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A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura

Leflunomide has not been previously associated with thrombotic thrombocytopenic purpura (TTP), a rare life-threatening clinical syndrome characterized by thrombotic microangiopathy (TMA) due to inability to cleave ADAMTS13. Here, we present the first case of leflunomide-induced TTP. Our patient deve...

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Autores principales: Shields, Misty Dawn, Skelton, William Paul, Laber, Damian A., Verbosky, Michael, Ashraf, Noman
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elmer Press 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8256916/
https://www.ncbi.nlm.nih.gov/pubmed/34267852
http://dx.doi.org/10.14740/jh837
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author Shields, Misty Dawn
Skelton, William Paul
Laber, Damian A.
Verbosky, Michael
Ashraf, Noman
author_facet Shields, Misty Dawn
Skelton, William Paul
Laber, Damian A.
Verbosky, Michael
Ashraf, Noman
author_sort Shields, Misty Dawn
collection PubMed
description Leflunomide has not been previously associated with thrombotic thrombocytopenic purpura (TTP), a rare life-threatening clinical syndrome characterized by thrombotic microangiopathy (TMA) due to inability to cleave ADAMTS13. Here, we present the first case of leflunomide-induced TTP. Our patient developed encephalopathy, thrombocytopenia, anemia and hyperbilirubinemia 2 months after starting leflunomide. Schistocytes were noted on peripheral smear and ADAMTS13 activity was low (< 5%), consistent with acquired TTP. He received therapeutic plasma exchange, corticosteroids, rituximab and caplacizumab with normalization of hemolysis labs and ADAMTS13 activity. However, pancytopenia persisted, raising the suspicion for leflunomide toxicity. Oral cholestyramine treatment was empirically started before teriflunomide (a leflunomide metabolite) level was found to be elevated. Blood counts normalized after cholestyramine and have remained normal at last follow-up over a year later. This is the first reported case of TTP precipitated by leflunomide. Our case highlights the importance of recognizing drugs as an etiology of TMA and adds leflunomide to this list.
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spelling pubmed-82569162021-07-14 A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura Shields, Misty Dawn Skelton, William Paul Laber, Damian A. Verbosky, Michael Ashraf, Noman J Hematol Case Report Leflunomide has not been previously associated with thrombotic thrombocytopenic purpura (TTP), a rare life-threatening clinical syndrome characterized by thrombotic microangiopathy (TMA) due to inability to cleave ADAMTS13. Here, we present the first case of leflunomide-induced TTP. Our patient developed encephalopathy, thrombocytopenia, anemia and hyperbilirubinemia 2 months after starting leflunomide. Schistocytes were noted on peripheral smear and ADAMTS13 activity was low (< 5%), consistent with acquired TTP. He received therapeutic plasma exchange, corticosteroids, rituximab and caplacizumab with normalization of hemolysis labs and ADAMTS13 activity. However, pancytopenia persisted, raising the suspicion for leflunomide toxicity. Oral cholestyramine treatment was empirically started before teriflunomide (a leflunomide metabolite) level was found to be elevated. Blood counts normalized after cholestyramine and have remained normal at last follow-up over a year later. This is the first reported case of TTP precipitated by leflunomide. Our case highlights the importance of recognizing drugs as an etiology of TMA and adds leflunomide to this list. Elmer Press 2021-06 2021-06-16 /pmc/articles/PMC8256916/ /pubmed/34267852 http://dx.doi.org/10.14740/jh837 Text en Copyright 2021, Shields et al. https://creativecommons.org/licenses/by-nc/4.0/This article is distributed under the terms of the Creative Commons Attribution Non-Commercial 4.0 International License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Shields, Misty Dawn
Skelton, William Paul
Laber, Damian A.
Verbosky, Michael
Ashraf, Noman
A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title_full A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title_fullStr A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title_full_unstemmed A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title_short A Novel Case of Leflunomide-Induced Thrombotic Thrombocytopenic Purpura
title_sort novel case of leflunomide-induced thrombotic thrombocytopenic purpura
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8256916/
https://www.ncbi.nlm.nih.gov/pubmed/34267852
http://dx.doi.org/10.14740/jh837
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