal mantle cell lymphoma in their early development found incidentally associated with advanced colonic adenocarcinoma. We present herein the clinical, histopathological, immunohistochemical, and molecular genetic features of these two cases. In one case, a single polypoid mass was found with invasion limited to mucosa and submucosa of the terminal ileum and without lymph node compromise. In the second case, there were multiple mucosal aggregates of neoplastic cells without formation of polyps. Regional lymph nodes in the latter case showed either partial or complete involvement by lymphoma. In both cases, immunohistochemistry (CD20+, CD5+, cyclin D1+, CD10-, and CD23-), and demonstration of clonal immunoglobulin heavy chain and bcl-1 gene rearrangements by PCR analysis confirmed the diagnosis of mantle cell lymphoma.
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Affiliation(s)
- K Kanehira
- Department of Pathology and Laboratory Medicine, University of Florida College of Medicine, Gainesville, FL, USA
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39
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Brouland JP, Poupard B, Nemeth J, Valleur P. Lipomatous polyposis of the colon with multiple lipomas of peritoneal folds and giant diverticulosis: report of a case. Dis Colon Rectum 2000; 43:1767-9. [PMID: 11156466 DOI: 10.1007/bf02236867] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
A case of multiple lipomatosis exclusively located in the colon is reported in a young male (33 years). It is characterized by a great number of lipomas with polyposis growth appearance, multiple lipomas of peritoneal folds, and giant diverticulosis probably caused by weakened areas of colonic wall induced by the lipomas.
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Affiliation(s)
- J P Brouland
- Service d'Anatomie et de Cytologie Pathologiques, H pital Lariboisière, Paris, France
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40
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Hashimoto Y, Nakamura N, Kuze T, Ono N, Abe M. Multiple lymphomatous polyposis of the gastrointestinal tract is a heterogenous group that includes mantle cell lymphoma and follicular lymphoma: analysis of somatic mutation of immunoglobulin heavy chain gene variable region. Hum Pathol 1999; 30:581-7. [PMID: 10333231 DOI: 10.1016/s0046-8177(99)90205-8] [Citation(s) in RCA: 48] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
Multiple lymphomatous polyposis (MLP) is characterized by multiple polyps involving long segments of the gastrointestinal (GI) tract. MLP is thought to represent mantle cell lymphoma (MCL) of the GI tract; however, some cases of follicular lymphoma (FL) of the GI tract are found with a multiple polypoid appearance. In the present study, to clarify the cellular origin of MLP, clonal immunoglobulin heavy chain (IgH) gene rearrangement of four cases with MLP was amplified by polymerase chain reaction (PCR) and analyzed for the presence of somatic mutation. The IgH variable (VH) region sequences of three cases (CD5+ CD10- cyclin D1+) showed a little somatic mutation compared with the closest published germline. The other case (CD10+ CD5- cyclin D1-) was highly mutated and showed intraclonal heterogeneity (ongoing somatic hypermutation). These data indicate that three of the cases with MLP are derived from pregerminal center B cells (mantle zone B cells) and one case with MLP from germinal center B cells. Our study suggests that MLP is a heterogenous group that includes MCL and FL.
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Affiliation(s)
- Y Hashimoto
- 1st Department of Pathology, School of Medicine, Fukushima Medical University, Japan
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41
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Höfler G. Klassifikation und Stadieneinteilung gastrointestinaler Lymphome. Eur Surg 1998. [DOI: 10.1007/bf02620105] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/23/2022]
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42
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Yatabe Y, Nakamura S, Nakamura T, Seto M, Ogura M, Kimura M, Kuhara H, Kobayashi T, Taniwaki M, Morishima Y, Koshikawa T, Suchi T. Multiple polypoid lesions of primary mucosa-associated lymphoid-tissue lymphoma of colon. Histopathology 1998; 32:116-25. [PMID: 9543667 DOI: 10.1046/j.1365-2559.1998.00315.x] [Citation(s) in RCA: 46] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Abstract
AIMS This study was focused on unusual cases of mucosa-associated lymphoid tissue (MALT) lymphoma presenting with multiple polypoid lesions of the colon and rectum with a special reference to the differential diagnosis of mantle cell lymphoma. METHODS AND RESULTS The lesions of these five cases grossly showed a segmental distribution of nodular protrusions in three patients and of innumerable small polyps in two. These patterns of involvement simulated those of multiple lymphomatous polyposis, known as the gastrointestinal presentation of mantle cell lymphoma (MCL), and caused a differential diagnostic problem between MALT lymphoma and MCL, which have different prognostic and therapeutic implications. Their histological features are almost indistinguishable from each other, especially in the small biopsy specimens via endoscope. The most important procedure for their differentiation is cyclin D1 immunohistochemistry and its negative reaction provides strong indication of MALT lymphoma. Of interest, one case showed a rare karyotypic abnormality of t(11;18)(q21;q21), which has been reported specifically in MALT lymphoma. CONCLUSIONS This study has indicated that the multiple polypoid lesions of the colon occur not only in MCL, but also in MALT lymphoma, making differential diagnosis between the two entities necessary, and cyclin D1 immunohistochemistry is indispensable for distinguishing between them.
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MESH Headings
- Adult
- Aged
- Colonic Polyps/genetics
- Colonic Polyps/immunology
- Colonic Polyps/pathology
- Colorectal Neoplasms/genetics
- Colorectal Neoplasms/immunology
- Colorectal Neoplasms/pathology
- Diagnosis, Differential
- Female
- Humans
- Immunohistochemistry
- Lymphoma, B-Cell, Marginal Zone/genetics
- Lymphoma, B-Cell, Marginal Zone/immunology
- Lymphoma, B-Cell, Marginal Zone/pathology
- Lymphoma, Non-Hodgkin/pathology
- Male
- Middle Aged
- Neoplasms, Multiple Primary/genetics
- Neoplasms, Multiple Primary/immunology
- Neoplasms, Multiple Primary/pathology
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Affiliation(s)
- Y Yatabe
- Department of Pathology and Clinical Laboratories, Aichi Cancer Center Hospital, Japan
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43
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44
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Honda K, Mizuno M, Matsumoto T, Kuroki F, Hoshika K, Iida M, Sakurai T, Shimizu M. A case of systemic malignant lymphoma with intestinal involvement of lymphomatous polyposis type. J Clin Gastroenterol 1997; 25:362-4. [PMID: 9412922 DOI: 10.1097/00004836-199707000-00015] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Abstract
Multiple lymphomatous polyposis is a rare type of intestinal lymphoma characterized by non-Hodgkin's lymphoma of follicular mantle cell origin and extremely poor prognosis. We report a case of systemic lymphoma with the intestinal involvement of multiple lymphomatous polyposis. Although radiographic and endoscopic features of the case were compatible with multiple lymphomatous polyposis, histologic evidence suggested the diagnosis of diffuse large cell lymphoma rather than mantle cell lymphoma. Our case seems to be unique in its histologic findings and also in its prognosis, because the patient has been alive for more than 50 months after diagnosis.
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Affiliation(s)
- K Honda
- Division of Gastroenterology, Kawasaki Medical School, Kurashiki, Japan
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45
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Abstract
BACKGROUND The term "pseudolymphoma" has been used to describe lymphoid lesions that show: 1) borderline features between hyperplasia and neoplasia or 2) benign tumor-like enlargement of lymphoid tissue. The term pseudolymphoma has been applied to lesions in many anatomic locations, with the stomach being one of the more common sites. In spite of the frequent use of this term, neither the histologic criteria nor the clinical significance of this lesion have ever been clearly defined. Since the description of mucosa-associated lymphoid tissue (MALT) and its corresponding MALT-type lymphomas, the value of the term gastric pseudolymphoma has come into question. METHODS The authors reviewed all cases diagnosed as gastric pseudolymphomas at the Armed Forces Institute of Pathology from 1970 to 1985. This period predated the description of low grade B-cell lymphoma of MALT-type. The cases were reclassified in light of current criteria and correlated with immunohistochemical findings and follow-up information. No patients were treated with chemotherapy or radiation therapy. RESULTS Seventy-seven of 97 cases formerly diagnosed as pseudolymphoma were determined to be malignant lymphomas; the majority (51 cases) were extranodal marginal zone B-cell lymphomas (MALT-type). The remaining cases included reactive lymphoid hyperplasia associated with chronic follicular gastritis (15 cases) and atypical lymphoid infiltrates (5 cases). CONCLUSIONS Most cases previously considered to be gastric pseudolymphomas are, by current criteria, malignant lymphomas. A small proportion are benign reactive lymphoid hyperplasias. Those cases of borderline or inconclusive nature are best diagnosed as atypical lymphoid infiltrates. The term gastric pseudolymphoma should be abandoned.
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Affiliation(s)
- S L Abbondanzo
- Department of Hematologic and Lymphatic Pathology, Armed Forces Institute of Pathology, Washington, DC 20306-6000, USA
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46
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Abstract
Multiple lymphomatous polyposis (MLP) is a distinctive and particularly rare clinical type of malignant gastrointestinal lymphoma, which is classified as B-cell centrocytic non-Hodgkin's lymphoma. this rare entity has been recently reclassified as mantle cell lymphoma. We herein report three additional cases of MLP involving various segments of the gastrointestinal tract. MLP has an aggressive biologic behavior and a relatively poor prognosis and must be treated accordingly as a high-grade lymphoma with systemic chemotherapy.
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Affiliation(s)
- A Kadayifçi
- Department of Internal Medicine, Hacettepe University Faculty of Medicine, Ankara, Turkey
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47
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Case records of the Massachusetts General Hospital. Weekly clinicopathological exercises. Case 8-1997. A 65-year-old man with recurrent abdominal pain for five years. N Engl J Med 1997; 336:786-93. [PMID: 9052658 DOI: 10.1056/nejm199703133361108] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/03/2023]
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48
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Yatabe Y, Nakamura S, Seto M, Kuroda H, Kagami Y, Suzuki R, Ogura M, Kojima M, Koshikawa T, Ueda R, Suchi T. Clinicopathologic study of PRAD1/cyclin D1 overexpressing lymphoma with special reference to mantle cell lymphoma. A distinct molecular pathologic entity. Am J Surg Pathol 1996; 20:1110-22. [PMID: 8764748 DOI: 10.1097/00000478-199609000-00009] [Citation(s) in RCA: 63] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/02/2023]
Abstract
Mantle cell lymphomas (MCLs) are frequently associated with the overexpression of PRAD1/cyclin D1, activated by 11q13 translocation and its molecular counterpart BCL-1 gene rearrangement. We recently described the correlation of positive nuclear staining using monoclonal antibody against a PRAD1/cyclin D1 product with mRNA overexpression in MCLs. In the present study, we immunohistochemically investigated the PRAD1/cyclin D1 protein in a large series of 334 lymphoproliferative disorders, including 39 cases of MCLs on paraffin sections. Based on the cyclin D1 positivity, CD5 expression, and the morphologic features of the tumor tissue, four groups of MCL-related lesions were identified among the B-cell lymphomas examined: 36 cases with cyclin D1 overexpression, 35 (95%) of which exhibited CD5-positivity and MCL-morphology (Group 1); four cases of lymphomas with MCL morphology and CD5 expression but lacking cyclin D1 overexpression (Group II); four cases of lymphomas without cyclin D1 overexpression and surface CD5 but that fall within the morphologic boundaries of MCLs (Group III); and 11 cases of CD5-positive diffuse large cell lymphomas without cyclin D1 overexpression (Group IV). The Group I cases demonstrated quite homogeneous clinicopathologic features identical to those of MCLs. This group showed a poor prognosis (11% had 5-year survival), which is highly contrasted with that of Group II (100%). Although the four groups of MCL-related lesions sometimes overlapped in their histologic or phenotypic spectrums, each appeared to show distinct clinicopathologic and prognostic profiles. Our study provides a basis for further clarification of the nature of the neoplasms of Groups II, III, and IV. Moreover, this comprehensive study may indicate that the overexpression of PRAD1/cyclin D1 is biologically essential to defining MCLs.
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Affiliation(s)
- Y Yatabe
- Department of Pathology and Clinical Laboratories, Aichi Cancer Center Hospital, Nagoya, Japan
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49
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Robert ME, Kuo FC, Longtine JA, Sklar JL, Schrock T, Weidner N. Diffuse colonic mantle cell lymphoma in a patient with presumed ulcerative colitis: detection of a precursor monoclonal lymphoid population using polymerase chain reaction and immunohistochemistry. Am J Surg Pathol 1996; 20:1024-31. [PMID: 8712289 DOI: 10.1097/00000478-199608000-00011] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/01/2023]
Abstract
Primary lymphoma of the colon, a rare and typically late complication of ulcerative colitis, exhibits high-grade morphology and behavior when it occurs. Recently, several reports of colonic lymphoma masquerading as ulcerative colitis have been described. These previous reports described inflammatory mucosal changes typical of ulcerative colitis as being present in superficial biopsies, leading to the initial diagnosis of ulcerative colitis; however, further workup resulted in a diagnosis of primary colonic lymphoma within several months in these cases, and all symptoms and mucosal changes resolved after treatment of the lymphoma. Herein we report a case of mantle cell lymphoma arising in the colon and rectum in a 71-year-old woman with a 4-year history of ulcerative colitis. Immunoglobulin heavy-chain gene rearrangements were detected using the polymerase chain reaction procedure in fixed tissue in the lymphoma as well as in a prior resection specimen that histologically appeared to show only changes of severe ulcerative colitis. This finding suggests that an indolent lymphoid proliferation may have been the underlying disease in this patient and raises questions about the role of colonic lymphoma in causing mucosal injury.
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Affiliation(s)
- M E Robert
- Department of Pathology, University of California at San Francisco, USA
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50
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Tsutsumi Y, Osamura RY, Makuuchi H. Intermediate lymphocytic lymphoma massively involving the gastrointestinal tract. Pathol Int 1996; 46:380-3. [PMID: 8809886 DOI: 10.1111/j.1440-1827.1996.tb03625.x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/02/2023]
Abstract
A case is presented of intermediate lymphocytic lymphoma seen in a 53 year old male, which extensively infiltrated systemic organs, including the entire digestive tract from the esophagus to the rectum. Peyer's patch invasion was evident. Multiple intestinal perforations caused death 5 months later. Surface marker studies suggested the marginal zone origin for this CD20+ B cell malignancy.
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Affiliation(s)
- Y Tsutsumi
- Department of Pathology, Tokai University School of Medicine, Isehara, Japan
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