Journal of Pathology and Translational Medicine

Case Studies

Uncommon granulomatous manifestation in Epstein-Barr virus–positive follicular dendritic cell sarcoma: a case report: Henry Goh Di Shen, Yue Zhang, Wei Qiang Leow; J Pathol Transl Med. 2025;59(2):133-138. Published online October 31, 2024; DOI: https://doi.org/10.4132/jptm.2024.09.27

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Hepatic Epstein-Barr virus–positive inflammatory follicular dendritic cell sarcoma (EBV+ IFDCS) represents a rare form of liver malignancy. The absence of distinct clinical and radiological characteristics, compounded by its rare occurrence, contributes to a challenging diagnosis. Here, we report a case of a 54-year-old Chinese female with a background of chronic hepatitis B virus treated with entecavir and complicated by advanced fibrosis presenting with a liver mass found on her annual surveillance ultrasound. Hepatectomy was performed under clinical suspicion of hepatocellular carcinoma. Immunomorphologic characteristics of the tumor were consistent with EBV+ IFDCS with distinct non-caseating granulomatous inflammation. Our case illustrates the importance of considering EBV+ IFDCS in the differential diagnosis of hepatic inflammatory lesions. Awareness of this entity and its characteristic features is essential for accurately diagnosing and managing this rare neoplasm.

Citations

Citations to this article as recorded by

Mesenchymal Tumors of the Liver: An Update Review
Joon Hyuk Choi, Swan N. Thung
Biomedicines.2025; 13(2): 479. CrossRef

Recurrent Indeterminate Dendritic Cell Tumor of the Skin: Jin Woo Joo, Taek Chung, Yoon Ah Cho, Sang Kyum Kim; J Pathol Transl Med. 2018;52(4):243-247. Published online April 5, 2018; DOI: https://doi.org/10.4132/jptm.2018.03.27

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Abstract PDF

Indeterminate dendritic cell tumor (IDCT) is a dendritic cell tumor that displays histologic features similar to those of Langerhans cells. The origin of the indeterminate cells may represent precursors of Langerhans cells or skin dendritic cells. IDCT is extremely rare, and tumor progression and predictive factors are not well known. Here, we report a case of a 61-year-old man who presented with a papule on his back and was finally diagnosed with IDCT based on histology and immunohistochemistry. The tumor recurred three months after surgical excision.

Citations

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Pathologic characteristics of histiocytic and dendritic cell neoplasms
Sun Och Yoon
Blood Research.2024;[Epub] CrossRef
An unusual cause of erythroderma
Chng Wei Qiang, Benjamin Ho Wen Yang, Wang Shi, Lim Kok Hing, Laura Hui Li Yao
Clinical and Experimental Dermatology.2024; 50(1): 227. CrossRef
Indeterminate cell histiocytosis: A systematic review of the literature with a comprehensive revision of clinical, histopathological, and molecular features
Simone Zanella, Emilio Berti, Arturo Bonometti
Journal of the European Academy of Dermatology and Venereology.2023; 37(8): 1559. CrossRef

Follicular Dendritic Cell Sarcoma of the Inflammatory Pseudotumor-like Variant Presenting as a Colonic Polyp: Shien-Tung Pan, Chih-Yuan Cheng, Nie-Sue Lee, Peir-In Liang, Shih-Sung Chuang; Korean J Pathol. 2014;48(2):140-145. Published online April 28, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.2.140

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Abstract PDF

Follicular dendritic cell (FDC) sarcoma is rare and is classified either as conventional type or inflammatory pseudotumor (IPT)-like variant. Extranodal presentation is uncommon and nearly all gastrointestinal FDC tumors are of the conventional type. IPT-like variant tumors occur almost exclusively in the liver and spleen and are consistently associated with Epstein-Barr virus (EBV). Here we report the case of a 78-year-old woman with an IPT-like FDC sarcoma presenting as a pedunculated colonic polyp. Histologically, scanty atypical ovoid to spindle cells were mixed with a background of florid lymphoplasmacytic infiltrate, which led to an initial misdiagnosis of pseudolymphoma. These atypical cells expressed CD21, CD23, CD35, and D2-40, and were positive for EBV by in situ hybridization, confirming the diagnosis. The patient was free of disease five months after polypectomy without adjuvant therapy. Although extremely rare, the differential diagnosis for colonic polyp should include FDC sarcoma to avoid an erroneous diagnosis. A review of the 24 cases of IPT-like FDC sarcoma reported in the literature reveal that this tumor occurs predominantly in females with a predilection for liver and spleen, and has a strong association with EBV.

Citations

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Epstein-barr virus (EBV)-positive inflammatory pseudotumor-like follicular dendritic cell sarcoma (IPT-like FDCS) presenting as thrombocytopenia: A case report and literature review
Jiawei Jin, Xiaolong Zhu, Yi Wan, Yang Shi
Heliyon.2024; 10(12): e32997. CrossRef
EBV-positive inflammatory follicular dendritic cell sarcoma of the colon with clonal immunoglobulin gene rearrangement: A case report and literature review
Xia Xu, Xiuzhen Li, Qun Deng, Kaihang Yu, Jinfan Li
Heliyon.2024; 10(11): e31947. CrossRef
Challenges in the Diagnosis of Epstein-Barr Virus-positive Inflammatory Follicular Dendritic Cell Sarcoma
Yan Li, Xia Yang, Lili Tao, Weimei Zeng, Min Zuo, Shuo Li, Liyan Wu, Yanshong Lin, Ziying Zhang, Jingping Yun, Yuhua Huang
American Journal of Surgical Pathology.2023; 47(4): 476. CrossRef
Epstein-Barr Virus-Positive Inflammatory Follicular Dendritic Cell Sarcoma Presenting as a Colonic Polyp: Report of a Case with a Literature Review
Jiahui Hu, Dongdong Huang, Chengfu Xu, Yi Chen, Han Ma, Zhe Shen
Medicina.2023; 59(7): 1341. CrossRef
A Clinicopathology Review and Update of Epstein–Barr Virus-Associated Mesenchymal Tumors
Oswald Zhao Jian Lee, Noorjehan Omar, Joshua K. Tay, Victor Kwan Min Lee
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Granulomatous splenic mass with necrosis revealing an EBV-positive inflammatory follicular dendritic cell sarcoma
Irena Antonia Ungureanu, Renato Micelli Lupinacci, Marie Parrens, Jean-François Emile
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Case report: Hepatic inflammatory pseudotumor-like follicular dendritic cell sarcoma: A rare case and minireview of the literature
Fan Ding, Chao Wang, Chi Xu, Hui Tang
Frontiers in Medicine.2022;[Epub] CrossRef
Follicular dendritic cell sarcoma of gastrointestinal tract with two emerging distinct subtypes: a case report and systemic review
Hongxing Gui, Jigisha Chaudhari, Rifat Mannan
Diagnostic Pathology.2022;[Epub] CrossRef
Surgical treatment of liver inflammatory pseudotumor-like follicular dendritic cell sarcoma: A case report
Li-Yue Fu, Jiu-Liang Jiang, Meng Liu, Jun-Jun Li, Kai-Ping Liu, Hai-Tao Zhu
World Journal of Gastrointestinal Oncology.2022; 14(11): 2288. CrossRef
Inflammatory pseudotumor-like follicular/fibroblastic dendritic cell sarcoma: focus on immunohistochemical profile and association with Epstein-Barr virus
Francesca Pagliuca, Andrea Ronchi, Annamaria Auricchio, Eva Lieto, Renato Franco
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Raul S. Gonzalez, Anwar Raza, Robert Propst, Oyedele Adeyi, Justin Bateman, Sabrina C. Sopha, Janet Shaw, Aaron Auerbach
Archives of Pathology & Laboratory Medicine.2021; 145(5): 607. CrossRef
Hepatic inflammatory pseudotumor-like follicular dendritic cell tumor: a case report
Ana Daniela Pascariu, Andreea Ioana Neagu, Andrei Valentin Neagu, Alexandru Băjenaru, Cezar Iulian Bețianu
Journal of Medical Case Reports.2021;[Epub] CrossRef
Inflammatory pseudotumor-like follicular dendritic cell sarcoma: Literature review of 67 cases
Hao Wu, Peng Liu, Xiao-Ran Xie, Jing-Shu Chi, Huan Li, Can-Xia Xu
World Journal of Meta-Analysis.2021; 9(1): 1. CrossRef
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Xiang-Nan Jiang, Yan Zhang, Tian Xue, Jie-Yu Chen, Alex C.L. Chan, Wah Cheuk, John K.C. Chan, Xiao-Qiu Li
American Journal of Surgical Pathology.2021; 45(6): 765. CrossRef
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Hepatic inflammatory pseudotumor-like follicular dendritic cell tumor with hepatic lymphoma history
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Clinicopathological characteristics of extranodal follicular dendritic cell sarcoma: A report of two cases
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Inflammatory pseudotumour-like follicular dendritic cell tumour of the colon with plasmacytosis mimicking EBV-positive lymphoproliferative disorder
Ying-Ren Chen, Chi-Lin Lee, Yen-Chien Lee, Kung-Chao Chang
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Beware the inflammatory cell-rich colonic polyp: a rare case of EBV-positive inflammatory pseudotumour-like follicular dendritic cell sarcoma with increased IgG4-positive plasma cells
Lynne Goh, Nan Zun Teo, Lai Mun Wang
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Epstein–Barr virus‐positive inflammatory follicular dendritic cell sarcoma presenting as a solitary colonic mass: two rare cases and a literature review
Xiaokang Ke, Huihua He, Qingping Zhang, Jingping Yuan, Qilin Ao
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Bi-Xi Zhang, Zhi-Hong Chen, Yu Liu, Yuan-Jun Zeng, Yan-Chun Li
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Epstein-Barr virus (EBV)–associated lymphoid proliferations, a 2018 update
Sherif A. Rezk, Xiaohui Zhao, Lawrence M. Weiss
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Rossana Kazemimood, Farid Saei Hamedani, Asma Sharif, Sujata Gaitonde, Elizabeth Wiley, Pier Cristoforo Giulianotti, John Vincent Groth
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Case Report

Extranodal Follicular Dendritic Cell Sarcoma with Rapid Growth in Parapharynx: A Case Report: Jung-Soo Pyo, Guhyun Kang, Sung-Im Do, Seoung Wan Chae, Kyungeun Kim, Sang Hyuk Lee, Yoon-La Choi, Joon Hyuk Choi, Jin Hee Sohn, Dong-Hoon Kim; Korean J Pathol. 2012;46(3):306-310. Published online June 22, 2012; DOI: https://doi.org/10.4132/KoreanJPathol.2012.46.3.306

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Abstract PDF

Follicular dendritic cell sarcoma (FDCS) is a rare malignancy arising from the antigen-presenting cells in the lymph node and extranodal tissue. We describe a 31-year-old male patient who presented with a swelling of the left parapharynx. The radiologic findings showed a 4.7×4.5×1.9 cm-sized, ill-defined mass in the left parapharyngeal space. A fine-needle aspiration cytology was performed and it showed scattered, irregular, cohesive clusters of tumor cells with a spindle-to-ovoid shape with irregular contours in a background of lymphocytes. Based on these findings, a diagnosis of spindle cell neoplasm was made. The surgically resected tumor was composed of elongated, ovoid or polygonal cells showing positive immunohistochemistry for CD21, CD23, and CD35. Postoperatively, the residual tumor was observed to undergo a rapidly growth. There is an overlap in the cytologic and histologic findings between FDCS of the parapharynx and other tumors. Pathologists should therefore be aware of its characteristics not only to provide an accurate diagnosis but also to recommend the appropriate clinical management.

Citations

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Extranodal Follicular Dendritic Cell Sarcoma of the Head and Neck Region: A Clinicopathological Study of 7 Cases
Nasir Ud Din, Zubair Ahmad, Shabina Rahim, Karen Fritchie, Muhammad Usman Tariq, Arsalan Ahmed
International Journal of Surgical Pathology.2023; 31(6): 1067. CrossRef
Cytomorphology of follicular dendritic cell sarcoma: a report of 7 cases with an emphasis on the diagnostic challenges
Cody Weimholt, Jalal B. Jalaly, Cedric Bailey
Journal of the American Society of Cytopathology.2023; 12(3): 229. CrossRef
Follicular dendritic cells
Seham A. Abd El‐Aleem, Entesar Ali Saber, Neven M. Aziz, Hani El‐Sherif, Asmaa M. Abdelraof, Laiche Djouhri
Journal of Cellular Physiology.2022; 237(4): 2019. CrossRef
Clinicopathological characteristics of extranodal follicular dendritic cell sarcoma: A report of two cases
Xing Zhao, Dayong Sun, Gang Zhang
Oncology Letters.2021;[Epub] CrossRef
Cytological diagnosis of follicular dendritic cell sarcoma: A case report and review of literature
A. Dutta, P. Arun, P. Roy, I. Arun
Cytopathology.2018; 29(5): 461. CrossRef
Follicular dendritic cells and related sarcoma
Fabio Facchetti, Luisa Lorenzi
Seminars in Diagnostic Pathology.2016; 33(5): 262. CrossRef
Extranodal follicular dendritic cell sarcoma: A clinicopathological report of four cases and a literature review
RUI-FEN WANG, WEI HAN, LEI QI, LI-HUI SHAN, ZHENG-CAI WANG, LI-FENG WANG
Oncology Letters.2015; 9(1): 391. CrossRef
Follicular Dendritic Cell Sarcoma of Parapharyngeal Space: A Case Report and Review of the Literature
Turki Al-Hussain, Muhammad Saleem, Suresh Babu Velagapudi, Mohammad Anas Dababo
Head and Neck Pathology.2015; 9(1): 135. CrossRef
Clinical and pathological features of head and neck follicular dendritic cell sarcoma
Ji Li, Min-Li Zhou, Shui-Hong Zhou
Hematology.2015; 20(10): 571. CrossRef

Original Article

Distribution of Dendritic Cells and Regulatory T-Cells in Cutaneous Lymphomas.: Changyoung Yoo, Young Seon Hong, Baik Kee Cho, Sang Ho Kim, Sang In Shim, Chang Suk Kang; Korean J Pathol. 2010;44(6):581-588.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.6.581

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Abstract PDF: BACKGROUND
Dendritic cells (DCs) play an important role in immune reactions. This study was designed to identify the distribution patterns of DCs and regulatory T-cells (Tregs) in cutaneous lymphomas.
METHODS
Immunohistochemistry was used to determine langerin expression on Langerhans cells, CD11b on inflammatory DCs, CD209 and CD11c on dermal DCs, CD303 on plasmacytic DCs, and Foxp3 on Tregs in 81 cases of cutaneous lymphomas.
RESULTS
Various DCs and Tregs were identified in most cutaneous lymphomas. Plasmacytic DCs, inflammatory DCs and Tregs were identified mainly in tumor areas, whereas dermal DCs were distributed both in the tumor and stromal areas. Among DCs, dermal DCs were most prominently identified in the cutaneous lymphomas not only in the tumor area but also in the stroma. The intense stromal infiltration of dermal DCs was consistent finding in T-cell lymphomas. Diffuse large B-cell lymphoma (DLBCL), not otherwise specified also showed intense stromal infiltration of dermal DCs, but stromal infiltration in DLBCL, leg type was relatively scant.
CONCLUSIONS
The results suggest that all types of DCs and Tregs are involved in cutaneous lymphoma tumor immunity. Among them dermal DCs may play a dominant role.

Case Report

Cytology of Follicular Dendritic Cell Sarcoma on Intraoperative Touch Imprint Smears: A Case Report.: Ju Young Song, Xian Ji Jin, Jee Young Han, Lucia Kim, In Suh Park, Joon Mee Kim, Young Chae Chu, Suk Jin Choi; Korean J Pathol. 2009;43(6):589-593.; DOI: https://doi.org/10.4132/KoreanJPathol.2009.43.6.589

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Abstract PDF

Follicular dendritic cell (FDC) sarcoma is a neoplastic proliferation of FDCs. Because its cytologic findings can vary widely, both the cytomorphology and histopathology of FDC sarcoma can impose a significant diagnostic dilemma. We present cytologic features of FDC sarcoma assessed by intraoperative touch imprint. Intra-abdominal lymphadenopathies were noted in 54-year-old male with hepatitis B-virus associated liver cirrhosis. In contrast to cytologic features of classical FDC sarcoma, the tumor cells featured a large epithelioid or Reed-Sternberg cell-like shape scattered in a background with abundant inflammatory cells, which led to a mistaken diagnosis of malignant lymphoma. However, in accordance with cytologic features previously described in the literature, the tumor cells were characterized by a fragile cytoplasm with cytoplasmic processes in dendritic or reticulated patterns reminiscent of the ultrastructural features of FDC. Cytoplasmic features rendering nuclei with a tendency to form clusters or syncytial aggregates associated with reactive lymphocytes appear to be the most valuable finding in diagnosis of FDC sarcoma.

Citations

Citations to this article as recorded by

Cytomorphology of follicular dendritic cell sarcoma: a report of 7 cases with an emphasis on the diagnostic challenges
Cody Weimholt, Jalal B. Jalaly, Cedric Bailey
Journal of the American Society of Cytopathology.2023; 12(3): 229. CrossRef
A Cytological Review of Follicular Dendritic Cell-Derived Tumors with Emphasis on Follicular Dendritic Cell Sarcoma and Unicentric Castleman Disease
José A. Jiménez-Heffernan, Cristina Díaz del Arco, Magdalena Adrados
Diagnostics.2022; 12(2): 406. CrossRef
Cytological diagnosis of follicular dendritic cell sarcoma: A case report and review of literature
A. Dutta, P. Arun, P. Roy, I. Arun
Cytopathology.2018; 29(5): 461. CrossRef

Original Article

S100 Protein Positive Dendritic Cells in Liver Diseases.: Ghil Suk Yoon, Inchul Lee, Eunsil Yu; Korean J Pathol. 1998;32(8):590-595.

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Abstract: We describe S100 protein positive dendritic cells (S100+DCs) in various liver diseases including chronic viral hepatitis B and C (20 cases), liver cirrhosis (3 cases), hepatocellular carcinoma (2 cases), hepatolithiasis (6 cases), primary biliary cirrhosis (PBC) (2 cases), liver allograft rejection (9 cases), bile duct paucity (1 case), and Wilson's disease (1 case). By immunohistochemical analysis, S100+DCs were absent in fetal and normal livers, while they were variably present in inflammatory liver diseases. In chronic hepatitis and active cirrhosis, S100+DCs were most frequently located in periportal area, at lymphoid follicles within the portal tract, and at foci of spotty necrosis within the lobule. Frequency and intensity of S100+DCs were not related to etiologies of liver diseases, but they were correlated with the activity of hepatitis. In PBC, S100+DCs were found between biliary epithelial cells of the septal bile ducts, as well as, the periductal area of the portal tracts. A posttransplantation liver with features of moderate acute rejection revealed many S100+DCs in polymorphous portal infiltrates. In hepatocellular carcinomas, many S100+DCs were scattered between tumor cells. In the case of the Wilson's disease, S100+DCs were not noted. Presence of S100+DCs in various inflammatory liver diseases indicates that they play a central role as antigen presenting cells in immune responses of inflammatory liver diseases.

Case Report

Epstein-Barr virus-associated Inflammatory Pseudotumor-like Follicular Dendritic Cell Tumor in the Spleen of a Patient with Diffuse Large B Cell Lymphoma: A Case Report and Review of the Literature.: Sun Och Yoon, Hyoungsuk Ko, Baek hui Kim, Ghee Young Kwon, Yoon Kyung Jeon, Chul Woo Kim; Korean J Pathol. 2007;41(3):198-202.

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Abstract PDF: We report a case of an Epstein-Barr virus (EBV)-associated inflammatory pseudotumor-like follicular dendritic cell tumor (IPT-like FDC tumor). The tumor occurred in the spleen of a 64-year-old woman with a history of a diffuse large B-cell lymphoma (DLBCL) of neck nodes that presented four years ago. The splenectomy specimen revealed a 5 cm-sized, tan-colored and well-circumscribed mass. Histologically, spindle or ovoid cells with large vesicular nuclei were admixed with abundant inflammatory cells. Immunohistochemically, spindle cells were positive for FDC marker CD35, but negative for CD20, CD30 and ALK. EBV was detected almost exclusively in spindle cells by EBER in situ hybridization. IPT-like FDC tumors are rare, and are recognized as a distinctive clinicopathologic variant of FDC tumors. Among only 18 similar cases reported in the English language literature, the present case is the first case of a patient with a history of DLBCL.

Original Article

Macrophage/dendritic Cell Marker Staining Characteristics of Langerhans cell Granulomatosis(Histiocytosis X).: Sang Ae Yoon, In Sun Kim; Korean J Pathol. 1992;26(3):310-313.

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Abstract PDF: Histiocytosis X is characterized by aggregates of Langerhans cells with other inflammatory cells. These Langerhans cells are antigen-presenting cells to T lymphocytes and identified by characteristic morphology, ultrastructural demonstration of Birbeck granules and immunologic reactivity with OKT-6 and HLA-DR antibodies. In this report, the tumor arising in a 2-years-old baby was examined byimmunostaining with several macrophage/dendritic cell markers. The main tumor cells showed cytoplasmic and nuclear staining with S-100 protein and ring-like surface and paranuclear staining with PNA. However, they were negative for follicular dendritic cell marker CD21, macrophage markers lysozyme, Mac 387, alpha-1 antitrypsin and CD68, and interdigitating reticulum cell marker ID4 and ID5. These observations demonstrate the usefulness of S-100 protein and PNA for the identification of Langerhans cells in paraffin-embedded tissue.

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