Woods-Samuels P, Wong C, Mathias S L, Scott A F, Kazazian H H, Antonarakis S E
Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, Maryland 21205.
Genomics. 1989 Apr;4(3):290-6. doi: 10.1016/0888-7543(89)90332-7.
We have characterized an insertional event in IVS-10 of the factor VIII gene in a pedigree containing a hemophilia A patient (JH-25). The inserted DNA is a 5' truncated L1 element that is 681 bp long followed by a 3'66-bp poly(A) tract. The L1 element is inserted 154 bp 5' to the start of exon 11 and is flanked by a 13- to 17-bp target site duplication. The L1 insertion is present in four generations of the patient's family. The maternal grandfather who carries the insertion does not have hemophilia A, indicating that the insertion is not the cause of hemophilia A in the patient. We have sequenced this insertion and two previously reported de novo L1 insertions in the factor VIII gene in patients JH-27 (3785 bp) and JH-28 (2132 bp). The three nucleotide sequences differ by 0.2-0.8%. All three of these L1 insertions have open reading frames (ORFs) (1192, 642, and 157 aa) and the three derived amino acid sequences are 98-99% identical. The previously reported sequence similarity between L1 3' ORFs and the polymerase domain of reverse transcriptases is maintained in the ORFs of the JH-27 and JH-28 L1 insertions. The presence of open reading frames and the close sequence similarity of these recently inserted L1 elements provide indirect evidence for the existence of a set of functional L1 elements that encode one or more proteins necessary for their retrotransposition.
我们对一个包含甲型血友病患者(JH - 25)的家系中凝血因子VIII基因IVS - 10的插入事件进行了特征分析。插入的DNA是一个5'端截短的L1元件,长681 bp,后面跟着一个3'端66 bp的多聚腺苷酸尾。该L1元件插入到外显子11起始位点上游154 bp处,两侧有13至17 bp的靶位点重复序列。L1插入存在于患者家族的四代人中。携带该插入的外祖父没有甲型血友病,这表明该插入不是患者甲型血友病的病因。我们对这个插入序列以及先前报道的患者JH - 27(3785 bp)和JH - 28(2132 bp)凝血因子VIII基因中的两个新生L1插入序列进行了测序。这三个核苷酸序列的差异为0.2 - 0.8%。所有这三个L1插入序列都有开放阅读框(ORF)(分别为1192、642和157个氨基酸),并且三个推导的氨基酸序列有98 - 99%的同一性。先前报道的L1 3'端ORF与逆转录酶聚合酶结构域之间的序列相似性在JH - 27和JH - 28的L1插入序列的ORF中得以保留。这些最近插入的L1元件存在开放阅读框以及紧密的序列相似性,为存在一组功能性L1元件提供了间接证据,这些元件编码其逆转录转座所需的一种或多种蛋白质。