Abstract

Abstract: Autoimmunity is often observed among individuals with primary immune deficiencies; however, the frequency and role of autoimmunity in Schimke immuno-osseous dysplasia (SIOD) has not been fully assessed. SIOD, which is caused by mutations of SMARCAL1 , is a rare autosomal recessive disease with its prominent features being skeletal dysplasia, T cell deficiency, and renal failure. We present a child with severe SIOD who developed rituximab resistant Evans syndrome (ES). Consistent with observations in several other immunodeficiency disorders, a review of SIOD patients showed that approximately a fifth of SIOD patients have some features of autoimmune disease. To our best knowledge this case represents the first patient with SIOD and rituximab resistant ES and the first study of autoimmune disease in SIOD.

Details

Title
Rituximab resistant evans syndrome and autoimmunity in Schimke immuno-osseous dysplasia
Author
Zieg, Jakub; Krepelova, Anna; Baradaran-Heravi, Alireza; Levtchenko, Elena; Guillén-Navarro, Encarna; Balascakova, Miroslava; Sukova, Martina; Seeman, Tomas; Dusek, Jiri; Simankova, Nadezda; Rosik, Tomas; Skalova, Sylva; Lebl, Jan; Boerkoel, Cornelius F
Pages
27
Publication year
2011
Publication date
2011
Publisher
BioMed Central
Source type
Scholarly Journal
Language of publication
English
ProQuest document ID
902265039
Copyright
© 2011 Zieg et al; licensee BioMed Central Ltd. 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.