%0 Journal Article
%T Prognosis of refractory neurosarcoidosis altered by thalidomide: a case report
%A J Chad Hoyle
%A Herbert B Newton
%A Steven Katz
%J Journal of Medical Case Reports
%D 2008
%I BioMed Central
%R 10.1186/1752-1947-2-27
%X 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.Our case report presents a difficult, refractory case of neurosarcoidosis that demonstrates an altered prognosis based on the addition of thalidomide.Sarcoidosis is a granulomatous, inflammatory disease that most commonly involves the lungs, skin, and eyes, but can also affect the nervous system in about 5% of cases [1]. Though it is common for the disease to spontaneously remit, nervous system involvement is a marker for a more resistant course [2]. Because of this, nervous system involvement is a definite indication for corticosteroid treatment. Unfortunately, 25% will still have a refractory course with steroid treatment and, even more concerning, 20¨C40% of those refractory patients will not respond to any level of current conventional immunosuppression [1,3].Despite the identification of several possible candidates, the exact inciting antigen responsible for the formation of sarcoid granulomas remains uncertain. However, the resultant inflammatory cascade involved in mediating granuloma formation has been mapped out more accurately. It involves macrophages or dendritic cells ingesting the antigen with resultant peptide fragments bound to MHC-II complexes that ignite a polarized TH-1 inflammatory cascade. This involves Interleukin-2, Interleukin-12, interferon-gamma (IFN-¦Ã), and tumor necrosis factor-alpha (TNF-¦Á). Specifically, TNF-¦Á has gained clinical attention with the availability of TNF-¦Á blocking agents [4]. The prognostic outcomes have been based on conventional immunosuppression that attacks this process in a non-specific fashion, but we present a case th
%U http://www.jmedicalcasereports.com/content/2/1/27