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Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report
INTRODUCTION: Sarcoidosis is a multisystem disease characterized by noncaseating granulomas in the lungs, skin, lymph nodes, and, rarely, the nervous system. Granuloma formation in sarcoidosis is mediated by increased secretion of interferon-gamma, interleukin-2, and tumor necrosis factor-alpha. 25%...
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Formato: | Texto |
Lenguaje: | English |
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BioMed Central
2008
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2249602/ https://www.ncbi.nlm.nih.gov/pubmed/18226232 http://dx.doi.org/10.1186/1752-1947-2-27 |
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author | Hoyle, J Chad Newton, Herbert B Katz, Steven |
author_facet | Hoyle, J Chad Newton, Herbert B Katz, Steven |
author_sort | Hoyle, J Chad |
collection | PubMed |
description | INTRODUCTION: Sarcoidosis is a multisystem disease characterized by noncaseating granulomas in the lungs, skin, lymph nodes, and, rarely, the nervous system. Granuloma formation in sarcoidosis is mediated by increased secretion of interferon-gamma, interleukin-2, and tumor necrosis factor-alpha. 25% of patients with neurosarcoidosis are steroid resistant and another 20–40% are resistant to any conventional immunosuppression, but the typical agents suppress the immune system in a non-specific fashion. Thalidomide has been shown to have activity specific to the inflammatory mediators of sarcoidosis, has been shown to be beneficial in cutaneous sarcoidosis, and provides an interesting observation in our patient with refractory neurosarcoidosis. CASE PRESENTATION: A 40 year old African-american female presented with refractory neurosarcoidosis. Over the course of several years, the patient was treated with high dose steroids, imuran, cytoxan, and cyclosporine without benefit. Then, the patient received thalidomide, slowly escalating to 650 mg. After 2 months radiologic improvement was noted and after 6 months clinical stabilization and improvement became apparent. CONCLUSION: Our case report presents a difficult, refractory case of neurosarcoidosis that demonstrates an altered prognosis based on the addition of thalidomide. |
format | Text |
id | pubmed-2249602 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2008 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-22496022008-02-22 Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report Hoyle, J Chad Newton, Herbert B Katz, Steven J Med Case Reports Case Report INTRODUCTION: Sarcoidosis is a multisystem disease characterized by noncaseating granulomas in the lungs, skin, lymph nodes, and, rarely, the nervous system. Granuloma formation in sarcoidosis is mediated by increased secretion of interferon-gamma, interleukin-2, and tumor necrosis factor-alpha. 25% of patients with neurosarcoidosis are steroid resistant and another 20–40% are resistant to any conventional immunosuppression, but the typical agents suppress the immune system in a non-specific fashion. Thalidomide has been shown to have activity specific to the inflammatory mediators of sarcoidosis, has been shown to be beneficial in cutaneous sarcoidosis, and provides an interesting observation in our patient with refractory neurosarcoidosis. CASE PRESENTATION: A 40 year old African-american female presented with refractory neurosarcoidosis. Over the course of several years, the patient was treated with high dose steroids, imuran, cytoxan, and cyclosporine without benefit. Then, the patient received thalidomide, slowly escalating to 650 mg. After 2 months radiologic improvement was noted and after 6 months clinical stabilization and improvement became apparent. CONCLUSION: Our case report presents a difficult, refractory case of neurosarcoidosis that demonstrates an altered prognosis based on the addition of thalidomide. BioMed Central 2008-01-28 /pmc/articles/PMC2249602/ /pubmed/18226232 http://dx.doi.org/10.1186/1752-1947-2-27 Text en Copyright © 2008 Hoyle et al; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( (http://creativecommons.org/licenses/by/2.0) ), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Hoyle, J Chad Newton, Herbert B Katz, Steven Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title | Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title_full | Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title_fullStr | Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title_full_unstemmed | Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title_short | Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
title_sort | prognosis of refractory neurosarcoidosis altered by thalidomide: a case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2249602/ https://www.ncbi.nlm.nih.gov/pubmed/18226232 http://dx.doi.org/10.1186/1752-1947-2-27 |
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