Cargando…

Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature

TAFRO syndrome is a novel disease concept characterized by Thrombocytopenia, Anasarca, myeloFibrosis, Renal dysfunction, Organomegaly, multiple lymphadenopathy and a histopathological pattern of atypical Castleman’s disease. A 58-year-old man was diagnosed as TAFRO syndrome by clinical and histopath...

Descripción completa

Detalles Bibliográficos
Autores principales: Matsuhisa, Takaharu, Takahashi, Noriyuki, Nakaguro, Masato, Sato, Motoki, Inoue, Eri, Teshigawara, Shiho, Ozawa, Yukihiro, Kondo, Takeshi, Nakamura, Shigeo, Sato, Juichi, Ban, Nobutaro
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nagoya University 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6728207/
https://www.ncbi.nlm.nih.gov/pubmed/31579342
http://dx.doi.org/10.18999/nagjms.81.3.519
_version_ 1783449393820598272
author Matsuhisa, Takaharu
Takahashi, Noriyuki
Nakaguro, Masato
Sato, Motoki
Inoue, Eri
Teshigawara, Shiho
Ozawa, Yukihiro
Kondo, Takeshi
Nakamura, Shigeo
Sato, Juichi
Ban, Nobutaro
author_facet Matsuhisa, Takaharu
Takahashi, Noriyuki
Nakaguro, Masato
Sato, Motoki
Inoue, Eri
Teshigawara, Shiho
Ozawa, Yukihiro
Kondo, Takeshi
Nakamura, Shigeo
Sato, Juichi
Ban, Nobutaro
author_sort Matsuhisa, Takaharu
collection PubMed
description TAFRO syndrome is a novel disease concept characterized by Thrombocytopenia, Anasarca, myeloFibrosis, Renal dysfunction, Organomegaly, multiple lymphadenopathy and a histopathological pattern of atypical Castleman’s disease. A 58-year-old man was diagnosed as TAFRO syndrome by clinical and histopathological findings. After receiving intensive immunosuppressive therapy, his thrombocytopenia and anasarca had not improved. He developed complications such as methicillin-resistant Staphylococcus aureus sepsis, gastrointestinal bleeding, peritonitis caused by Stenotrophomonas maltophilia, gastrointestinal perforation, and disseminated candidiasis resulting in death. Autopsy revealed disseminated candidiasis and hemophagocytic lymphohistiocytosis, with no evidence of TAFRO syndrome. During treatment, we regarded his lasting thrombocytopenia and anasarca as insufficient control of TAFRO syndrome. However, the autopsy revealed that thrombocytopenia was caused by secondary hemophagocytic lymphohistiocytosis caused by over-immunosuppression. We reviewed the published literature to identify indicators of adequate treatment, which suggested improvement of platelet count and anasarca several weeks after initial therapy. This indicated that we could not depend on the platelet count and anasarca in acute medical care after initial treatment. We should treat TAFRO syndrome based on patients’ clinical status and obviate the risk of treatment-related complications caused by over-immunosuppression.
format Online
Article
Text
id pubmed-6728207
institution National Center for Biotechnology Information
language English
publishDate 2019
publisher Nagoya University
record_format MEDLINE/PubMed
spelling pubmed-67282072019-10-02 Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature Matsuhisa, Takaharu Takahashi, Noriyuki Nakaguro, Masato Sato, Motoki Inoue, Eri Teshigawara, Shiho Ozawa, Yukihiro Kondo, Takeshi Nakamura, Shigeo Sato, Juichi Ban, Nobutaro Nagoya J Med Sci Case Report TAFRO syndrome is a novel disease concept characterized by Thrombocytopenia, Anasarca, myeloFibrosis, Renal dysfunction, Organomegaly, multiple lymphadenopathy and a histopathological pattern of atypical Castleman’s disease. A 58-year-old man was diagnosed as TAFRO syndrome by clinical and histopathological findings. After receiving intensive immunosuppressive therapy, his thrombocytopenia and anasarca had not improved. He developed complications such as methicillin-resistant Staphylococcus aureus sepsis, gastrointestinal bleeding, peritonitis caused by Stenotrophomonas maltophilia, gastrointestinal perforation, and disseminated candidiasis resulting in death. Autopsy revealed disseminated candidiasis and hemophagocytic lymphohistiocytosis, with no evidence of TAFRO syndrome. During treatment, we regarded his lasting thrombocytopenia and anasarca as insufficient control of TAFRO syndrome. However, the autopsy revealed that thrombocytopenia was caused by secondary hemophagocytic lymphohistiocytosis caused by over-immunosuppression. We reviewed the published literature to identify indicators of adequate treatment, which suggested improvement of platelet count and anasarca several weeks after initial therapy. This indicated that we could not depend on the platelet count and anasarca in acute medical care after initial treatment. We should treat TAFRO syndrome based on patients’ clinical status and obviate the risk of treatment-related complications caused by over-immunosuppression. Nagoya University 2019-08 /pmc/articles/PMC6728207/ /pubmed/31579342 http://dx.doi.org/10.18999/nagjms.81.3.519 Text en http://creativecommons.org/licenses/by-nc-nd/4.0/ This is an Open Access article distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. To view the details of this license, please visit (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Matsuhisa, Takaharu
Takahashi, Noriyuki
Nakaguro, Masato
Sato, Motoki
Inoue, Eri
Teshigawara, Shiho
Ozawa, Yukihiro
Kondo, Takeshi
Nakamura, Shigeo
Sato, Juichi
Ban, Nobutaro
Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title_full Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title_fullStr Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title_full_unstemmed Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title_short Fatal case of TAFRO syndrome associated with over-immunosuppression: a case report and review of the literature
title_sort fatal case of tafro syndrome associated with over-immunosuppression: a case report and review of the literature
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6728207/
https://www.ncbi.nlm.nih.gov/pubmed/31579342
http://dx.doi.org/10.18999/nagjms.81.3.519
work_keys_str_mv AT matsuhisatakaharu fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT takahashinoriyuki fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT nakaguromasato fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT satomotoki fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT inoueeri fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT teshigawarashiho fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT ozawayukihiro fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT kondotakeshi fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT nakamurashigeo fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT satojuichi fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature
AT bannobutaro fatalcaseoftafrosyndromeassociatedwithoverimmunosuppressionacasereportandreviewoftheliterature