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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...

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Autores principales: De Masi, Roberto, Accoto, Salvatore, Orlando, Stefania, De Blasi, Vincenzo, Pasca, Sergio, Scarpello, Rocco, Spagnolo, Leo, Idolo, Adele, De Donno, Antonella
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2015
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.
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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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