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Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls

Thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome is a rare condition with diverse clinical and pathological characteristics related to multi-organ damage. We report a case of TAFRO syndrome complicated by immune thrombocytopenia with prolonged fever and...

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Autores principales: Sato, Hironori, Kanno, Atsuhiro, Sato, Minato, Endo, Akari, Ito, Hiroki, Ohara, Takahiro, Shirota, Yuko, Sumitomo, Kazuhiro, Mori, Takefumi, Furukawa, Katsutoshi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10611515/
https://www.ncbi.nlm.nih.gov/pubmed/37901232
http://dx.doi.org/10.3389/fimmu.2023.1266187
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author Sato, Hironori
Kanno, Atsuhiro
Sato, Minato
Endo, Akari
Ito, Hiroki
Ohara, Takahiro
Shirota, Yuko
Sumitomo, Kazuhiro
Mori, Takefumi
Furukawa, Katsutoshi
author_facet Sato, Hironori
Kanno, Atsuhiro
Sato, Minato
Endo, Akari
Ito, Hiroki
Ohara, Takahiro
Shirota, Yuko
Sumitomo, Kazuhiro
Mori, Takefumi
Furukawa, Katsutoshi
author_sort Sato, Hironori
collection PubMed
description Thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome is a rare condition with diverse clinical and pathological characteristics related to multi-organ damage. We report a case of TAFRO syndrome complicated by immune thrombocytopenia with prolonged fever and thrombocytopenia for several weeks. A 61-year-old man was transferred with sepsis caused by Enterococcus faecalis, and developed disseminated intravascular coagulation. Antibiotics treatment was initiated: however, low-grade fever and thrombocytopenia persisted despite the adequate antimicrobial treatment. Systemic edema, pleural effusion, and ascites had developed before hospitalization, and renal and liver function had deteriorated, resulting in progressive multi-organ damage. Prednisolone 40 mg/day was initiated based on the assumption of a condition in which excessive production of inflammatory cytokines would lead to systemic deterioration and fatal organ damage. Subsequently, the fever resolved, and renal function began to normalize. However, thrombocytopenia did not show much recovery trend after Helicobacter pylori eradication therapy and initiation of thrombopoietin receptor agonists. Bone marrow biopsy results showed normal bone marrow with no malignant findings. Alternatively, significant clinical signs met the diagnostic criteria for TAFRO syndrome, and a renal biopsy revealed thrombotic microangiopathy, which is also reasonable for renal involvement in TAFRO syndrome. The use of cyclosporine remarkably corrected the thrombocytopenia. We considered this a case of TAFRO syndrome that developed after sepsis with disseminated intravascular coagulation and performed the differential diagnosis of prolonged thrombocytopenia and excluded it. Although TAFRO syndrome is a unique disease concept, diagnostic criteria may consist of nonspecific elements such as generalized edema, thrombocytopenia, persistent fever, and elevated inflammatory response, and there are many differential conditions to exclude, requiring caution in diagnosing TAFRO syndrome.
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spelling pubmed-106115152023-10-28 Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls Sato, Hironori Kanno, Atsuhiro Sato, Minato Endo, Akari Ito, Hiroki Ohara, Takahiro Shirota, Yuko Sumitomo, Kazuhiro Mori, Takefumi Furukawa, Katsutoshi Front Immunol Immunology Thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome is a rare condition with diverse clinical and pathological characteristics related to multi-organ damage. We report a case of TAFRO syndrome complicated by immune thrombocytopenia with prolonged fever and thrombocytopenia for several weeks. A 61-year-old man was transferred with sepsis caused by Enterococcus faecalis, and developed disseminated intravascular coagulation. Antibiotics treatment was initiated: however, low-grade fever and thrombocytopenia persisted despite the adequate antimicrobial treatment. Systemic edema, pleural effusion, and ascites had developed before hospitalization, and renal and liver function had deteriorated, resulting in progressive multi-organ damage. Prednisolone 40 mg/day was initiated based on the assumption of a condition in which excessive production of inflammatory cytokines would lead to systemic deterioration and fatal organ damage. Subsequently, the fever resolved, and renal function began to normalize. However, thrombocytopenia did not show much recovery trend after Helicobacter pylori eradication therapy and initiation of thrombopoietin receptor agonists. Bone marrow biopsy results showed normal bone marrow with no malignant findings. Alternatively, significant clinical signs met the diagnostic criteria for TAFRO syndrome, and a renal biopsy revealed thrombotic microangiopathy, which is also reasonable for renal involvement in TAFRO syndrome. The use of cyclosporine remarkably corrected the thrombocytopenia. We considered this a case of TAFRO syndrome that developed after sepsis with disseminated intravascular coagulation and performed the differential diagnosis of prolonged thrombocytopenia and excluded it. Although TAFRO syndrome is a unique disease concept, diagnostic criteria may consist of nonspecific elements such as generalized edema, thrombocytopenia, persistent fever, and elevated inflammatory response, and there are many differential conditions to exclude, requiring caution in diagnosing TAFRO syndrome. Frontiers Media S.A. 2023-10-13 /pmc/articles/PMC10611515/ /pubmed/37901232 http://dx.doi.org/10.3389/fimmu.2023.1266187 Text en Copyright © 2023 Sato, Kanno, Sato, Endo, Ito, Ohara, Shirota, Sumitomo, Mori and Furukawa https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Sato, Hironori
Kanno, Atsuhiro
Sato, Minato
Endo, Akari
Ito, Hiroki
Ohara, Takahiro
Shirota, Yuko
Sumitomo, Kazuhiro
Mori, Takefumi
Furukawa, Katsutoshi
Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title_full Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title_fullStr Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title_full_unstemmed Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title_short Case Report: A case of TAFRO syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
title_sort case report: a case of tafro syndrome with severe and prolonged thrombocytopenia: diagnostic pitfalls
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10611515/
https://www.ncbi.nlm.nih.gov/pubmed/37901232
http://dx.doi.org/10.3389/fimmu.2023.1266187
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