Dostal A, Linnankivi T, Somer M, Kähkönen M, Litzman J, Tienari P
Center for Neurologic Diseases, Brigham Women Hospital and Harvard Medical School, Boston, MA 02115, USA.
Int J Immunogenet. 2007 Jun;34(3):143-7. doi: 10.1111/j.1744-313X.2007.00652.x.
This study presents a clinical report of the Finnish chromosome t(18q; 10p) translocation family with an overview of eight other selected immunoglobulin A (IgA)-deficient 18q deletion (18q-) patients from seven published articles. The family members show features common to 18q- syndrome such as mental retardation, multiple facial dysmorphism, foot/hand deformities, abnormal myelination of brain white matter, and a spectrum of immunological/infectious disorders including IgA deficiency (IgAD). Genotype-phenotype correlation study of the unbalanced t(18q-; 10p+) translocation family members and other 18q- syndrome reports led to definition of a potential susceptibility gene locus for IgAD at distal region of 18q22.3-q23 between markers D18S812-18qter. The haplo-insufficiency of the 18q22.3-q23 gene region is suggested to be a cause of the IgAD phenotype in 18q- individuals. This 7 Mb IgAD critical region shows significant association with susceptibility region for celiac disease that is frequently connected to IgAD.
本研究报告了芬兰染色体t(18q; 10p)易位家族的临床情况,并概述了从七篇已发表文章中选取的其他八例免疫球蛋白A(IgA)缺陷型18q缺失(18q-)患者。家族成员表现出18q-综合征的常见特征,如智力迟钝、多种面部畸形、手足畸形、脑白质髓鞘形成异常,以及一系列免疫/感染性疾病,包括IgA缺陷(IgAD)。对不平衡t(18q-; 10p+)易位家族成员和其他18q-综合征报告进行的基因型-表型相关性研究,确定了18q22.3-q23远端区域(介于标记D18S812与18qter之间)为IgAD的一个潜在易感基因位点。18q22.3-q23基因区域的单倍体不足被认为是18q-个体中IgAD表型的一个原因。这个7 Mb的IgAD关键区域与乳糜泻的易感区域有显著关联,而乳糜泻常与IgAD相关。