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Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption
BACKGROUND: Selective immune adsorption (SIA) is an emerging method for treating immune-mediated neurological diseases, given its superior safety profile compared to plasma exchange (PEX). However, the available literature concerning Multiple Sclerosis includes no cases of SIA applied to steroid-ref...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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BioMed Central
2015
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4521477/ https://www.ncbi.nlm.nih.gov/pubmed/26227815 http://dx.doi.org/10.1186/s12883-015-0377-2 |
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author | De Masi, Roberto Accoto, Salvatore Orlando, Stefania De Blasi, Vincenzo Pasca, Sergio Scarpello, Rocco Spagnolo, Leo Idolo, Adele De Donno, Antonella |
author_facet | De Masi, Roberto Accoto, Salvatore Orlando, Stefania De Blasi, Vincenzo Pasca, Sergio Scarpello, Rocco Spagnolo, Leo Idolo, Adele De Donno, Antonella |
author_sort | De Masi, Roberto |
collection | PubMed |
description | BACKGROUND: Selective immune adsorption (SIA) is an emerging method for treating immune-mediated neurological diseases, given its superior safety profile compared to plasma exchange (PEX). However, the available literature concerning Multiple Sclerosis includes no cases of SIA applied to steroid-refractory rebound after Fingolimod discontinuation. CASE PRESENTATION: Here we report the case of a 32-year-old woman suffering from multiple sclerosis treated with Fingolimod and admitted to a Multiple Sclerosis Centre after drug discontinuation due to the occurrence of lymphopenia. During the few weeks preceding admission, the patient experienced progressive and severe neurological deterioration that did not respond to an initial cycle of pulsed high doses of intravenous 6-methyl prednisolone (IVMP). Given the ineffectiveness of a second cycle of IVMP, the patient was treated with plasma immunoadsorption, leading to dramatic functional recovery. The patient then started a neuro-rehabilitation program. About one month after the final SIA procedure the patient started Natalizumab-based therapy, while maintaning a stable neurological condition. We noted significant modification of C3/C4 complement components and total gamma globulin concentrations (IgG) during SIA. CONCLUSIONS: Our observations show that however serious, steroid-refractory neurological deterioration occurring after Fingolimod discontinuation in multiple sclerosis can be treated with selective immune-adsorption therapy which thus represents a good alternative in these cases. It could be speculated that this clinical condition was associated with pattern II of demyelination, given the good response to a form of treatment that acts on autoantibodies. Thus, SIA represented an effective therapeutic strategy for this case of relapsed MS as steroid-resistent rebound post Fingolimod cessation. |
format | Online Article Text |
id | pubmed-4521477 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2015 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-45214772015-08-01 Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption De Masi, Roberto Accoto, Salvatore Orlando, Stefania De Blasi, Vincenzo Pasca, Sergio Scarpello, Rocco Spagnolo, Leo Idolo, Adele De Donno, Antonella BMC Neurol Case Report BACKGROUND: Selective immune adsorption (SIA) is an emerging method for treating immune-mediated neurological diseases, given its superior safety profile compared to plasma exchange (PEX). However, the available literature concerning Multiple Sclerosis includes no cases of SIA applied to steroid-refractory rebound after Fingolimod discontinuation. CASE PRESENTATION: Here we report the case of a 32-year-old woman suffering from multiple sclerosis treated with Fingolimod and admitted to a Multiple Sclerosis Centre after drug discontinuation due to the occurrence of lymphopenia. During the few weeks preceding admission, the patient experienced progressive and severe neurological deterioration that did not respond to an initial cycle of pulsed high doses of intravenous 6-methyl prednisolone (IVMP). Given the ineffectiveness of a second cycle of IVMP, the patient was treated with plasma immunoadsorption, leading to dramatic functional recovery. The patient then started a neuro-rehabilitation program. About one month after the final SIA procedure the patient started Natalizumab-based therapy, while maintaning a stable neurological condition. We noted significant modification of C3/C4 complement components and total gamma globulin concentrations (IgG) during SIA. CONCLUSIONS: Our observations show that however serious, steroid-refractory neurological deterioration occurring after Fingolimod discontinuation in multiple sclerosis can be treated with selective immune-adsorption therapy which thus represents a good alternative in these cases. It could be speculated that this clinical condition was associated with pattern II of demyelination, given the good response to a form of treatment that acts on autoantibodies. Thus, SIA represented an effective therapeutic strategy for this case of relapsed MS as steroid-resistent rebound post Fingolimod cessation. BioMed Central 2015-07-31 /pmc/articles/PMC4521477/ /pubmed/26227815 http://dx.doi.org/10.1186/s12883-015-0377-2 Text en © De Masi et al. 2015 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 De Masi, Roberto Accoto, Salvatore Orlando, Stefania De Blasi, Vincenzo Pasca, Sergio Scarpello, Rocco Spagnolo, Leo Idolo, Adele De Donno, Antonella Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title | Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title_full | Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title_fullStr | Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title_full_unstemmed | Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title_short | Dramatic recovery of steroid-refractory relapsed multiple sclerosis following Fingolimod discontinuation using selective immune adsorption |
title_sort | dramatic recovery of steroid-refractory relapsed multiple sclerosis following fingolimod discontinuation using selective immune adsorption |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4521477/ https://www.ncbi.nlm.nih.gov/pubmed/26227815 http://dx.doi.org/10.1186/s12883-015-0377-2 |
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