Cargando…

Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept

BACKGROUND: Several disorders may present with mononeuritis multiplex and the etiological diagnosis can be challenging. CASE PRESENTATION: We report a 42 year-old female who presented with severe lower limb neuropathic pain, asymmetric weakness and sensory impairment and was diagnosed with mononeuri...

Descripción completa

Detalles Bibliográficos
Autores principales: Marques, Inês Brás, Giovannoni, Gavin, Marta, Monica
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4275932/
https://www.ncbi.nlm.nih.gov/pubmed/25494723
http://dx.doi.org/10.1186/s12883-014-0237-5
_version_ 1782350195066929152
author Marques, Inês Brás
Giovannoni, Gavin
Marta, Monica
author_facet Marques, Inês Brás
Giovannoni, Gavin
Marta, Monica
author_sort Marques, Inês Brás
collection PubMed
description BACKGROUND: Several disorders may present with mononeuritis multiplex and the etiological diagnosis can be challenging. CASE PRESENTATION: We report a 42 year-old female who presented with severe lower limb neuropathic pain, asymmetric weakness and sensory impairment and was diagnosed with mononeuritis multiplex. Biopsy showed a granulomatous vasculitic process with eosinophils, scarce granulomata and axonal neuropathy and granulomatosis with poliangiitis was assumed. Steroids, cyclophosphamide, alemtuzumab, azathioprine, mycophenolate mofetil and rituximab were used, all with transient and insufficient response. Skin biopsy performed in a further exacerbation allowed sarcoidosis diagnosis. Infliximab and, later, adalimumab induced good clinical and laboratorial response, but neutralizing antibodies developed to both drugs, so etanercept was tried with good clinical response. CONCLUSIONS: To the best of our knowledge, this is the first report of sarcoidosis successfully treated with etanercept. This drug may be considered in refractory sarcoidosis after other TNF-α inhibitors failure, having the advantage of not being associated with neutralizing antibodies development.
format Online
Article
Text
id pubmed-4275932
institution National Center for Biotechnology Information
language English
publishDate 2014
publisher BioMed Central
record_format MEDLINE/PubMed
spelling pubmed-42759322014-12-25 Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept Marques, Inês Brás Giovannoni, Gavin Marta, Monica BMC Neurol Case Report BACKGROUND: Several disorders may present with mononeuritis multiplex and the etiological diagnosis can be challenging. CASE PRESENTATION: We report a 42 year-old female who presented with severe lower limb neuropathic pain, asymmetric weakness and sensory impairment and was diagnosed with mononeuritis multiplex. Biopsy showed a granulomatous vasculitic process with eosinophils, scarce granulomata and axonal neuropathy and granulomatosis with poliangiitis was assumed. Steroids, cyclophosphamide, alemtuzumab, azathioprine, mycophenolate mofetil and rituximab were used, all with transient and insufficient response. Skin biopsy performed in a further exacerbation allowed sarcoidosis diagnosis. Infliximab and, later, adalimumab induced good clinical and laboratorial response, but neutralizing antibodies developed to both drugs, so etanercept was tried with good clinical response. CONCLUSIONS: To the best of our knowledge, this is the first report of sarcoidosis successfully treated with etanercept. This drug may be considered in refractory sarcoidosis after other TNF-α inhibitors failure, having the advantage of not being associated with neutralizing antibodies development. BioMed Central 2014-12-11 /pmc/articles/PMC4275932/ /pubmed/25494723 http://dx.doi.org/10.1186/s12883-014-0237-5 Text en © Marques et al.; licensee BioMed Central. 2014 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Marques, Inês Brás
Giovannoni, Gavin
Marta, Monica
Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title_full Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title_fullStr Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title_full_unstemmed Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title_short Mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
title_sort mononeuritis multiplex as the first presentation of refractory sarcoidosis responsive to etanercept
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4275932/
https://www.ncbi.nlm.nih.gov/pubmed/25494723
http://dx.doi.org/10.1186/s12883-014-0237-5
work_keys_str_mv AT marquesinesbras mononeuritismultiplexasthefirstpresentationofrefractorysarcoidosisresponsivetoetanercept
AT giovannonigavin mononeuritismultiplexasthefirstpresentationofrefractorysarcoidosisresponsivetoetanercept
AT martamonica mononeuritismultiplexasthefirstpresentationofrefractorysarcoidosisresponsivetoetanercept