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无义突变导致阿非利堪德牛出现遗传性甲状腺肿,并揭示了甲状腺球蛋白转录本的可变剪接。

A nonsense mutation causes hereditary goitre in the Afrikander cattle and unmasks alternative splicing of thyroglobulin transcripts.

作者信息

Ricketts M H, Simons M J, Parma J, Mercken L, Dong Q, Vassart G

出版信息

Proc Natl Acad Sci U S A. 1987 May;84(10):3181-4. doi: 10.1073/pnas.84.10.3181.

Abstract

The hereditary goitre of Afrikander cattle is an autosomal recessive disease characterized in homozygotes by the production of abnormal thyroglobulin (Tg) and the coexistence in the thyroid of normal-sized 8.4-kilobase (kb) Tg mRNA with a misspliced 7.3-kb message having lost exon 9. We have cloned and sequenced the cDNA segment corresponding to the abnormal exon 8-exon 10 junction and the relevant genomic DNA region. The mutation responsible for the disease is a cytosine to thymine transition creating a stop codon at position 697 in exon 9. The original reading frame is maintained in the 7.3-kb mRNA, which, as it lacks the mutated exon, is translatable into a potentially functional protein. This puzzling phenotype in which a mutated exon is apparently removed selectively from transcripts by alternative splicing leads us to suggest that the 7.3-kb transcript could be present in normal animals. Using a sensitive oligonucleotide hybridization assay, we have demonstrated that a 7.3-kb mRNA lacking exon 9 does exist in normal thyroids as a minor mRNA species. As it is fully translatable, the 7.3-kb mRNA is expected to be more stable than the normal-sized 8.4-kb message. This probably accounts for the higher proportion of 7.3-kb transcript found in the goitre.

摘要

非洲瘤牛的遗传性甲状腺肿是一种常染色体隐性疾病,其纯合子的特征是产生异常甲状腺球蛋白(Tg),并且在甲状腺中正常大小的8.4千碱基(kb)Tg mRNA与缺失外显子9的错配剪接的7.3-kb信息共存。我们已经克隆并测序了与异常外显子8 - 外显子10连接相对应的cDNA片段以及相关的基因组DNA区域。导致该疾病的突变是胞嘧啶到胸腺嘧啶的转变,在外显子9的第697位产生了一个终止密码子。原始阅读框在7.3-kb mRNA中得以保留,由于它缺少突变的外显子,所以可以翻译成一种潜在的功能性蛋白质。这种令人困惑的表型,即突变的外显子显然通过可变剪接从转录本中被选择性去除,使我们推测7.3-kb转录本可能存在于正常动物中。使用灵敏的寡核苷酸杂交分析,我们已经证明在正常甲状腺中确实存在缺少外显子9的7.3-kb mRNA,作为一种次要的mRNA种类。由于它可以完全翻译,预计7.3-kb mRNA比正常大小的8.4-kb信息更稳定。这可能解释了在甲状腺肿中发现的7.3-kb转录本比例较高的原因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f297/304832/15e429af3771/pnas00275-0111-a.jpg

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