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LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE
Central nervous system (CNS) manifestations are rare complications of relapsing polychondritis (RP). The majority of patients respond well to glucocorticoid therapy, but need to maintain it. Some patients are refractory to initial glucocorticoid therapy and to additional immunosuppressants, and end...
Autores principales: | , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nagoya University
2014
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4345688/ https://www.ncbi.nlm.nih.gov/pubmed/25741046 |
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author | KONDO, TAKESHI FUKUTA, MAMIKO TAKEMOTO, AYUMU TAKAMI, YUICHIRO SATO, MOTOKI TAKAHASHI, NORIYUKI SUZUKI, TOMIO SATO, JUICHI ATSUTA, NAOKI SOBUE, GEN TAKAHASHI, YUKITOSHI BAN, NOBUTARO |
author_facet | KONDO, TAKESHI FUKUTA, MAMIKO TAKEMOTO, AYUMU TAKAMI, YUICHIRO SATO, MOTOKI TAKAHASHI, NORIYUKI SUZUKI, TOMIO SATO, JUICHI ATSUTA, NAOKI SOBUE, GEN TAKAHASHI, YUKITOSHI BAN, NOBUTARO |
author_sort | KONDO, TAKESHI |
collection | PubMed |
description | Central nervous system (CNS) manifestations are rare complications of relapsing polychondritis (RP). The majority of patients respond well to glucocorticoid therapy, but need to maintain it. Some patients are refractory to initial glucocorticoid therapy and to additional immunosuppressants, and end up with an outcome worse than at therapy initiation. The standardized therapeutic protocol for this condition has not been established. The effects of anti-tumor necrosis factor (TNF) -α agents have been reported recently. We experienced a patient with RP and limbic encephalitis who was refractory to initial high-dose glucocorticoid, but subsequently responded to infliximab and did not show deterioration of signs and symptoms after stopping therapy. We report this case together with a systematic literature review. This is the first case report of RP with CNS manifestations successfully treated by an anti-TNF-α agent without recurrence after discontinuation. |
format | Online Article Text |
id | pubmed-4345688 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2014 |
publisher | Nagoya University |
record_format | MEDLINE/PubMed |
spelling | pubmed-43456882015-03-04 LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE KONDO, TAKESHI FUKUTA, MAMIKO TAKEMOTO, AYUMU TAKAMI, YUICHIRO SATO, MOTOKI TAKAHASHI, NORIYUKI SUZUKI, TOMIO SATO, JUICHI ATSUTA, NAOKI SOBUE, GEN TAKAHASHI, YUKITOSHI BAN, NOBUTARO Nagoya J Med Sci Case Report Central nervous system (CNS) manifestations are rare complications of relapsing polychondritis (RP). The majority of patients respond well to glucocorticoid therapy, but need to maintain it. Some patients are refractory to initial glucocorticoid therapy and to additional immunosuppressants, and end up with an outcome worse than at therapy initiation. The standardized therapeutic protocol for this condition has not been established. The effects of anti-tumor necrosis factor (TNF) -α agents have been reported recently. We experienced a patient with RP and limbic encephalitis who was refractory to initial high-dose glucocorticoid, but subsequently responded to infliximab and did not show deterioration of signs and symptoms after stopping therapy. We report this case together with a systematic literature review. This is the first case report of RP with CNS manifestations successfully treated by an anti-TNF-α agent without recurrence after discontinuation. Nagoya University 2014-08 /pmc/articles/PMC4345688/ /pubmed/25741046 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 KONDO, TAKESHI FUKUTA, MAMIKO TAKEMOTO, AYUMU TAKAMI, YUICHIRO SATO, MOTOKI TAKAHASHI, NORIYUKI SUZUKI, TOMIO SATO, JUICHI ATSUTA, NAOKI SOBUE, GEN TAKAHASHI, YUKITOSHI BAN, NOBUTARO LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title | LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title_full | LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title_fullStr | LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title_full_unstemmed | LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title_short | LIMBIC ENCEPHALITIS ASSOCIATED WITH RELAPSING POLYCHONDRITIS RESPONDED TO INFLIXIMAB AND MAINTAINED ITS CONDITION WITHOUT RECURRENCE AFTER DISCONTINUATION: A CASE REPORT AND REVIEW OF THE LITERATURE |
title_sort | limbic encephalitis associated with relapsing polychondritis responded to infliximab and maintained its condition without recurrence after discontinuation: a case report and review of the literature |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4345688/ https://www.ncbi.nlm.nih.gov/pubmed/25741046 |
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