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TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis

OBJECTIVE: To assess the efficacy of tumour necrosis factor-α (TNF-α) inhibitors used as steroid-sparing monotherapy in central nervous system (CNS) parenchymal sarcoidosis. METHODS: The French Multiple Sclerosis and Neuroinflammation Centers retrospectively identified patients with definite or prob...

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Autores principales: Hilezian, Frédéric, Maarouf, Adil, Boutiere, Clemence, Rico, Audrey, Demortiere, Sarah, Kerschen, Philippe, Sene, Thomas, Bensa-Koscher, Caroline, Giannesini, Claire, Capron, Jean, Mekinian, Arsene, Camdessanché, Jean-Philippe, Androdias, Géraldine, Marignier, Romain, Collongues, Nicolas, Casez, Olivier, Coclitu, Catalina, Vaillant, Mathieu, Mathey, Guillaume, Ciron, Jonathan, Pelletier, Jean, Audoin, Bertrand
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BMJ Publishing Group 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8292597/
https://www.ncbi.nlm.nih.gov/pubmed/34103339
http://dx.doi.org/10.1136/jnnp-2020-325665
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author Hilezian, Frédéric
Maarouf, Adil
Boutiere, Clemence
Rico, Audrey
Demortiere, Sarah
Kerschen, Philippe
Sene, Thomas
Bensa-Koscher, Caroline
Giannesini, Claire
Capron, Jean
Mekinian, Arsene
Camdessanché, Jean-Philippe
Androdias, Géraldine
Marignier, Romain
Collongues, Nicolas
Casez, Olivier
Coclitu, Catalina
Vaillant, Mathieu
Mathey, Guillaume
Ciron, Jonathan
Pelletier, Jean
Audoin, Bertrand
author_facet Hilezian, Frédéric
Maarouf, Adil
Boutiere, Clemence
Rico, Audrey
Demortiere, Sarah
Kerschen, Philippe
Sene, Thomas
Bensa-Koscher, Caroline
Giannesini, Claire
Capron, Jean
Mekinian, Arsene
Camdessanché, Jean-Philippe
Androdias, Géraldine
Marignier, Romain
Collongues, Nicolas
Casez, Olivier
Coclitu, Catalina
Vaillant, Mathieu
Mathey, Guillaume
Ciron, Jonathan
Pelletier, Jean
Audoin, Bertrand
author_sort Hilezian, Frédéric
collection PubMed
description OBJECTIVE: To assess the efficacy of tumour necrosis factor-α (TNF-α) inhibitors used as steroid-sparing monotherapy in central nervous system (CNS) parenchymal sarcoidosis. METHODS: The French Multiple Sclerosis and Neuroinflammation Centers retrospectively identified patients with definite or probable CNS sarcoidosis treated with TNF-α inhibitors as steroid-sparing monotherapy. Only patients with CNS parenchymal involvement demonstrated by MRI and imaging follow-up were included. The primary outcome was the minimum dose of steroids reached that was not associated with clinical or imaging worsening during a minimum of 3 months after dosing change. RESULTS: Of the identified 38 patients with CNS sarcoidosis treated with TNF-α inhibitors, 23 fulfilled all criteria (13 females). Treatments were infliximab (n=22) or adalimumab (n=1) for a median (IQR) of 24 (17–40) months. At treatment initiation, the mean (SD) age was 41.5 (10.5) years and median (IQR) disease duration 22 (14–49.5) months. Overall, 60% of patients received other immunosuppressive agents before a TNF-α inhibitor. The mean (SD) minimum dose of steroids was 31.5 (33) mg before TNF-α inhibitor initiation and 6.5 (5.5) mg after (p=0.001). In all, 65% of patients achieved steroids dosing <6 mg/day; 61% showed clinical improvement, 30% stability and 9% disease worsening. Imaging revealed improvement in 74% of patients and stability in 26%. CONCLUSION: TNF-α inhibitors can greatly reduce steroids dosing in patients with CNS parenchymal sarcoidosis, even refractory. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that TNF-α inhibitor used as steroid-sparing monotherapy is effective for patients with CNS parenchymal sarcoidosis.
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spelling pubmed-82925972021-08-05 TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis Hilezian, Frédéric Maarouf, Adil Boutiere, Clemence Rico, Audrey Demortiere, Sarah Kerschen, Philippe Sene, Thomas Bensa-Koscher, Caroline Giannesini, Claire Capron, Jean Mekinian, Arsene Camdessanché, Jean-Philippe Androdias, Géraldine Marignier, Romain Collongues, Nicolas Casez, Olivier Coclitu, Catalina Vaillant, Mathieu Mathey, Guillaume Ciron, Jonathan Pelletier, Jean Audoin, Bertrand J Neurol Neurosurg Psychiatry Neuro-Inflammation OBJECTIVE: To assess the efficacy of tumour necrosis factor-α (TNF-α) inhibitors used as steroid-sparing monotherapy in central nervous system (CNS) parenchymal sarcoidosis. METHODS: The French Multiple Sclerosis and Neuroinflammation Centers retrospectively identified patients with definite or probable CNS sarcoidosis treated with TNF-α inhibitors as steroid-sparing monotherapy. Only patients with CNS parenchymal involvement demonstrated by MRI and imaging follow-up were included. The primary outcome was the minimum dose of steroids reached that was not associated with clinical or imaging worsening during a minimum of 3 months after dosing change. RESULTS: Of the identified 38 patients with CNS sarcoidosis treated with TNF-α inhibitors, 23 fulfilled all criteria (13 females). Treatments were infliximab (n=22) or adalimumab (n=1) for a median (IQR) of 24 (17–40) months. At treatment initiation, the mean (SD) age was 41.5 (10.5) years and median (IQR) disease duration 22 (14–49.5) months. Overall, 60% of patients received other immunosuppressive agents before a TNF-α inhibitor. The mean (SD) minimum dose of steroids was 31.5 (33) mg before TNF-α inhibitor initiation and 6.5 (5.5) mg after (p=0.001). In all, 65% of patients achieved steroids dosing <6 mg/day; 61% showed clinical improvement, 30% stability and 9% disease worsening. Imaging revealed improvement in 74% of patients and stability in 26%. CONCLUSION: TNF-α inhibitors can greatly reduce steroids dosing in patients with CNS parenchymal sarcoidosis, even refractory. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that TNF-α inhibitor used as steroid-sparing monotherapy is effective for patients with CNS parenchymal sarcoidosis. BMJ Publishing Group 2021-08 2021-06-08 /pmc/articles/PMC8292597/ /pubmed/34103339 http://dx.doi.org/10.1136/jnnp-2020-325665 Text en © Author(s) (or their employer(s)) 2021. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ. https://creativecommons.org/licenses/by-nc/4.0/This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) .
spellingShingle Neuro-Inflammation
Hilezian, Frédéric
Maarouf, Adil
Boutiere, Clemence
Rico, Audrey
Demortiere, Sarah
Kerschen, Philippe
Sene, Thomas
Bensa-Koscher, Caroline
Giannesini, Claire
Capron, Jean
Mekinian, Arsene
Camdessanché, Jean-Philippe
Androdias, Géraldine
Marignier, Romain
Collongues, Nicolas
Casez, Olivier
Coclitu, Catalina
Vaillant, Mathieu
Mathey, Guillaume
Ciron, Jonathan
Pelletier, Jean
Audoin, Bertrand
TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title_full TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title_fullStr TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title_full_unstemmed TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title_short TNF-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal CNS sarcoidosis
title_sort tnf-α inhibitors used as steroid-sparing maintenance monotherapy in parenchymal cns sarcoidosis
topic Neuro-Inflammation
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8292597/
https://www.ncbi.nlm.nih.gov/pubmed/34103339
http://dx.doi.org/10.1136/jnnp-2020-325665
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