Nakahashi Y, Miyazaki H, Kadota Y, Naitoh Y, Inoue K, Yamamoto M, Hayashi N, Taketani S
Third Department of Internal Medicine, Kansai Medical University, Osaka, Japan.
Hum Genet. 1993 May;91(4):303-6. doi: 10.1007/BF00217346.
We investigated the molecular basis of ferrochelatase in a Japanese patient with erythropoietic protoporphyria (EPP), complicated by fatal liver failure, and defined a novel point mutation in the ferrochelatase gene. cDNAs were synthesized using Epstein-Barr-virus-transformed lymphoblastoid cells from the proband. cDNA clones encoding ferrochelatase in the proband were isolated by amplification using the polymerase chain reaction. There were two sizes of ferrochelatase cDNAs; one was normal in size, the other being smaller. Sequence analysis of the abnormally sized cDNA clones revealed that they lacked exon 9 of the ferrochelatase gene. Genomic DNA analysis demonstrated that the proband had the abnormal allele and that it contained a G to A point mutation at the first position of the donor site of intron 9. An identical mutation was detected in the affected family members of the proband by allele-specific oligonucleotide hybridization analysis. EPP is inherited in an autosomal dominant manner in this family.
我们研究了一名患有红细胞生成性原卟啉症(EPP)并并发致命性肝功能衰竭的日本患者体内铁螯合酶的分子基础,并确定了铁螯合酶基因中的一个新的点突变。使用先证者的爱泼斯坦-巴尔病毒转化的淋巴母细胞系合成cDNA。通过聚合酶链反应扩增,从先证者中分离出编码铁螯合酶的cDNA克隆。铁螯合酶cDNA有两种大小;一种大小正常,另一种较小。对异常大小的cDNA克隆进行序列分析发现,它们缺少铁螯合酶基因的外显子9。基因组DNA分析表明,先证者具有异常等位基因,并且该等位基因在第9内含子供体位点的第一个位置存在G到A的点突变。通过等位基因特异性寡核苷酸杂交分析,在先证者的患病家庭成员中检测到相同的突变。在这个家族中,EPP以常染色体显性方式遗传。