Germline mutations of the LKB1 (STK11) gene in Peutz-Jeghers patients

被引:0
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作者
Wang, ZJ
Churchman, M
Avizienyte, E
McKeown, C
Davies, S
Evans, DGR
Ferguson, A
Ellis, I
Xu, WH
Yan, ZY
Aaltonen, LA
Tomlinson, IPM [1 ]
机构
[1] Univ Oxford, John Radcliffe Hosp, Tumour Genet Grp, Dept Clin Med, Oxford OX3 9DU, England
[2] Beijing Univ Med, Sch Med 1, Dept Surg, Beijing, Peoples R China
[3] Univ Helsinki, Haartman Inst, Dept Med Genet, FIN-00014 Helsinki, Finland
[4] Birmingham Womens Hosp, Dept Clin Genet, Birmingham B15 2TG, W Midlands, England
[5] Univ Wales Hosp, Inst Med Genet, Cardiff CF4 4XN, S Glam, Wales
[6] St Marys Hosp, Dept Clin Genet, Manchester M13 0JH, Lancs, England
[7] Western Gen Hosp, Dept Gastroenterol, Edinburgh EH4 2XU, Midlothian, Scotland
[8] Royal Liverpool Childrens Hosp, Dept Clin Genet, Liverpool L12 2AP, Merseyside, England
[9] Imperial Canc Res Fund, Mol & Populat Genet Lab, London WC2A 3PX, England
关键词
Peutz-Jeghers syndrome; LKB1/STK11;
D O I
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中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Germline mutations of the LKB1 (STK11) serine/threonine kinase gene (chromosome 19p13.3) cause Peutz-Jeghers syndrome, which is characterised by hamartomas of the gastrointestinal tract and typical pigmentation. Peutz-Jeghers syndrome carries an overall risk of cancer that may be up to 20 times that of the general population. Here, we report the results of a screen for germline LKB1 mutations by DNA sequencing in 12 Peutz-Jeghers patients (three sporadic and nine familial cases). Mutations were found in seven (58%) cases, in exons 1, 2, 4, 6, and 9. Five of these mutations, two of which are identical, are predicted to lead to a truncated protein (three frameshifts, two nonsense changes). A further mutation is an in frame deletion of 6 bp, resulting in a deletion of lysine and asparagine; the second of these amino acids is conserved between species. The seventh mutation is a missense change in exon 2, converting lysine to arginine, affecting non-conserved amino acids and of uncertain functional significance. Despite the fact that Peutz-Jeghers syndrome is usually an early onset disease with characteristic clinical features, predictive and diagnostic testing for LKB1 mutations will be useful for selected patients in both familial and non-familial contexts.
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页码:365 / 368
页数:4
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