Angioimmunoblastic T-cell lymphoma with autoimmune thrombocytopenia: A report of two cases

Oka, Kuniyuki; Nagayama, Reizo; Yatabe, Yasushi; Iijima, Shigeruko; Mori, Naoyoshi

doi:10.1016/j.prp.2009.04.002

Cited by 8 publications

(7 citation statements)

References 13 publications

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“…It has been speculated that tumor cells produce interleukin (IL)-6, which acts on the B cells, leading to hyperg-globulinemia, production of autoantibodies and development of ATP. 13 In the present case, however, the platelet count did not increase even at the time of complete remission of the AITL, despite the serum IL-6 level being within the normal range. Other unusual characteristics of our patient were that at the time of the relapse of the AITL, the tumor cells became negative for CD56, and showed complex additional chromosomal abnormalities and the morphological characteristics of blast cells.…”

Section: Introductioncontrasting

confidence: 71%

“…Various immunological abnormalities, including circulating immune complex positivity, Coombs positivity, production of autoantibodies such as antinuclear antibodies, hemolytic anemia, cold agglutinin positivity and polyclonal hyperg-globulinemia, have been reported in patients with AITL. 15 It is speculated that the tumor cells produce IL-6, which acts on the B cells, resulting in hyper-g-globulinemia and production of autoantibodies, 8,9,13 and consequently, in the development of ATP. Production of cytokines, such as tumor necrosis factor-a/b, IL-1, IL-4, IL-6 and interferon-g, has also been reported.…”

Section: Discussionmentioning

confidence: 99%

“…In addition, AITL associated with autoimmune thrombocytopenia (ATP) is also rare, and only 14 cases, including our present case, have been reported so far. [4][5][6][7][8][9][10][11][12][13] Furthermore, only in the present patient was the ATP a component of Evans syndrome. It has been speculated that tumor cells produce interleukin (IL)-6, which acts on the B cells, leading to hyperg-globulinemia, production of autoantibodies and development of ATP.…”

Section: Introductionmentioning

confidence: 97%

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CD56+ Angioimmunoblastic T-Cell Lymphoma With Evans Syndrome : A Case Report and Review of the Literature

Sekiguchi

Shimada

Imai

et al. 2013

J Clin Exp Hematopathol

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A 67-year-old man was diagnosed with CD56(+) angioimmunoblastic T cell lymphoma (AITL), which was associated with autoimmune thrombocytopenic purpura (ATP) and autoimmune hemolytic anemia (AIHA) (Evans syndrome). The ATP was refractory to Helicobacter pylori eradication therapy and steroid. Complete remission (CR) of both AITL and AIHA was achieved with THP-COP chemotherapy (pirarubicin, cyclophosphamide, vincristine, and prednisolone), but ATP was not improved promptly. AITL associated with ATP has been reported in only 14 cases. The present case was not related to the serum interleukin-6 levels, suggesting the possibility of an association with other factors. This case is the first report of Evans syndrome associated with AITL. The AITL relapsed 2 months after CR. The AITL tumor were CD56-positive at initial diagnosis and CD56-negative at relapse, and showed complex additional chromosomal abnormalities, and the morphological characteristics of blast cells. CD56(+) AITL are rare, although CD56 expression has not been investigated in many cases ; our observations suggest that CD56 expression and its significance in AITL should be investigated in the future. There has been only one other case of CD56(+) AITL, the patient died 4 months after the diagnosis. Our patient reported showed early relapse, central nervous system infiltration and was refractory to treatment, suggesting that CD56 positivity may be a poor prognostic marker in patients with AITL. [J Clin Exp Hematop 53(1): 37-47, 2013].

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Section: Introductioncontrasting

confidence: 71%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 97%

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CD56+ Angioimmunoblastic T-Cell Lymphoma With Evans Syndrome : A Case Report and Review of the Literature

Sekiguchi

Shimada

Imai

et al. 2013

J Clin Exp Hematopathol

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“…The possible mechanism that may explain the relation between AITL and PAN is represented by the induction of an autoimmune phenomenon by the lymphoma. It is well known that AITL cells produce cytokines such as interleukin 6 [31] and TNF-α [16], stimulating polyclonal B-cells and plasma cells to secrete antibodies forming circulating immune complexes (CIC). CIC complete the complement cascade, releasing vasoactive substances and chemotactic factors that cause accumulations of inflammatory cells and release of lysosomal enzymes by neutrophils, leading to injury of vessels walls and thrombosis [17].…”

Section: Discussionmentioning

confidence: 99%

Renal infarction due to polyarteritis nodosa in a patient with angioimmunoblastic T-cell lymphoma: a case report and a brief review of the literature

et al. 2012

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Angioimmunoblastic T-cell lymphoma is one of the most common subtypes of peripheral T-cell lymphoma (15-20% of all cases), accounting for approximately 1-2% of all non-Hodgkin lymphomas. It often presents autoimmune phenomena including hemolytic anemia, thrombocytopenia, glomerulonephrities and circulating immune complexes. Polyarteritis nodosa is an autoimmune disease characterized by necrotizing vasculitis of medium vessels, which rarely develops in association with hematological malignant disorders. Herein we report the case of a 40-year-old man who underwent lymph node biopsy in the suspicious of sarcoidosis. On the basis of histological and immunohistochemical findings, the diagnosis of angioimmunoblastic T-cell lymphoma was performed. The patient was successfully treated with cytarabine-based regimen for 6 cycles. Three months after the initial diagnosis of angioimmunoblastic T-cell lymphoma, a whole body computed tomography showed a lesion in the lower pole of the left kidney. Renal cell carcinoma was suspected, thus a nephrectomy was carried out. The histological findings were compatible with polyarteritis nodosa. To the best of our knowledge, the association between polyarteritis nodosa and angioimmunoblastic T-cell lymphoma has been described only once. This relation may be secondary to the induction of an autoimmune phenomenon by the lymphoma with the formation of circulating immune complexes, leading to vessels walls injury. A careful evaluation is needed in the management of angioimmunoblastic T-cell lymphoma patients with signs of renal failure in order to avoid delay of treatment and organ damage.

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“…1 It affects mainly people in the sixth and seventh decades of life (mean age of 59-64 years old) and is usually presented as a systemic disease. 3 Its prevalent among men, with a male-female ratio of 2:1. 1,2 AITL was initially described as angioimmunoblastic lymphadenopathy with dysproteinemia, and defined as a clinical syndrome characterized by generalized Linfoma de células t angioimunoblástico em paciente idoso e com diagnóstico prévio de Angioimmunoblastic T-cell lymphoma in elderly patient with a previous diagnosis of infectious mononucleosis: a challenge ...…”

Section: Introductionmentioning

confidence: 99%

Angioimmunoblastic T-cell lymphoma in elderly patient with a previous diagnosis of infectious mononucleosis: a challenge for cancer treatment - case report

Soares¹,

Brandão²,

Mata³

et al. 2015

Revista Médica De Minas Gerais

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Angioimmunoblastic T-cell lymphoma (AITL) is a rare subtype of peripheral T-cell lymphoma that represents about 2% of non-Hodgkin's lymphomas (LNH). It affects mainly men, in the sixth and seventh decades of life, and appears as a systemic disease. It has an aggressive behavior and responds poorly to chemotherapy, still with undefined standard treatment. This report aims to provide the poor prognosis in patients with AITL and the lack of treatment consolidation. The factors that may be associated with the poor response are being elderly, advanced stages, and constitutional symptoms.

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Angioimmunoblastic T-cell lymphoma with autoimmune thrombocytopenia: A report of two cases

Cited by 8 publications

References 13 publications

CD56+ Angioimmunoblastic T-Cell Lymphoma With Evans Syndrome : A Case Report and Review of the Literature

CD56+ Angioimmunoblastic T-Cell Lymphoma With Evans Syndrome : A Case Report and Review of the Literature

Renal infarction due to polyarteritis nodosa in a patient with angioimmunoblastic T-cell lymphoma: a case report and a brief review of the literature

Angioimmunoblastic T-cell lymphoma in elderly patient with a previous diagnosis of infectious mononucleosis: a challenge for cancer treatment - case report

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