Journal of Pathology and Translational Medicine

Case Study

Intravascular NK/T-cell lymphoma: a case report and literature review: Ji Min Na, Wookjae Jung, Minhye Kim, Yun-Hong Cheon, Jong Sil Lee, Dae Hyun Song, Jung Wook Yang; J Pathol Transl Med. 2023;57(6):332-336. Published online November 14, 2023; DOI: https://doi.org/10.4132/jptm.2023.10.30

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Intravascular lymphoma is characterized by an exclusively intravascular distribution of tumor cells. Intravascular natural killer/T-cell lymphoma (IVNKTL) is extremely rare, highly aggressive, commonly Epstein-Barr virus (EBV)–positive, and predominantly affects the skin and central nervous system. Here we report a case of IVNKTL diagnosed in a 67-year-old female, presenting with persistent intermittent fever and skin rashes throughout the body. Incisional biopsy of an erythematous lesion on the chest exhibited aggregation of medium to large-sized atypical lymphoid cells confined to the lumen of small vessels that were positive for CD3, granzyme B, and CD56 on immunohistochemistry and EBV-encoded RNA in situ hybridization. EBV DNA was also detected in serum after diagnosis. With a review of 26 cases of IVNKTL to date, we suggest that active biopsy based on EBV DNA detection may facilitate early diagnosis of IVNKTL.

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Cutaneous Intravascular Hematolymphoid Entities: A Review
Emily Hatheway Marshall, Bethany Brumbaugh, Allison Holt, Steven T. Chen, Mai P. Hoang
Diagnostics.2024; 14(7): 679. CrossRef

Case Reports

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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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.

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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

Extranodal NK/T Cell Lymphoma Accompanied by Heavy Eosinophilic Infiltration and Peripheral Blood Eosinophilia, Involving Skeletal Muscles.: Jin Ho Paik, Yoon Kyung Jeon, Heounjeong Go, Chul Woo Kim; Korean J Pathol. 2011;45:S70-S74.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S70

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Abstract PDF

The patient was a 52-year-old female with swelling in both lower legs and peripheral blood eosinophilia. Biopsy specimen revealed the heavy infiltration of eosinophils with sparse small lymphocytes showing mild atypia. The diagnosis was Kimura disease. The symptoms including eosinophilia were relieved by steroid treatment. At 17 months from initial biopsy, the patient developed swelling of the buttock. At 25 months, fever and dyspnea with multiple lung nodules developed. Wedge resection revealed multiple aggregates of CD3(+), CD56(+), Epstein-Barr virus(+) large atypical lymphocytes with necrosis. The patient was finally diagnosed with extranodal NK/T cell lymphoma (NKTL). Epstein-Barr virus in situ hybridization retrospectively performed on the previous biopsies demonstrated Epstein-Barr virus infection in small CD3(+) lymphocytes. The patient expired after 26 months despite chemotherapy. Blood eosinophilia correlated well with disease activity during the clinical course. This case shows not only unusual histologic features, which hampered the correct diagnosis, but also a unique clinical manifestation of NKTL.

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Muscular involvement of extranodal natural killer/T cell lymphoma misdiagnosed as polymyositis: A case report and review of literature
Li-Hui Liu, Qing Huang, Yun-Hai Liu, Jie Yang, Han Fu, Lin Jin
World Journal of Clinical Cases.2020; 8(5): 963. CrossRef
Extranodal natural killer/T-cell lymphoma with paraneoplastic eosinophilic myositis
Jayati Mallick, Jasmine Zain, Dennis D. Weisenburger
Human Pathology: Case Reports.2020; 21: 200391. CrossRef
Extranodal NK/T-cell Lymphoma Mimicking Granulomatous Myositis
Norihiko Kawaguchi, Rumiko Izumi, Masahiro Kobayashi, Maki Tateyama, Naoki Suzuki, Fumiyoshi Fujishima, Juichi Fujimori, Masashi Aoki, Ichiro Nakashima
Internal Medicine.2019; 58(2): 277. CrossRef
Uveitis and Myositis as Immune Complications in Chemorefractory NK/T-Cell Nasal-Type Lymphoma Successfully Treated with Allogeneic Stem-Cell Transplant
Maria José Gómez-Crespo, Aránzazu García-Raso, Jose Luis López-Lorenzo, Teresa Villaescusa, María Rodríguez-Pinilla, José Fortes, Cristina Serrano, Salma Machan, Pilar Llamas, Raúl Córdoba
Case Reports in Hematology.2016; 2016: 1. CrossRef
Prognostic implications of CD30 expression in extranodal natural killer/T-cell lymphoma according to treatment modalities
Wook Youn Kim, Soo Jeong Nam, Sehui Kim, Tae Min Kim, Dae Seog Heo, Chul-Woo Kim, Yoon Kyung Jeon
Leukemia & Lymphoma.2015; 56(6): 1778. CrossRef
Unusual case of metachronous EBV‐associated B‐cell and NK/T‐cell lymphoma mimicking polymyositis‐diagnostic challenges and pitfalls
Esther H.L. Chan, Suat‐Jin Lu, Fredrik Petersson, Kong‐Bing Tan, Wee‐Joo Chng, Siok‐Bian Ng
American Journal of Hematology.2014; 89(1): 110. CrossRef
CD30+ extranodal natural killer/T-cell lymphoma mimicking phlegmonous myositis: A case report
YAN-JIA YANG, YA-XIN LI, YAN-BIN LIU, MEI YANG, KAI LIU
Oncology Letters.2014; 7(5): 1419. CrossRef

Fine Needle Aspiration Cytology of Extranodal Marginal Zone B cell Lymphoma with Abundant Plasma Cells and Eosinophilic Histiocytes in Parotid Gland.: Youngseok Lee, Jungsuk An, Yang Seok Chae, Bom Woo Yeom, Jong Sang Choi, Chul Hwan Kim; Korean J Cytopathol. 2007;18(2):165-169.

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Abstract PDF: The authors present the fine needle aspiration cytology (FNAC) cytologic findings of a case of extranodal marginal zone B cell lymphoma (MZBCL), which featured abundant plasma cells and eosinophilic histiocytes arising in both parotid glands. A 49-year-old female presented with palpable masses in both parotid glands. She had been suffering from systemic lupus erythematosus and rheumatoid arthritis. The lesions were evaluated by FNAC and smears showed a small number of clusters of oncocytic cells with abundant eosinophilic granular cytoplasm and small nuclei, intermixed with small to medium-sized lymphoid cells containing round to lobulated nuclei, which suggested Warthin's tumor. Some of lymphoid cells had a plasmacytoid appearance, and some scattered large cells contained a large amount of eosinophilic cytoplasm. Bilateral superficial parotidectomy was performed and a histopathologic study indicated MZBCL with abundant plasma cells, intermixed with eosinophilic histiocytes. The presence of oncocytic cells and a mixture of lymphoid and plasma cells indicates Warthin's tumor, but the cytologic features of a relatively monotonous small to medium-sized lymphoid infiltrate suggest the possibility of MZBCL in the clinical setting of an FNAC study performed on a patient suffering from a connective tissue disease.

Ki-1 Positive Extranodal NK/T Cell Lymphoma, Nasal Type, Mistaken as Dermatomyositis: A Case Report and Literature Review.: Hyun Jung Kim, Eunah Shin, Jung Yeon Kim, Kyeongmee Park, Young Jin Yoo, Seung Sook Lee; Korean J Pathol. 2007;41(4):278-283.

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Abstract PDF: We report a case of a 38-year-old male with extranodal NK/T-cell lymphoma, nasal type, showing unusual clinical and pathological features. The patient was admitted for soft tissue swelling and tenderness in both legs. The patient had been treated intermittently 8 months prior for repeated muco-cutaneous ulcers. A muscle biopsy showed medium-sized atypical lymphoid cells with bizarre nuclei and plump cytoplasm, infiltrating to the skeletal muscle fibers with angiocentricity. The immunoresults were Ki-1+, CD56+, cytoplasmic CD3+, with EBV-in situ hybridization +. The patient rapidly deteriorated and died of sepsis and respiratory failure shortly after initiation of low-dose chemotherapy. A careful review of previous biopsies revealed scarce atypical lymphoid cells around vessels with similar immunoprofiles without the presence of Ki-1 positive cells. This case emphasizes that an extranodal NK/T-cell lymphoma may have a dermatomyositis-like diffuse presentation. Ki-1 co-expression can be an unexpected event in a process of the disease course; however, this should be validated with future studies.

Nesal T-cell Lymphoma associated with Hemophagocytic Syndrome: A case report.: Mee Sook Roh, Jin Sook Jeong, Sook Hee Hong; Korean J Pathol. 1994;28(5):541-543.

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Abstract PDF: Peripheral T-cell lymphoma is the generic group given to a family of tumors composed of neoplastic lymphocytes with phenotypic features of peripheral T-cells. Certain peripheral T-cell lymphomas develop a hemophagocytic syndrome that mimics malignant histiocytosis, both clinically and pathologically. We experienced a case of nasal T-cell lymphoma, histologically mimicking malignant histiocytosis in a 40-year-old male. The chief complaints were nasal obstruction and intermittent mild fever. Mild anemia, elevated SGOT and SGPT, polyclonal gammophthy, and moderate hepatomegaly were present. Two weeks later was present an enlarged cervical lymph node. The biopsied nasal mass showed angiocentric and angiodestructive peripheral T-cell lymphoma withextensive necrosis and marked erythrophagocytosis by non-neoplastic histiocytes. Subsequently, cervical lymph node was biopsied, which showed peripheral T-cell ltmphoma with extensive necrosis and erythrophagocytosis as well. The atypical lymphoid cells revealed pan-T(+), but CD4(-) and CD8(-), whereas the reactive histiocytes showed lysozyme(+), immunohistochemistry.

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