Aplastic Anemia and Idiopathic Thrombocytopenic Purpura with Antibody to Platelet Glycoprotein llb/llla Following Resection of Malignant Thymoma

Kobayashi, Hikaru; Kitano, Kiyoshi; Ishida, Fumihiro; Saitô, Hiroshi; Miyabayashi, Hideharu; Yonezawa, Takanori; Inada, Haruhiko; Suzuki, Akihiko; Furuta, Seiichi

doi:10.1159/000204372

Cited by 20 publications

(14 citation statements)

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“…In our patient, an attack on both stem cells and erythroid precursors by either CD8c cytotoxic T lymphocytes and CD16c natural killer cells is suspected, and serum inhibitor was demonstrated. Although we lack studies on the pathogenesis of TA-AA [24], based on the current literature, most PRCA and AA associated with thymomas seem to be caused by marrow suppression related to unbalanced T cell regulation [10,13,14,16,19,21,[27][28][29][30][31]. An excess of blood CD8c T lymphocytes, as observed in our patient, represents the most consistent finding [16,17,21,27,28], while serum erythropoietic inhibitors [29,30], and/or inhibitory effects of T lymphocytes on BFU-E and CFU-E [16,27,30] can be established in a few patients.…”

Section: Discussionmentioning

confidence: 98%

“…Hematologic dyscrasias, including PRCA and AA, are the hallmark of spindle-cell thymomas, including or not a lymphocytic component [8,17,24], often of the cortical phenotype [18,24]. Malignant thymomas can also be involved [6,10,31].…”

Section: Discussionmentioning

confidence: 99%

“…In fact, the overall long-term survival of patients with a thymoma is poor once cytopenias occur [3,8,17]. ALG and Cy-A, often successful in primary [5,7,10,17,23] as well as secondary chronic PRCA [15] and idiopathic AA [7,32,33], should be proposed in unresponsive cases. ALG, which resulted in permanent hematological remission in two cases of TA-AA [14,31], proved totally ineffective in our patient.…”

Section: Discussionmentioning

confidence: 99%

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Thymoma-associated pancytopenia: effectiveness of cyclosporine A

Liozon

Touati

Allegraud

et al. 1998

Annals of Hematology

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Aplastic anemia is a rare complication of thymoma and is properly documented in only few cases. Here, we report the case of a previously healthy, 65-year-old patient who was found simultaneously to have a spindle-cell thymoma and severe hypoplastic anemia with a mild infiltration of the bone marrow by CD4+ and CD8+ T lymphocytes, CD16+ natural killer cells, and a decrease in blood CD4/CD8 ratio. Cultures of marrow erythroid progenitors demonstrated serum inhibitor. While steroids, cyclophosphamide and antilymphocyte globulin failed to improve hematopoiesis, cyclosporine A (Cy-A) led to a partial, stable remission that was sustained for 4 years. Since Cy-A has been associated with good responses in three cases of thymoma-associated red cell aplasia, we recommend its use in cases of thymoma-associated cytopenias refractory to steroids and cyclophosphamide.

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Section: Discussionmentioning

confidence: 98%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Thymoma-associated pancytopenia: effectiveness of cyclosporine A

Liozon

Touati

Allegraud

et al. 1998

Annals of Hematology

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“…Aplastic anemia usually precedes the diagnosis of thymoma. Th ere have been only three cases described in the literature of aplastic anemia occurring as a late complication after thymectomy with the time interval between 3 and 48 months [1][2][3] . Ritchie et al described a single case of MG and aplastic anemia after thymectomy and remission of thymoma 4 .…”

Section: Discussionmentioning

confidence: 99%

Myasthenia Gravis Associated with Thymoma and Aplastic Anemia: Case Report

Grbelja¹,

Vrhovać

Ulamec³

2017

ACC

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SUMMARY -Myasthenia gravis is associated in 10 to 15 percent of patients with thymic tumors, rarely with aplastic anemia. We report a 45-year-old male diagnosed with myasthenia gravis associated with thymoma. We started treatment with pyridostigmine. After thymectomy, the patient received 30 irradiation sessions. In the postoperative course, he had mild worsening of myasthenia gravis, which improved with prednisone. Five months later, he developed severe aplastic anemia. He was dependent on blood supplement. After allogeneic transplantation of bone marrow, he improved but later he developed graft versus host disease. Myasthenia gravis was under good control with 480 mg of pyridostigmine per day.

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“…These cytopenias include agranulocytosis, pure red cell aplasia (PRCA), and aplastic anemia [7][8][9][10][11][12]. PRCA is characterized by normochromic, normocytic anemia, reticulocytopenia, and the absence of erythroblasts from an otherwise normal bone marrow [13].…”

Section: Introductionmentioning

confidence: 99%

Macrocytic anemia and thrombocytosis associated with thymoma: A case report

et al. 2000

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Thymomas are often associated with autoimmune disorders. We report on a 45-year-old female patient with thymoma and hypogammaglobulinemia (Good's syndrome) who developed symptomatic macrocytic anemia (Hb 4.4 g/dl, MCV 112 fl) and thrombocytosis (Plt 442 G/l). Besides hypogammaglobulinemia (IgG 589 mg/dl), an inverted ratio of CD4 + / CD8 + cells was seen. The bone marrow biopsy showed a slightly hypercellular bone marrow with normal granulopoiesis, normal megakaryopoiesis and a mild dyserythropoiesis without any ring-sideroblasts. The in-vitro stem cell culture from the bone marrow revealed an atypical growth of macroclusters, reduced BFU-E and CFU-GEMM colony growth, whereas the CFU-GM colony growth was within the normal range. The chromosomal analysis showed a normal karyotype. The plasma vitamin B 12 and folate levels were within normal ranges, and we could not detect any autoantibodies. These findings excluded the differential diagnoses pure red cell aplasia (PRCA) and pernicious anemia. After resection of the thymoma of mixed cell type, the macrocytic anemia and thrombocytosis disappeared. The clinical course was complicated by a cerebral palsy and a life-threatening fungal septicemia after surgery. In the third year after thymectomy, hyporegenerative macrocytic anemia and thrombocytosis reappeared and an immunosuppressive treatment with prednisolone (1 mg/kg BW) was started. After initiation of the prednisolone therapy, reticulocyte counts increased and macrocytic anemia as well as thrombocytosis disappeared. The normalization of these laboratory parameters during glucocorticoid therapy suggests that in rare cases the constellation of macrocytic anemia, thrombocytosis and hypogammaglobulinemia may be due to an underlying immunologic mechanism. Am.

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Aplastic Anemia and Idiopathic Thrombocytopenic Purpura with Antibody to Platelet Glycoprotein llb/llla Following Resection of Malignant Thymoma

Cited by 20 publications

References 10 publications

Thymoma-associated pancytopenia: effectiveness of cyclosporine A

Thymoma-associated pancytopenia: effectiveness of cyclosporine A

Myasthenia Gravis Associated with Thymoma and Aplastic Anemia: Case Report

Macrocytic anemia and thrombocytosis associated with thymoma: A case report

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