Acquired Amegakaryocytic Thrombocytopenia with Glycoprotein IIb/IIIa Antibody in a Patient with Mantle Cell Lymphoma
Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. We encountered a diagnostically c...
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| Published in | Internal Medicine Vol. 64; no. 8; pp. 1229 - 1234 |
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| Language | English |
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The Japanese Society of Internal Medicine
15.04.2025
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| Abstract | Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. We encountered a diagnostically challenging case involving a 66-year-old man who showed severe thrombocytopenic bleeding with isolated megakaryocytic hypoplasia, elevated serum thrombopoietin levels, glycoprotein IIb/IIIa antibody positivity, and prolonged platelet transfusion refractoriness following mantle cell lymphoma (MCL). Treatment with corticosteroids and intravenous immunoglobulin was ineffective, while a combination of multiagent chemotherapy, including rituximab, was beneficial for both thrombocytopenia and MCL. Ultimately, the patient was diagnosed with AAMT and immune thrombocytopenia (ITP)-like platelet destruction. This case suggests that AAMT and ITP are non-exclusive and sometimes overlap as components of a broad spectrum of platelet-related autoimmune diseases. |
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| AbstractList | Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. We encountered a diagnostically challenging case involving a 66-year-old man who showed severe thrombocytopenic bleeding with isolated megakaryocytic hypoplasia, elevated serum thrombopoietin levels, glycoprotein IIb/IIIa antibody positivity, and prolonged platelet transfusion refractoriness following mantle cell lymphoma (MCL). Treatment with corticosteroids and intravenous immunoglobulin was ineffective, while a combination of multiagent chemotherapy, including rituximab, was beneficial for both thrombocytopenia and MCL. Ultimately, the patient was diagnosed with AAMT and immune thrombocytopenia (ITP)-like platelet destruction. This case suggests that AAMT and ITP are non-exclusive and sometimes overlap as components of a broad spectrum of platelet-related autoimmune diseases. Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. We encountered a diagnostically challenging case involving a 66-year-old man who showed severe thrombocytopenic bleeding with isolated megakaryocytic hypoplasia, elevated serum thrombopoietin levels, glycoprotein IIb/IIIa antibody positivity, and prolonged platelet transfusion refractoriness following mantle cell lymphoma (MCL). Treatment with corticosteroids and intravenous immunoglobulin was ineffective, while a combination of multiagent chemotherapy, including rituximab, was beneficial for both thrombocytopenia and MCL. Ultimately, the patient was diagnosed with AAMT and immune thrombocytopenia (ITP)-like platelet destruction. This case suggests that AAMT and ITP are non-exclusive and sometimes overlap as components of a broad spectrum of platelet-related autoimmune diseases. Background Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. Case description We encountered a diagnostically challenging case involving a 66-year-old man who showed severe thrombocytopenic bleeding with isolated megakaryocytic hypoplasia, elevated serum thrombopoietin levels, glycoprotein IIb/IIIa antibody positivity, and prolonged platelet transfusion refractoriness following mantle cell lymphoma (MCL). Treatment with corticosteroids and intravenous immunoglobulin was ineffective, while a combination of multiagent chemotherapy, including rituximab, was beneficial for both thrombocytopenia and MCL. Ultimately, the patient was diagnosed with AMMT and immune thrombocytopenia (ITP)-like platelet destruction. Discussion This case suggests that AAMT and ITP are non-exclusive and sometimes overlap as components of a broad spectrum of platelet-related autoimmune diseases.Background Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved other-lineage hematopoiesis in the bone marrow. The etiology of AAMT remains poorly understood owing to its rarity. Case description We encountered a diagnostically challenging case involving a 66-year-old man who showed severe thrombocytopenic bleeding with isolated megakaryocytic hypoplasia, elevated serum thrombopoietin levels, glycoprotein IIb/IIIa antibody positivity, and prolonged platelet transfusion refractoriness following mantle cell lymphoma (MCL). Treatment with corticosteroids and intravenous immunoglobulin was ineffective, while a combination of multiagent chemotherapy, including rituximab, was beneficial for both thrombocytopenia and MCL. Ultimately, the patient was diagnosed with AMMT and immune thrombocytopenia (ITP)-like platelet destruction. Discussion This case suggests that AAMT and ITP are non-exclusive and sometimes overlap as components of a broad spectrum of platelet-related autoimmune diseases. |
| ArticleNumber | 4016-24 |
| Author | Ueno, Mayumi Ueki, Toshimitsu Ichikawa, Naoaki Kobayashi, Hikaru Sato, Keijiro Sumi, Masahiko Tokutake, Takayoshi |
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| Cites_doi | 10.1002/ajh.20510 10.1016/j.phoj.2021.09.001 10.1007/s00296-006-0202-8 10.1182/hematology.2020000137 10.1007/s002770050199 10.2491/jjsth.29.281 10.1111/j.1751-553X.2011.01387.x 10.1159/000484595 10.2169/internalmedicine.2929-19 10.1002/art.10420 10.1016/j.hoc.2009.09.001 10.2169/internalmedicine.5611-20 10.2492/inflammregen.29.40 10.3324/haematol.11934 10.1159/000499523 10.1182/blood-2009-01-129155 10.7759/cureus.27315 10.1093/rheumatology/kel010 10.3748/wjg.v23.i35.6540 |
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| Keywords | amegakaryocytic thrombocytopenia anti-glycoprotein IIb/IIIa antibody malignant lymphoma immune thrombocytopenia |
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| References | 11. Cohn CS. Platelet transfusion refractoriness: how do I diagnose and manage? Hematology Am Soc Hematol Educ Program 2020: 527-532, 2020. 19. Meraj F, Hanif HM, Khan MR, Mansoor N. Transient acquired amegakaryocytic thrombocytopenia associated with pediatric Hodgkin lymphoma: a case report. Pediatr Hematol Oncol 6: 203-206, 2021. 6. Kuwana M, Kaburaki J, Okazaki Y, Miyazaki H, Ikeda Y. Two types of autoantibody-mediated thrombocytopenia in patients with systemic lupus erythematosus. Rheumatology 45: 851-854, 2006. 17. Hauswirth AW, Skrabs C, Schutzinger C, et al. Autoimmune thrombocytopenia in non-Hodgkin's lymphomas. Haematologica 93: 447-450, 2008. 18. Kurihara Y, Taoka K, Takagi E, Toyama K, Nakazaki K, Kurokawa M. Treatment of secondary immune thrombocytopenia with non-Hodgkin lymphoma: a case report and literature review. Intern Med 60: 1583-1588, 2021. 4. Agarwal N, Spahr JE, Werner TL, Newton DL, Rodgers GM. Acquired amegakaryocytic thrombocytopenic purpura. Am J Hematol 81: 132-135, 2006. 1. Kuwana M. Acquired amegakaryocytic thrombocytopenia. Hematology 77: 66-70, 2018 (in Japanese). 7. Levy I, Laor R, Jiries N, Bejar J, Polliack A, Tadmor T. Amegakaryocytic thrombocytopenia and subsequent aplastic anemia associated with apparent Epstein-Barr virus infection. Acta Haemato 139: 7-11, 2018. 15. Yamanouchi J. Immune thrombocytopenia. Nihon Kessen Shiketsu Gakkaishi (Jpn J Thromb Hemost) 29: 281-287, 2018 (in Japanese). 20. Lugassy G. Non-Hodgkin's lymphoma presenting with amegakaryocytic thrombocytopenic purpura. Ann Hematol 73: 41-42, 1996. 8. Kuwana M, Okazaki Y, Kajihara M, et al. Autoantibody to c-Mpl (thrombopoietin receptor) in systemic lupus erythematosus: relationship to thrombocytopenia with megakaryocytic hypoplasia. Arthritis Rheum 46: 2148-2159, 2002. 9. Kuter DJ, Gernsheimer TB. Thrombopoietin and platelet production in chronic immune thrombocytopenia. Hematol Oncol Clin North Am 23: 1193-1211, 2009. 12. Ichimata S, Kobayashi M, Honda K, Shibata S, Matsumoto A, Kanno H. Acquired amegakaryocytic thrombocytopenia previously diagnosed as idiopathic thrombocytopenic purpura in a patient with hepatitis C virus infection. World J Gastroenterol 23: 6540-6545, 2017. 2. Tirthani E, Said MS, De Jesus O. Amegakaryocytic Thrombocytopenia. In: StatPearls. StatPearls Publishing, Treasure Island (FL), 2023. 16. Cines DB, Bussel JB, Liebman HA, Luning Prak ET. The ITP syndrome: pathogenic and clinical diversity. Blood 113: 6511-6521, 2009. 10. Kuwana M. Autoantibodies to platelets: roles in thrombocytopenia. Inflamm Regen 29: 40-46, 2009. 3. Son B, Park HS, Han HS, et al. A case of acquired amegakaryocytic thrombocytopenia with anti-c-mpl autoantibody: comparison with idiopathic thrombocytopenic purpura. Acta Haematol 142: 239-243, 2019. 21. Lewandowski K, Kowalik MM, Pawlaczyk R, Rogowski J, Hellmann A. Microscopic examination of bone marrow aspirate in healthy adults - comparison of two techniques of slide preparation. Int J Lab Hematol 34: 254-261, 2012. 5. Ikeda N, Hisano Y, Kamao T, Uno M, Mizushima T. Acquired amegakaryocytic thrombocytopenia associated with autoimmune hemolytic anemia. Cureus 14: e27315, 2022. 13. Her MY, Kim TH, Chang HK, Lee WS, Yoo DH. Successful treatment of acquired amegakaryocytic thrombocytopenia with cyclosporine in adult onset Still's disease. Rheumatol Int 27: 295-298, 2007. 14. Ichikawa T, Shimojima Y, Otuki T, Ueno K, Kishida D, Sekijima Y. Acquired amegakaryocytic thrombocytopenia in adult-onset Still's disease: successful combination therapy with tocilizumab and cyclosporine. Intern Med 58: 3473-3478, 2019. 11 12 13 14 15 16 17 18 19 1 2 3 4 5 6 7 8 9 20 10 21 |
| References_xml | – reference: 17. Hauswirth AW, Skrabs C, Schutzinger C, et al. Autoimmune thrombocytopenia in non-Hodgkin's lymphomas. Haematologica 93: 447-450, 2008. – reference: 19. Meraj F, Hanif HM, Khan MR, Mansoor N. Transient acquired amegakaryocytic thrombocytopenia associated with pediatric Hodgkin lymphoma: a case report. Pediatr Hematol Oncol 6: 203-206, 2021. – reference: 11. Cohn CS. Platelet transfusion refractoriness: how do I diagnose and manage? Hematology Am Soc Hematol Educ Program 2020: 527-532, 2020. – reference: 6. Kuwana M, Kaburaki J, Okazaki Y, Miyazaki H, Ikeda Y. Two types of autoantibody-mediated thrombocytopenia in patients with systemic lupus erythematosus. Rheumatology 45: 851-854, 2006. – reference: 10. Kuwana M. Autoantibodies to platelets: roles in thrombocytopenia. Inflamm Regen 29: 40-46, 2009. – reference: 12. Ichimata S, Kobayashi M, Honda K, Shibata S, Matsumoto A, Kanno H. Acquired amegakaryocytic thrombocytopenia previously diagnosed as idiopathic thrombocytopenic purpura in a patient with hepatitis C virus infection. World J Gastroenterol 23: 6540-6545, 2017. – reference: 4. Agarwal N, Spahr JE, Werner TL, Newton DL, Rodgers GM. Acquired amegakaryocytic thrombocytopenic purpura. Am J Hematol 81: 132-135, 2006. – reference: 9. Kuter DJ, Gernsheimer TB. Thrombopoietin and platelet production in chronic immune thrombocytopenia. Hematol Oncol Clin North Am 23: 1193-1211, 2009. – reference: 16. Cines DB, Bussel JB, Liebman HA, Luning Prak ET. The ITP syndrome: pathogenic and clinical diversity. Blood 113: 6511-6521, 2009. – reference: 7. Levy I, Laor R, Jiries N, Bejar J, Polliack A, Tadmor T. Amegakaryocytic thrombocytopenia and subsequent aplastic anemia associated with apparent Epstein-Barr virus infection. Acta Haemato 139: 7-11, 2018. – reference: 13. Her MY, Kim TH, Chang HK, Lee WS, Yoo DH. Successful treatment of acquired amegakaryocytic thrombocytopenia with cyclosporine in adult onset Still's disease. Rheumatol Int 27: 295-298, 2007. – reference: 1. Kuwana M. Acquired amegakaryocytic thrombocytopenia. Hematology 77: 66-70, 2018 (in Japanese). – reference: 8. Kuwana M, Okazaki Y, Kajihara M, et al. Autoantibody to c-Mpl (thrombopoietin receptor) in systemic lupus erythematosus: relationship to thrombocytopenia with megakaryocytic hypoplasia. Arthritis Rheum 46: 2148-2159, 2002. – reference: 18. Kurihara Y, Taoka K, Takagi E, Toyama K, Nakazaki K, Kurokawa M. Treatment of secondary immune thrombocytopenia with non-Hodgkin lymphoma: a case report and literature review. Intern Med 60: 1583-1588, 2021. – reference: 20. Lugassy G. Non-Hodgkin's lymphoma presenting with amegakaryocytic thrombocytopenic purpura. Ann Hematol 73: 41-42, 1996. – reference: 21. Lewandowski K, Kowalik MM, Pawlaczyk R, Rogowski J, Hellmann A. Microscopic examination of bone marrow aspirate in healthy adults - comparison of two techniques of slide preparation. Int J Lab Hematol 34: 254-261, 2012. – reference: 2. Tirthani E, Said MS, De Jesus O. Amegakaryocytic Thrombocytopenia. In: StatPearls. StatPearls Publishing, Treasure Island (FL), 2023. – reference: 5. Ikeda N, Hisano Y, Kamao T, Uno M, Mizushima T. Acquired amegakaryocytic thrombocytopenia associated with autoimmune hemolytic anemia. Cureus 14: e27315, 2022. – reference: 14. Ichikawa T, Shimojima Y, Otuki T, Ueno K, Kishida D, Sekijima Y. Acquired amegakaryocytic thrombocytopenia in adult-onset Still's disease: successful combination therapy with tocilizumab and cyclosporine. Intern Med 58: 3473-3478, 2019. – reference: 15. Yamanouchi J. Immune thrombocytopenia. Nihon Kessen Shiketsu Gakkaishi (Jpn J Thromb Hemost) 29: 281-287, 2018 (in Japanese). – reference: 3. Son B, Park HS, Han HS, et al. A case of acquired amegakaryocytic thrombocytopenia with anti-c-mpl autoantibody: comparison with idiopathic thrombocytopenic purpura. Acta Haematol 142: 239-243, 2019. – ident: 2 – ident: 4 doi: 10.1002/ajh.20510 – ident: 19 doi: 10.1016/j.phoj.2021.09.001 – ident: 13 doi: 10.1007/s00296-006-0202-8 – ident: 11 doi: 10.1182/hematology.2020000137 – ident: 20 doi: 10.1007/s002770050199 – ident: 15 doi: 10.2491/jjsth.29.281 – ident: 21 doi: 10.1111/j.1751-553X.2011.01387.x – ident: 7 doi: 10.1159/000484595 – ident: 14 doi: 10.2169/internalmedicine.2929-19 – ident: 1 – ident: 8 doi: 10.1002/art.10420 – ident: 9 doi: 10.1016/j.hoc.2009.09.001 – ident: 18 doi: 10.2169/internalmedicine.5611-20 – ident: 10 doi: 10.2492/inflammregen.29.40 – ident: 17 doi: 10.3324/haematol.11934 – ident: 3 doi: 10.1159/000499523 – ident: 16 doi: 10.1182/blood-2009-01-129155 – ident: 5 doi: 10.7759/cureus.27315 – ident: 6 doi: 10.1093/rheumatology/kel010 – ident: 12 doi: 10.3748/wjg.v23.i35.6540 |
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| Snippet | Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and preserved... Background Acquired amegakaryocytic thrombocytopenia (AAMT) is a rare disorder characterized by thrombocytopenia, marked megakaryocytic hypoplasia, and... |
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| SubjectTerms | Aged amegakaryocytic thrombocytopenia anti-glycoprotein IIb/IIIa antibody Autoantibodies - blood Autoimmune diseases Bone Marrow Diseases Case Report Chemotherapy Corticosteroids Hemopoiesis Humans Hypoplasia Idiopathic thrombocytopenic purpura immune thrombocytopenia Lymphoma, Mantle-Cell - complications Lymphoma, Mantle-Cell - diagnosis Lymphoma, Mantle-Cell - drug therapy Lymphoma, Mantle-Cell - immunology Male malignant lymphoma Mantle cell lymphoma Platelet Glycoprotein GPIIb-IIIa Complex - immunology Platelets Purpura, Thrombocytopenic Purpura, Thrombocytopenic, Idiopathic - diagnosis Rituximab Thrombocytopenia - blood Thrombocytopenia - complications Thrombocytopenia - diagnosis Thrombocytopenia - immunology Thrombopoietin |
| Title | Acquired Amegakaryocytic Thrombocytopenia with Glycoprotein IIb/IIIa Antibody in a Patient with Mantle Cell Lymphoma |
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