首页 | 本学科首页   官方微博 | 高级检索  
检索        


Structure and function of the Wiskott-Aldrich syndrome protein
Authors:Ochs Hans D  Notarangelo Luigi D
Institution:Departments of Pediatrics, University of Washington, Seattle, Washington 98109, USA. hans.ochs@seattlechildrens.org
Abstract:PURPOSE OF REVIEW: Mutations of the Wiskott-Aldrich syndrome protein can result in highly variable clinical symptoms that affect the hematopoietic/immunologic system. The responsible gene, WASP, has multiple domains, each with unique functions that were only recently fully recognized. RECENT FINDINGS: Two new comprehensive studies of patients with mutations of the Wiskott-Aldrich syndrome protein unequivocally demonstrated a strong phenotype-genotype correlation; the most predictive variable was the presence or absence of the Wiskott-Aldrich syndrome protein in the lymphoid cells from patients with X-linked thrombocytopenia or Wiskott-Aldrich syndrome, respectively. A third clinical study revealed a high rate (>70%) of autoimmune disorders in patients with classic Wiskott-Aldrich syndrome, possibly caused by immune dysregulation involving both T and B cell defects. In addition, the Wiskott-Aldrich syndrome protein is required for natural killer cell function by participating in the formation of immunologic synapses and facilitating the nuclear translocation of nuclear factor for activated T cell and nuclear factor-kappaB. Finally, the Wiskott-Aldrich syndrome protein was shown to play an important role in lymphoid development and in the maturation and function of myelomonocytic cells. SUMMARY: The progress made in dissecting the functions of the Wiskott-Aldrich syndrome protein has direct implications for our understanding of the distinct clinical phenotypes (Wiskott-Aldrich syndrome/X-linked thrombocytopenia; intermittent thrombocytopenia; congenital neutropenia), for making diagnostic and prognostic decisions, and for the selection of therapeutic strategies--from conservative symptomatic treatment to curative hematopoietic stem cell transplantation, or, in the future, gene therapy.
Keywords:
本文献已被 PubMed 等数据库收录!
设为首页 | 免责声明 | 关于勤云 | 加入收藏

Copyright©北京勤云科技发展有限公司  京ICP备09084417号