Angioimmunoblastic T-Cell Lymphoma Initially Presenting with Replacement of Bone Marrow and Peripheral Plasmacytosis

Sakai, Hisashi; Tanaka, Hideaki; Katsurada, Tatsuya; Yoshida, Yataro; Okamoto, Eiichi; Ohno, Hitoshi

doi:10.2169/internalmedicine.46.6121

Cited by 18 publications

(13 citation statements)

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“…The reactive plasmacytes commonly exhibited a CD19 + , CD38 + , CD138 -/+ , CD10 -, CD20 -, and CD56 -immunophenotype (Sakai et al 2007;Yamane et al 2007;Ahsanuddin et al 2011;Nagoshi et al 2013). In addition, 4 of 12 patients demonstrated proliferation of monoclonal plasma cells that were relatively mature in morphology; that originated from EBV-negative clones; and that were always associated with AITL lesions at the lymphoma site, but not in the bone marrow (Zettl et al 2002;Balague et al 2007;Huppmann et al 2013).…”

Section: Discussionmentioning

confidence: 99%

“…However, the potential mechanism involved in the development and progression of the concomitant plasma cell proliferation remains to be clarified. High levels of cytokines, such as IL-6, IL-10 and tumor necrosis factor-α, may serve as possible contributing factors (Balague et al 2007;Sakai et al 2007;Yamane et al 2007;Ahsanuddin et al 2011;Huppmann et al 2013;Nagoshi et al 2013). …”

Section: Discussionmentioning

confidence: 99%

“…Histopathologically, AITL derives from a unique subset of T cells which are called the follicular helper T (TFH) cells, and displays distinctive features that are characterized by effacement of the lymph node architecture, a paracortical polymorphic infiltrate, the proliferation of arborizing high endothelial venules (HEVs) and follicular dendritic cells (FDCs), and occasional background expansion of Epstein-Barr virus (EBV)-harboring B immunoblasts (Dogan et al 2003(Dogan et al , 2008Dunleavy et al 2007). Recently, several research groups focused on the behavior of plasma cells in AITL, which ranged from reactive plasmacytosis to striking clonal proliferation (Balague et al 2007;Sakai et al 2007;Yamane et al 2007;Ahsanuddin et al 2011;Huppmann et al 2013;Nagoshi et al 2013). However, the role of the plasma cell as a component of the microenvironment in AITL and the functional interaction between plasma cells and tumor cells have not yet been deciphered (Gaulard and de Leval 2014).…”

Section: Introductionmentioning

confidence: 99%

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Angioimmunoblastic T-Cell Lymphoma with Coexisting Plasma Cell Myeloma: A Case Report and Review of the Literature

Tang

Zhao

et al. 2015

Tohoku J. Exp. Med.

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Angioimmunoblastic T-cell lymphoma (AITL) is recognized as a distinct clinicopathological subtype of peripheral T-cell lymphomas. Its clinical features include generalized lymphadenopathy, constitutional symptoms, and autoimmune-related findings, such as hemolytic anemia. Pathologically, AITL is characterized by a polymorphous infiltrate in lymph nodes with prominent proliferation of high endothelial venules and follicular dendritic cells. We present an 80-year-old Chinese man with generalized lymphadenopathy and pulmonary infection, diagnosed as AITL based on the distinctive pathological findings and T-cell receptor gamma (TCR-γ) gene rearrangement analysis of lymph nodes. Importantly, the patient suffered from a coexisting plasma cell myeloma, as judged by monoclonal immunoglobulin in the serum, immature plasma cells, and rearrangement of the immunoglobulin heavy-chain (IgH) gene in the bone marrow. The patient received two courses of the chemotherapy but died of pneumonia 6 months after diagnosis. AITL can be accompanied by polyclonal or clonal proliferation of B lymphocytes; however, AITL are rarely associated with plasma cell proliferation. In fact, 14 AITL cases with plasma cell proliferation have been reported in the literature, but none of them manifested the infiltration of monoclonal immature plasma cells in the bone marrow. To the best of our knowledge, this is the first report of newly diagnosed, concurrent AITL and plasma cell myeloma, providing the evidence for the interplay between malignant T cells and plasma cell proliferation. A review of the literature has also supported a relationship between AITL and plasma cell proliferation. Awareness of this relationship is important for correct diagnosis and appropriate treatment of AITL.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Angioimmunoblastic T-Cell Lymphoma with Coexisting Plasma Cell Myeloma: A Case Report and Review of the Literature

Tang

Zhao

et al. 2015

Tohoku J. Exp. Med.

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“…A number of cases of AITL with increased plasmacytoid cells in the peripheral blood, which is typical of plasma cell leukemia, have been reported (3)(4)(5). In these reports, the plasmacytoid cell counts in the peripheral blood were markedly elevated, although they did not exhibit clonal expansion.…”

Section: Discussionmentioning

confidence: 99%

“…Patients with AITL, frequently exhibit anemia, thrombocytopenia and lymphopenia in the peripheral blood. However, it is rare for patients with AITL to have numerous plasma cells in the peripheral blood, which is a feature more typical of plasma cell leukemia (3)(4)(5). The current report presents a case of a patient with AITL, in whom leukemic change and plasmacytosis were observed in the peripheral blood, bone marrow and lymph nodes.…”

Section: Introductionmentioning

confidence: 99%

A case of CD10-negative angioimmunoblastic T cell lymphoma with leukemic change and increased plasma cells mimicking plasma cell leukemia: A case report

et al. 2015

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Abstract. Angioimmunoblastic T cell lymphoma (AITL) is a peripheral T cell lymphoma, known to express CD3 and CD4, and, frequently, also CD10 and c-Maf-1. Hypergammaglobulinemia is not particularly rare in patients with AITL. However, AITL in conjunction with plasmacytosis in the peripheral blood is rare. The current report presents a case of CD10-negative AITL demonstrating leukemic change and plasmacytosis in the peripheral blood mimicking plasma cell leukemia. A 78-year-old male was admitted to hospital due to systemic lymph node enlargement, high serum IgG and IgA, and increased counts of plasmacytoid cells and lymphoid cells with atypical nuclei in the peripheral blood. Initially, plasma cell leukemia was suspected, due to the extreme increase in the number of plasma cells in the peripheral blood. However, the plasma cells did not show clonal expansion on examination by flow cytometry. Based on histological analyses, following a biopsy of an enlarged lymph node, the patient was diagnosed with AITL. This case suggests that when hypergammaglobulinemia and increases in B-lineage cells are observed, AITL should be considered in addition to disorders of B-lineage cells. IntroductionAITL is the second most frequent subtype of peripheral T cell lymphoma in the Western world, accounting for ~25-30% of peripheral T cell lymphoma cases and 2-4% of all cases of lymphoma (1). It has been reported that AITL presents with a number of clinical symptoms, including fever, weight loss, chills, skin rashes, pruritis, lymphadenopathy, hepatosplenomegaly, anemia, thrombocytopenia and hypergammaglobulinemia (2). Patients with AITL, frequently exhibit anemia, thrombocytopenia and lymphopenia in the peripheral blood. However, it is rare for patients with AITL to have numerous plasma cells in the peripheral blood, which is a feature more typical of plasma cell leukemia (3-5). The current report presents a case of a patient with AITL, in whom leukemic change and plasmacytosis were observed in the peripheral blood, bone marrow and lymph nodes. Case reportA 78-year-old male was admitted to hospital, due to systemic lymph node swelling and an elevated number of white blood cells in comparison with normal levels, which had been detected by his family doctor 10 days previously. Upon physical examination, lymph nodes in the cervical region, supraclavicular fossa, axillary fossa and inguinal fossa were found to be enlarged to 40 mm. The peripheral white blood cell count was elevated and numerous plasmacytoid cells without dysmorphic features, in addition to small-to-medium-sized lymphoid cells with atypical nuclei, were observed in his peripheral blood, as shown in Table I, Fig. 1 and Fig. 2A and B. 'Other' in the leukocyte classification in Table I, includes plasmacytoid cells (19% of whole white blood cells) and lymphoid cells with nuclear atypia (22% of whole white blood cells). Anemia and low platelet numbers were also observed.

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2021

Flow Cytometry of Hematological Malignancies

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Angioimmunoblastic T-Cell Lymphoma Initially Presenting with Replacement of Bone Marrow and Peripheral Plasmacytosis

Abstract: Abstract

Cited by 18 publications

References 10 publications

Angioimmunoblastic T-Cell Lymphoma with Coexisting Plasma Cell Myeloma: A Case Report and Review of the Literature

Angioimmunoblastic T-Cell Lymphoma with Coexisting Plasma Cell Myeloma: A Case Report and Review of the Literature

A case of CD10-negative angioimmunoblastic T cell lymphoma with leukemic change and increased plasma cells mimicking plasma cell leukemia: A case report

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