Prognosis of refractory neurosarcoidosis altered by thalidomide: a case reportReport as inadecuate




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Journal of Medical Case Reports

, 2:27

First Online: 28 January 2008Received: 14 September 2007Accepted: 28 January 2008DOI: 10.1186-1752-1947-2-27

Cite this article as: Hoyle, J.C., Newton, H.B. & Katz, S. J Med Case Reports 2008 2: 27. doi:10.1186-1752-1947-2-27

Abstract

IntroductionSarcoidosis 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 presentationA 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.

ConclusionOur case report presents a difficult, refractory case of neurosarcoidosis that demonstrates an altered prognosis based on the addition of thalidomide.

AbbreviationsODright eye

OSleft eye

MRIMagnetic Resonance Imaging

CTCat Scan

MHC-IIMajor Histocompatibility-II

IFN-γinterferon-gamma

TNF-αTumor necrosis factor-alpha

MSMultiple Sclerosis

ACEangiotensin converting enzyme

QDdaily

QODevery other day

BIDtwice daily

TH-1T helper-1

STEPSSystem for Thalidomide Education and Prescribing Safety

Electronic supplementary materialThe online version of this article doi:10.1186-1752-1947-2-27 contains supplementary material, which is available to authorized users.

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Author: J. Chad Hoyle - Herbert B. Newton - Steven Katz

Source: https://link.springer.com/







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