Roscioli Tony, Cliffe Simon T, Bloch Donald B, Bell Christopher G, Mullan Glenda, Taylor Peter J, Sarris Maria, Wang Joanne, Donald Jennifer A, Kirk Edwin P, Ziegler John B, Salzer Ulrich, McDonald George B, Wong Melanie, Lindeman Robert, Buckley Michael F
Centre for Vascular Research, University of New South Wales, Sydney 2052, Australia.
Nat Genet. 2006 Jun;38(6):620-2. doi: 10.1038/ng1780. Epub 2006 Apr 30.
We describe mutations in the PML nuclear body protein Sp110 in the syndrome veno-occlusive disease with immunodeficiency, an autosomal recessive disorder of severe hypogammaglobulinemia, combined T and B cell immunodeficiency, absent lymph node germinal centers, absent tissue plasma cells and hepatic veno-occlusive disease. This is the first report of the involvement of a nuclear body protein in a human primary immunodeficiency and of high-penetrance genetic mutations in hepatic veno-occlusive disease.
我们描述了PML核体蛋白Sp110在静脉闭塞性疾病伴免疫缺陷综合征中的突变情况,该综合征是一种常染色体隐性疾病,表现为严重低丙种球蛋白血症、T和B细胞联合免疫缺陷、淋巴结生发中心缺失、组织浆细胞缺失以及肝静脉闭塞性疾病。这是关于核体蛋白参与人类原发性免疫缺陷以及肝静脉闭塞性疾病中高外显率基因突变的首次报道。