Botto M, Fong K Y, So A K, Rudge A, Walport M J
Department of Medicine, Royal Postgraduate Medical School, London, United Kingdom.
J Clin Invest. 1990 Oct;86(4):1158-63. doi: 10.1172/JCI114821.
Hereditary deficiency of complement component C3 in a 10-yr-old boy was studied. C3 could not be detected by RIA of serum from the patient. Segregation of C3 S and C3 F allotypes within the family confirmed the presence of a null gene for C3, for which the patient was homozygous. 30 exons have been characterized, spanning the entire beta chain of C3 and the alpha chain as far as the C3d region. Sequence analysis of the exons derived from the C3 null gene showed no abnormalities in the coding sequences. A GT-AT mutation at the 5' donor splice site of the intervening sequence 18 was found in the C3 null gene. Exons 17-21 were amplified by the polymerase chain reaction (PCR) from first-strand cDNA synthesized from mRNA obtained from peripheral blood monocytes stimulated with LPS. This revealed a 61-bp deletion in exon 18, resulting from splicing of a cryptic 5' donor splice site in exon 18 with the normal 3' splice site in exon 19. This deletion leads to a disturbance of the reading frame of the mRNA with a stop codon 17 bp downstream from the abnormal splice in exon 18. His parents had both the normal and abnormal C3 mRNA and were shown to be heterozygous for this mutation by sequence analysis of genomic DNA amplified by PCR. Similar splice mutants have previously been reported in the beta-globin, phenylalanine hydroxylase, and porphobilinogen deaminase genes. This mutation is sufficient to cause the deficiency of C3 in the patient.
对一名10岁男孩遗传性补体成分C3缺乏症进行了研究。通过放射免疫分析法未在该患者血清中检测到C3。家族内C3 S和C3 F同种异型的分离证实存在C3无效基因,该患者为该基因的纯合子。已鉴定出30个外显子,涵盖C3的整个β链以及直至C3d区域的α链。对源自C3无效基因的外显子进行序列分析,结果显示编码序列无异常。在C3无效基因中,发现内含子序列18的5'供体剪接位点处存在GT-AT突变。通过聚合酶链反应(PCR)从用脂多糖刺激的外周血单核细胞获得的mRNA合成的第一链cDNA中扩增外显子17 - 21。这揭示了外显子18中61个碱基对的缺失,这是由于外显子18中一个隐蔽的5'供体剪接位点与外显子19中的正常3'剪接位点拼接所致。这种缺失导致mRNA阅读框紊乱,在异常剪接下游17个碱基对处出现终止密码子。他的父母同时具有正常和异常的C3 mRNA,通过对PCR扩增的基因组DNA进行序列分析,显示他们是该突变的杂合子。先前在β-珠蛋白、苯丙氨酸羟化酶和胆色素原脱氨酶基因中也报道过类似的剪接突变体。这种突变足以导致患者C3缺乏。