Heisterkamp N, Mulder M P, Langeveld A, ten Hoeve J, Wang Z, Roe B A, Groffen J
Department of Pathology, Children's Hospital of Los Angeles, California 90027, USA.
Genomics. 1995 Sep 20;29(2):451-6. doi: 10.1006/geno.1995.9982.
A high percentage of patients with DiGeorge syndrome and velo-cardio-facial syndrome have interstitial deletions on chromosome 22q11. The shortest region of overlap is currently estimated to be around 55 kb. Two segments of DNA from chromosome 22q11, located 160 kb apart, were cloned because they contained NotI restriction enzyme sites. In the current study we demonstrate that these segments are absent from chromosomes 22 carrying microdeletions of two different DiGeorge patients. Fluorescence in situ and Southern blot hybridization was further used to show that this locus is within the DiGeorge critical region. Phylogenetically conserved sequences adjacent to one human cell lines. cDNAs isolated with a probe from this segment showed it to contain the gene for teh human mitochondrial citrate transporter protein. Deletion of this gene in DiGeorge syndrome and velocardio-facial syndrome may contribute to the mental deficiency seen in the patients.
高比例的迪乔治综合征和腭心面综合征患者在22号染色体q11区域存在间质性缺失。目前估计最短重叠区域约为55 kb。来自22号染色体q11的两段DNA,相距160 kb,因其含有NotI限制性酶切位点而被克隆。在本研究中,我们证明携带两名不同迪乔治综合征患者微缺失的22号染色体上不存在这些片段。荧光原位杂交和Southern印迹杂交进一步表明该基因座位于迪乔治关键区域内。与一个人类细胞系相邻的系统发育保守序列。用该片段的探针分离的cDNA表明它包含人类线粒体柠檬酸转运蛋白的基因。迪乔治综合征和腭心面综合征中该基因的缺失可能导致患者出现智力缺陷。