Thrombocytopenia and Anemia with Anti-c-Mpl antibodies Effectively Treated with Cyclosporine in a Patient with Rheumatoid Arthritis and Chronic Renal Failure

Hashimoto, Akinori; Kanisawa, Yuji; Fujimi, Akihito; Nakajima, Chisa; Hayasaka, Naotaka; Yamada, Shota; Okuda, Toshinori; Minami, Shinya; Yamauchi, Naofumi; Iwasaki, Shigeo; Suzuki, Akira; Kato, Junji

doi:10.2169/internalmedicine.55.5190

Cited by 8 publications

(7 citation statements)

References 13 publications

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“…AATP is a rare disease, and only 64 cases have been reported in the past 2 decades 1 , 5 , 6 , 12 - 16 , 21 - 23 . In the present case, bone marrow examination should have performed when we made a tentative diagnosis of ITP because thrombocytopenic conditions include ITP, AATP, and other thrombocytopenic disorders.…”

Section: Discussionmentioning

confidence: 72%

“…An autoimmune mechanism has been considered as the cause of AATP such as an antibody directed against TPO 1 or c-Mpl, 21 , 22 , 24 or cytotoxic immune cells against megakaryocyte precursors or progenitors 23 , 25 , 26 . In support of an autoimmune mechanism, associations with other autoimmune diseases, such as systemic lupus erythematosus, 21 , 24 , 25 rheumatoid arthritis, 12 Sjögren’s syndrome, 18 and adult-onset Still’s disease, 7 have been reported. As another cause, the involvement of estrogen has been suggested 5 .…”

Section: Discussionmentioning

confidence: 99%

“…As therapeutic strategies for AATP, glucocorticoid, 1 , 3 , 4 , 7 , 8 , 10 - 12 , 22 , 23 , 25 , 26 high-dose IVIg, 1 , 7 , 8 , 22 , 23 anti-thymocyte globulin, 4 , 9 cyclosporine A, 7 , 9 - 12 , 23 rituximab, 10 , 11 splenectomy, 8 allogeneic hematopoietic stem cell transplantation, 8 and agonists for c-Mpl, 10 androgens, 5 and vincristine 7 , 8 have been employed. Of these, glucocorticoid therapy, which is widely used and effective for ITP, is less effective for AATP.…”

Section: Discussionmentioning

confidence: 99%

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Marked rebound thrombocytosis in response to glucocorticoids in a patient with acquired amegakaryocytic thrombocytopenia

Nishino¹,

Kodaka²,

Sawada³

et al. 2018

JCEH

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Acquired amegakaryocytic thrombocytopenia (AATP) is a rare disease characterized by thrombocytopenia and the disappearance of marrow megakaryocytes. A 43-year-old man was admitted because of thrombocytopenia of 1.0×10 9 /L. Bone marrow aspirate demonstrated normal hematopoiesis lacking megakaryocytes, and AATP was diagnosed. The serum concentration of thrombopoietin (TPO) was high (7.72 fmol/mL). Prednisolone (PSL) at 60 mg/day was started and the platelet count recovered to 1,335×10 9 /L; however, excessive megakaryocytopoiesis and subsequent decline in platelet count were noted 14 days later. At the peak platelet count, the TPO remained at 3.79 fmol/mL and returned to a normal level of 0.40 fmol/mL during the period of normal platelet count after PSL tapering. The marked thrombocytosis in response to prednisolone may have been caused by the high TPO after the resolution of suppressed megakaryopoiesis. Marked rebound thrombocytosis beyond 1,000×10 9 /L after successful PSL treatment for AATP has not been previously reported.

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Section: Discussionmentioning

confidence: 72%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Marked rebound thrombocytosis in response to glucocorticoids in a patient with acquired amegakaryocytic thrombocytopenia

Nishino¹,

Kodaka²,

Sawada³

et al. 2018

JCEH

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“…CsA can inhibit T lymphocytes through the following two pathways: i) CsA inhibits the generation of interleukin (IL)-2 ( 15 ); ii) CsA inhibits generation of the IL-2 receptor ( 16 ). CsA has been widely used in the treatment of various diseases, including aplastic anemia ( 17 ), nephritic syndrome ( 18 ), rheumatoid arthritis ( 19 ) and psoriasis ( 20 ). Studies have shown that CsA inhibits cyclophilin-D (Cyp-D), and then prevents the formation of mPTP ( 21 ).…”

Section: Introductionmentioning

confidence: 99%

Therapeutic efficacy of cyclosporin A against spinal cord injury in rats with hyperglycemia

Zhi-rong

Guo

et al. 2018

Mol Med Report

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The present study aimed to explore the therapeutic effects of cyclosporin A (CsA) on spinal cord injury (SCI) in rats with hyperglycemia and to identify a novel potential method to treat SCI in the presence of hyperglycemia. Female Sprague-Dawley (SD) rats were randomly allocated into four groups: Sham, SCI, SCI+hyperglycemia and SCI+hyperglycemia+CsA groups. Streptozotocin-induced hyperglycemic SD rats and a weight-drop contusion SCI model were established. The Basso, Beattie, Bresnahan scale and inclined plane test were used to evaluate the neurological function of the rats. Flow cytometric assay was performed to detect the apoptotic rates of cells in the spinal cord. ELISA and western blot analysis were performed to determine the levels of interleukin (IL)-10, tumor necrosis factor (TNF)-α, cyclophilin-D (Cyp-D) and apoptosis-inducing factor (AIF). The results demonstrated that CsA significantly improved the neurological function of the SCI rats with hyperglycemia. CsA markedly reduced the number of apoptotic cells exaggerated by hyperglycemia in the spinal cord of the SCI rats. CsA significantly decreased the expression levels of IL-10, TNF-α, Cyp-D and AIF in the spinal cord of the SCI rats. Overall, the present study revealed a significant role of CsA in the treatment of SCI in the presence of hyperglycemia by inhibiting the apoptosis of spinal cord cells.

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“…It can either occur isolated or be associated with systemic lupus erythematodus (SLE) (Cela et al, 2010;Ernestho-ghoud et al, 2015) and other autoimmune diseases (Hashimoto et al, 2016), as well as hematological malignancies such as non-Hodgkin lymphoma (Lugassy, 1996).…”

Section: Introductionmentioning

confidence: 99%

Acquired Amegakaryocytic Thrombocytopenic Purpura Progressing into Aplastic Anemia

et al. 2017

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Acquired amegakaryocytic thrombocytopenic purpura (AATP) is a rare hematological disorder characterized by severe thrombocytopenia and a complete or near-to complete absence of megakaryocytes in the bone marrow, while granulopoiesis, as well as erythropoiesis are usually preserved. Although autoimmune mechanisms are believed to be causative, the exact underlying pathogenesis is not known. To date, only few cases have been reported and management of this disease remains controversial with immunosuppression being the treatment modality of choice in the majority of patients. In this article, we report a case of newly acquired AATP without an associated autoimmune disease, refractory to corticoids, intravenous immunoglobulin (IVIG) and second-generation TPO (thrombopoietin) agonists, which have recently been approved for the treatment of thrombocytopenia. Finally, in accordance with other reports, disease progression into aplastic anemia has occurred.

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Thrombocytopenia and Anemia with Anti-c-Mpl antibodies Effectively Treated with Cyclosporine in a Patient with Rheumatoid Arthritis and Chronic Renal Failure

Cited by 8 publications

References 13 publications

Marked rebound thrombocytosis in response to glucocorticoids in a patient with acquired amegakaryocytic thrombocytopenia

Marked rebound thrombocytosis in response to glucocorticoids in a patient with acquired amegakaryocytic thrombocytopenia

Therapeutic efficacy of cyclosporin A against spinal cord injury in rats with hyperglycemia

Acquired Amegakaryocytic Thrombocytopenic Purpura Progressing into Aplastic Anemia

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