Malignant thymoma complicated by amegakaryocytic thrombocytopenic purpura

Maslovsky, Igor; Gefel, Dov; Uriev, L.; Dor, David Ben; Lugassy, Gilles

doi:10.1016/j.ejim.2005.09.005

Cited by 18 publications

(27 citation statements)

References 6 publications

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“…Immunophenotyping of peripheral blood T-cells expressing T-cell receptor γß discriminated celiac disease from autoimmune enteropathy, which commonly expresses T-cell receptor αβ 18. Megakaryocytic hypoplasia has been reported after resection of a thymoma in previous studies,2,19 and the association of thymoma with pure red cell aplasia is well known 20. Amegakaryocytic thrombocytopenia following thymoma may represent an early presentation of impending aplastic anemia, with the time interval between thymoma resection and development of aplastic anemia between 3 and 48 months 2,21.…”

Section: Discussionmentioning

confidence: 93%

“…Megakaryocytic hypoplasia has been reported after resection of a thymoma in previous studies,2,19 and the association of thymoma with pure red cell aplasia is well known 20. Amegakaryocytic thrombocytopenia following thymoma may represent an early presentation of impending aplastic anemia, with the time interval between thymoma resection and development of aplastic anemia between 3 and 48 months 2,21. Aplastic anemia may be related with active thymoma or with thymoma in remission 22.…”

Section: Discussionmentioning

confidence: 96%

“…Thymoma is the most common tumor in the anterior mediastinum1 and originates from thymus epithelial cells 2. It seems that paraneoplastic syndromes associated with thymoma have an underling autoimmune characterization 3.…”

Section: Introductionmentioning

confidence: 99%

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Two thymus-related autoimmune disorders: a case report and review of the literature

Mirtavoos-Mahyari¹,

Khosravi²,

Esfahani-Monfared³

et al. 2014

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Thymoma is the most common tumor in the anterior mediastinum. A 56-year-old man presented unremitting and periodic chronic diarrhea of 9 weeks duration, and clinical examination revealed a huge nonhomogeneous mass lesion in the right lung and leukocytosis. He was treated with CHOP regimen (cyclophosphamide 1,200 mg/m2, doxorubicin 50 mg/m2, vincristine 1.5 mg/m2, and prednisolone 75 mg/m2 × 5 days) based on lung mass computed tomography-guided biopsy, but he was reevaluated because neither symptom improved. Surprisingly, celiac disease was documented with increased titer of immunoglobulin antibodies to gliadin and tissue transglutaminase. Lung mass rebiopsy and thymectomy demonstrated thymoma. After surgery, the patient showed aplastic anemia that responded well to cyclosporine. At 2-year follow-up, the patient’s hematologic status and diarrhea were completely recovered and no symptom and/or sign of thymoma recurrence was seen.

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Section: Discussionmentioning

confidence: 93%

Section: Discussionmentioning

confidence: 96%

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Two thymus-related autoimmune disorders: a case report and review of the literature

Mirtavoos-Mahyari¹,

Khosravi²,

Esfahani-Monfared³

et al. 2014

OTT

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“…Limited case reports on the coexistence of thymoma, PRCA, and AAMT, a seen in our case, have been published. 3,6-8 These cases are summarized in Table 1. Unlike in our case, however, in which all conditions occurred simultaneously, in each of these previous cases, the diseases occurred separately, indicating that thymoma-related hematologic disorders can change over time.…”

Section: Discussionmentioning

confidence: 99%

Invasive Thymoma with Pure Red Cell Aplasia and Amegakaryocytic Thrombocytopenia

et al. 2016

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We here describe a case involving a 67-yearold female patient who was referred to our hospital due to severe anemia (hemoglobin, 5.0 g/dL), thrombocytopenia (platelet count, 0.6 × 104/μL), and a mediastinal shadow with calcification noted on X-ray. On admission, an anterior mediastinal tumor was detected, and bone marrow biopsy revealed few megakaryocytes and severely reduced numbers of erythroid cells. The diagnosis was thymoma with pure red cell aplasia (PRCA) and acquired amegakaryocytic thrombocytopenia (AAMT). On Day 8 of admission, the patient received immunosuppressive therapy together with cyclosporine for the 2 severe hematologic diseases, which were stabilized within 2 months. Subsequently, total thymectomy was performed. The diagnosis of the tumor invading the left lung was invasive thymoma, Masaokakoga stage III. The histological diagnosis was World Health Organization type AB. Thymoma accompanied with PRCA and AAMT is very rare, and, based on our case, immunotherapeutic therapy for the hematologic disorders should precede surgical intervention.

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“…APMA is characterized by severe thrombocytopenia resulting from marked decrease or absence of megakaryocytes in the marrow in the presence of otherwise normal erythropoiesis and granulopoiesis. APMA can be either idiopathic or caused by a variety of conditions, such as acquired clonal cytogenetic abnormalities, drug sensitivity, toxin exposure, infectious diseases such as viral infection [1,2], immune diseases such as lupus erythematosus [3], systemic sclerosis [4], eosinophilic fasciitis [5], and malignancy [6,7]. Patients with acquired amegakaryocytic thrombocytopenia may have additional hematological abnormalities such as macrocytosis or dyserythropoiesis, abnormalities which may indicate potential future progression to aplastic anemia or myelodysplasia [8][9][10].…”

Section: Introductionmentioning

confidence: 99%

Acquired pure megakaryocytic aplasia successfully treated with cyclosporine

Omri¹,

Ibrahim²,

Taha³

et al. 2010

Turk J Hematol

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Acquired pure megakaryocytic aplasia is a rare hematological disorder characterized by thrombocytopenia with absent or markedly reduced megakaryocytes in the bone marrow. We report a case of a 25-year-old male diagnosed as acquired pure megakaryocytic aplasia. Treatment with prednisone and intravenous immunoglobulin failed, but he was successfully treated with cyclosporine, with complete remission after 90 days and normal platelet count maintained thereafter. (Turk J Hematol 2010; 27: 289-93) Key words: Acquired amegakaryocytic thrombocytopenia, steroids, cyclosporine

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Malignant thymoma complicated by amegakaryocytic thrombocytopenic purpura

Cited by 18 publications

References 6 publications

Two thymus-related autoimmune disorders: a case report and review of the literature

Two thymus-related autoimmune disorders: a case report and review of the literature

Invasive Thymoma with Pure Red Cell Aplasia and Amegakaryocytic Thrombocytopenia

Acquired pure megakaryocytic aplasia successfully treated with cyclosporine

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