Journal of Pathology and Translational Medicine

Case Studies

Gastric crystal-storing histiocytosis with concomitant mucosa-associated lymphoid tissue lymphoma: Mee Joo, Nam-Hoon Kim; J Pathol Transl Med. 2020;54(4):332-335. Published online May 22, 2020; DOI: https://doi.org/10.4132/jptm.2020.04.20

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Crystal-storing histiocytosis (CSH) is a rare entity that is characterized by intrahistiocytic accumulation of crystallized immunoglobulins. CSH is not a malignant process per se, but the majority of CSH cases are associated with underlying lymphoproliferative disorder. Although CSH can occur in a variety of organs, gastric CSH is very rare. We present a localized gastric CSH with concomitant mucosaassociated lymphoid tissue (MALT) lymphoma, manifesting as an ulcer bleeding in a 56-year-old man. Histologically, the biopsied gastric mucosa demonstrated expansion of the lamina propria by prominent collections of large eosinophilic mononuclear cells containing fibrillary crystalloid inclusions. Immunohistochemical studies revealed that the crystal-storing cells were histiocytes harboring kappa light chain-restricted immunoglobulin crystals. Within the lesion, atypical centrocyte-like cells forming lymphoepithelial lesions were seen, consistent with MALT lymphoma. Since this entity is rare and unfamiliar, difficulties in diagnosis may arise. Particularly, in this case, the lymphomatous area was obscured by florid CSH, making the diagnosis more challenging.

Citations

Citations to this article as recorded by

Crystal-storing histiocytosis in the stomach: A case report and review of the literature
Linghong Kong, Liyan Xue, Yanfeng Zhong, Shenglan Wang, Danfeng Zheng, Lining Wang, Yang Jiao, Xinpeng Zhang, Huizhong Xue, Xiaogang Liu
Frontiers in Oncology.2022;[Epub] CrossRef
Lambda-Restricted Crystal-Storing Histiocytosis of Stomach: A Case Report and Review of Literature
Nalini Bansal, Pankaj Puri, Nishant Nagpal, Rahul Naithani, Rahul Gupta
Cureus.2021;[Epub] CrossRef
Immunoglobulin-Storing Histiocytosis: A Case Based Systemic Review
Hanne Wiese-Hansen, Friedemann Leh, Anette Lodvir Hemsing, Håkon Reikvam
Journal of Clinical Medicine.2021; 10(9): 1834. CrossRef

Primary hepatic extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue: Soyeon Choi, Ji Hye Kim, Kyungbin Kim, Misung Kim, Hye Jeong Choi, Young Min Kim, Jae Hee Suh, Min Jung Seo, Hee Jeong Cha; J Pathol Transl Med. 2020;54(4):340-345. Published online April 15, 2020; DOI: https://doi.org/10.4132/jptm.2020.03.18

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Abstract PDF

Extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue (MALT lymphoma), is one of the specific type of low-grade B-cell lymphoma not infrequently found worldwide. It typically involves mucosal sites such as stomach and conjunctiva; however, primary hepatic MALT lymphoma has been extremely rarely reported. We describe a case of hepatic MALT lymphoma in a 70-year-old male patient who underwent left hepatectomy due to the incidentally detected liver masses at a medical checkup. The resected specimen revealed multinodular masses consisting of small-to-intermediate-sized lymphoid cells with serpentine pattern and focal lymphoepithelial lesions. The tumor cells were diffusely positive for CD20 and Bcl-2 but negative for CD3, CD10, CD5, CD23, CD43, and cyclinD1. The Ki-67 labeling index was 10% and immunoglobulin heavy chain gene rearrangement study confirmed monoclonal proliferation. In this paper, we discuss several unique clinicopathologic characteristics which will be helpful to the differential diagnosis of hepatic MALT lymphoma.

Citations

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“Speckled Enhancement” on Gd-EOB-DTPA Enhanced MR Imaging of Primary Hepatic Mucosa-associated Lymphoid Tissue Lymphoma
Ryota Hyodo, Yasuo Takehara, Ayumi Nishida, Masaya Matsushima, Shinji Naganawa
Magnetic Resonance in Medical Sciences.2023; 22(3): 273. CrossRef
Primary hepatic extranodal marginal zone B-cell mucosa-associated lymphoid tissue lymphoma treated by laparoscopic partial hepatectomy: a case report
Keisuke Okura, Satoru Seo, Hironori Shimizu, Hiroto Nishino, Tomoaki Yoh, Ken Fukumitsu, Takamichi Ishii, Koichiro Hata, Hironori Haga, Etsuro Hatano
Surgical Case Reports.2023;[Epub] CrossRef
Incidental Findings in Pediatric Patients: How to Manage Liver Incidentaloma in Pediatric Patients
Andrius Cekuolis, Dagmar Schreiber-Dietrich, Rasa Augustinienė, Heike Taut, Judy Squires, Edda L. Chaves, Yi Dong, Christoph F. Dietrich
Cancers.2023; 15(8): 2360. CrossRef
Primary hepatic mucosa‐associated lymphoid tissue lymphoma: Case report and literature review
Wing Yu Lau, Kit‐Man Ho, Fiona Ka‐Man Chan, Shi Lam, Kai‐Chi Cheng
Surgical Practice.2022; 26(1): 56. CrossRef
18F-FDG Versus 68Ga-FAPI PET/CT in Visualizing Primary Hepatic Extranodal Marginal Zone Lymphoma of Mucosa-Associated Lymphoid Tissue
Yizhen Pang, Long Zhao, Qihang Shang, Tinghua Meng, Haojun Chen
Clinical Nuclear Medicine.2022; 47(4): 375. CrossRef
Primary hepatopancreatobiliary lymphoma: Pathogenesis, diagnosis, and management
Qianwen Wang, Kangze Wu, Xuzhao Zhang, Yang Liu, Zhouyi Sun, Shumei Wei, Bo Zhang
Frontiers in Oncology.2022;[Epub] CrossRef
Positive effect of Bifidobacterium animalis subsp. lactis VHProbi YB11 in improving gastrointestinal movement of mice having constipation
Hongchang Cui, Qian Wang, Congrui Feng, Chaoqun Guo, Jingyan Zhang, Xinping Bu, Zhi Duan
Frontiers in Microbiology.2022;[Epub] CrossRef
A case of primary hepatic extranodal marginal zone B-cell mucosa-associated lymphoid tissue (MALT) lymphoma treated by radiofrequency ablation (RFA), and a literature review
Zhe Xu, Chong Pang, Jidong Sui, Zhenming Gao
Journal of International Medical Research.2021; 49(3): 030006052199953. CrossRef

Original Article

Colorectal epithelial neoplasm associated with gut-associated lymphoid tissue: Yo Han Jeon, Ji Hyun Ahn, Hee Kyung Chang; J Pathol Transl Med. 2020;54(2):135-145. Published online January 29, 2020; DOI: https://doi.org/10.4132/jptm.2019.11.06

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Abstract PDF: Background
Colorectal epithelial neoplasm extending into the submucosal gut-associated lymphoid tissue (GALT) can cause difficulties in the differential diagnosis. Regarding GALT-associated epithelial neoplasms, a few studies favor the term “GALT carcinoma” while other studies have mentioned the term “GALT-associated pseudoinvasion/epithelial misplacement (PEM)”.
Methods
The clinicopathologic characteristics of 11 cases of colorectal epithelial neoplasm associated with submucosal GALT diagnosed via endoscopic submucosal dissection were studied.
Results
Eight cases (72.7%) were in males. The median age was 59 years, and age ranged from 53 to 73. All cases had a submucosal tumor component more compatible with GALT-associated PEM. Eight cases (72.7%) were located in the right colon. Ten cases (90.9%) had a non-protruding endoscopic appearance. Nine cases (81.8%) showed continuity between the submucosal and surface adenomatous components. Nine cases showed (81.8%) focal defects or discontinuation of the muscularis mucosae adjacent to the submucosal GALT. No case showed hemosiderin deposits in the submucosa or desmoplastic reaction. No case showed single tumor cells or small clusters of tumor cells in the submucosal GALT. Seven cases (63.6%) showed goblet cells in the submucosa. No cases showed oncocytic columnar cells lining submucosal glands.
Conclusions
Our experience suggests that pathologists should be aware of the differential diagnosis of GALT-associated submucosal extension by colorectal adenomatous neoplasm. Further studies are needed to validate classification of GALT-associated epithelial neoplasms.

Case Study

Micronodular Thymoma with Lymphoid Stroma in a Multilocular Thymic Cyst: A Case Study: Na Rae Kim, Jae Ik Lee, Seung Yeon Ha; Korean J Pathol. 2013;47(4):392-394. Published online August 26, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.4.392

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Abstract PDF

Herein, we report a case of micronodular thymoma with lymphoid stroma in a previously healthy 73-year-old male. Thymectomy was performed. The solid and macrocystic masses were encapsulated with focal invasion. The solid portion consisted of nodules of bland-looking spindle or round epithelial cells in lymphoid stroma containing prominent germinal centers. The epithelial cells had moderate amount of cytoplasm and occasional mucin production. The cystic portion was lined with cuboidal epithelium. According to World Health Organization (WHO) classification, the mass was diagnosed as a micronodular thymoma with lymphoid stroma accompanied by a pre-existing multilocular thymic cyst. Micronodular thymoma with lymphoid stroma, a possible variant of type A thymoma, is an extremely rare tumor. This so-called "unusual" variant may imply the schematic weakness of the current WHO classification that cannot cover all morphologic types. Further study is recommended for clarification of this variant and its incorporation into the current classification.

Citations

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GTF2Imutation in micronodular thymoma with lymphoid stroma
Andrea Bille, Katherine Fryer, Andrew Wallace, Daisuke Nonaka
Journal of Clinical Pathology.2024; 77(2): 125. CrossRef
Thymic epithelial tumours: histopathological classification and differential diagnosis
Jan von der Thüsen
Histopathology.2024; 84(1): 196. CrossRef
Micronodular Thymomas With Prominent Cystic Changes: A Clinicopathological and Immunohistochemical Study of 25 Cases
Diana M. Oramas, Cesar A. Moran
International Journal of Surgical Pathology.2021; 29(4): 352. CrossRef
Two cases of resection of micronodular thymoma with lymphoid stroma
Seiji Omura, Kyohei Masai, Kaoru Kaseda, Keisuke Asakura, Tomoyuki Hishida, Hisao Asamura
The Journal of the Japanese Association for Chest Surgery.2021; 35(6): 705. CrossRef
Two surgical cases of micronodular thymoma with lymphoid stroma
Yusuke Kita, Yoshimasa Tokunaga, Taku Okamoto
The Journal of the Japanese Association for Chest Surgery.2020; 34(2): 166. CrossRef
Thoracoscopic Thymectomy for Large Thymic Cyst: Myasthenia Gravis With Thymoma Concealed by Thymic Cyst
Motoki Yano, Hiroki Numanami, Takashi Akiyama, Rumiko Taguchi, Chihiro Furuta, Akari Iwakoshi, Masayuki Haniuda
Surgical Laparoscopy, Endoscopy & Percutaneous Techniques.2019; 29(3): e34. CrossRef
A resected case of micronodular thymoma with lympoid stroma
Hiromitsu Domen, Yasuhiro Hida, Yasunari Takakuwa, Yuki Iijima, Kazuomi Ichinokawa, Hidehisa Yamada
The Journal of the Japanese Association for Chest Surgery.2019; 33(5): 504. CrossRef
Thymoma and thymic carcinoma associated with multilocular thymic cyst: a clinicopathologic analysis of 18 cases
Xuxia Shen, Yan Jin, Lei Shen, Yihua Sun, Haiquan Chen, Yuan Li
Diagnostic Pathology.2018;[Epub] CrossRef
Micronodular thymoma with lymphoid stroma: Two cases, one in a multilocular thymic cyst, and literature review
Linlin Qu, Yan Xiong, Qian Yao, Bo Zhang, Ting Li
Thoracic Cancer.2017; 8(6): 734. CrossRef
Cystic Micronodular Thymoma. Report of a Case
Mlika M
Journal of Clinical, Medical and Experimental Images.2017; 1(1): 001. CrossRef
A Rare Case of Mixed Type A Thymoma and Micronodular Thymoma with Lymphoid Stroma
Yoon Jin Cha, Joungho Han, Jimin Kim, Kyung Soo Lee, Young Mog Shim
Journal of Pathology and Translational Medicine.2015; 49(1): 75. CrossRef
Micronodular thymic neoplasms: case series and literature review with emphasis on the spectrum of differentiation
Wadad S Mneimneh, Yesim Gökmen-Polar, Kenneth A Kesler, Patrick J Loehrer Sr, Sunil Badve
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Original Articles

Expression of bcl-2 and p53 Protein in Primary Gastric Lymphomas.: Young Rok Cho, Yu Na Kang, Sang Sook Lee, Hong Suk Song, Soo Sang Sohn, Dong Sug Kim; Korean J Pathol. 1998;32(11):978-984.

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Abstract: The bcl-2 gene is a proto-oncogene which extends cell survival by blocking apoptosis. Bcl-2 expression has been detected in many types of nodal and MALT lymphoma. The p53 gene is a tumor suppressor gene and p53 mutation is the most common genetic alteration in human malignancies. The relationship between the expression of bcl-2 and p53 protein in primary gastric lymphoma has been rarely reported. The authors investigated the expression of bcl-2 and p53 protein in 37 cases of primary gastric lymphoma by immunohistochemical method using bcl-2 and p53 monoclonal antibodies. There were five cases of low grade B-cell MALT lymphomas and thirty two cases of high grade B-cell lymphomas. Fifteen of 37 cases (41%) showed bcl-2 protein expression in the cytoplasm of tumor cells and 26 cases (70%) showed p53 protein expression in the nucleus of tumor cells. Bcl-2 protein was detected in 4 of 5 (80%) low grade MALT lymphomas, and in 11 of 32 (34%) high grade lymphomas. There was no significant correlation between bcl-2 expression and histologic grade of primary gastric lymphomas (p>0.05). p53 protein was positive in 25 of 32 (78%) high grade lymphomas, and in 1 of 5 (20%) low grade MALT lymphomas. The expression of p53 protein is significantly higher in high grade lymphoma than in low grade MALT lymphoma (p<0.05). The p53 expression in the bcl-2 negative cases (86%) was significantly higher than in the bcl-2 positive cases (47%). There was an inverse relationship between bcl-2 and p53 expression in primary gastric lymphoma. These results suggest that bcl-2 and p53 expression in primary gastric lymphoma may be involved in the transition from low grade MALT lymphoma to high grade lymphoma.

Methylotion Analysis of p16/INK4A in Gastric Low-Grade Mucosa-Associated Lymphoid Tissue Lymphomas after Helicobacter pylori Eradication Therapy.: Young A Kim, Sung Shin Park, Bo Young Lee, You Sun Kim, In Sung Song, Chul Woo Kim; Korean J Pathol. 2002;36(1):13-20.

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Abstract PDF: BACKGROUND
Inactivation of p16 has been associated with promoter region hypermethylation in different types of malignancies, including non-Hodgkin's lymphomas (NHLs). This loss of p16 was found frequently in cases of mucosa-associated lymphoid tissue (MALT) lymphomas. Recent studies indicate that promoter hypermethylation is often an early event in tumor progression in the follow-up of NHLs.
METHODS
To investigate the usefulness of p16 methylation in the diagnosis and follow-up of gastric low-grade MALT lymphomas, we analyzed methylation status of p16 using methylation-specific polymerase chain reaction methods in the sequential biopsy specimens of 13 patients with gastric low-grade MALT lymphomas undergoing Helicobacter pylori eradication therapy.
RESULTS
Five of thriteen cases showed p16 hypermethylation upon diagnosis. In four of five methylation positive cases, abnormal methylation was detected in the specimen even after the treatment, although there were no histologic evidence of disease. This methylation disappeared in the later samples of two of the cases, and they have remained in complete remission. Immunohistochemically, the loss of p16 protein expression was detected in one of three methylation-positive cases, and in none of the methylation-negative cases.
CONCLUSIONS
These results suggest that p16 methylation is relatively fequent in low-grade gastric MALT lymphomas, and it may have clinical applications in the management and follow-up of low-grade gastric MALT lymphomas.

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