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PMID: 19602640 Published · ppublish English Journal Article Research Support, N.I.H., Intramural

The ClinSeq Project: piloting large-scale genome sequencing for research in genomic medicine.

Genome research ·Vol. 19 ·No. 9 ·2009-09-00 ·Pages 1665-74

Biesecker LG, Mullikin JC, Facio FM, Turner C, Cherukuri PF, Blakesley RW, Bouffard GG, Chines PS, Cruz P, Hansen NF, Teer JK, Maskeri B, Young AC, NISC Comparative Sequencing Program, Manolio TA, Wilson AF, Finkel T, Hwang P, Arai A, Remaley AT, Sachdev V, Shamburek R, Cannon RO, Green ED

Abstract

ClinSeq is a pilot project to investigate the use of whole-genome sequencing as a tool for clinical research. By piloting the acquisition of large amounts of DNA sequence data from individual human subjects, we are fostering the development of hypothesis-generating approaches for performing research in genomic medicine, including the exploration of issues related to the genetic architecture of disease, implementation of genomic technology, informed consent, disclosure of genetic information, and archiving, analyzing, and displaying sequence data. In the initial phase of ClinSeq, we are enrolling roughly 1000 participants; the evaluation of each includes obtaining a detailed family and medical history, as well as a clinical evaluation. The participants are being consented broadly for research on many traits and for whole-genome sequencing. Initially, Sanger-based sequencing of 300-400 genes thought to be relevant to atherosclerosis is being performed, with the resulting data analyzed for rare, high-penetrance variants associated with specific clinical traits. The participants are also being consented to allow the contact of family members for additional studies of sequence variants to explore their potential association with specific phenotypes. Here, we present the general considerations in designing ClinSeq, preliminary results based on the generation of an initial 826 Mb of sequence data, the findings for several genes that serve as positive controls for the project, and our views about the potential implications of ClinSeq. The early experiences with ClinSeq illustrate how large-scale medical sequencing can be a practical, productive, and critical component of research in genomic medicine.

MeSH Terms
Aged Atherosclerosis/genetics Biomedical Research Cardiovascular Diseases/genetics Cohort Studies Female Genome, Human Genomics Humans Male Pedigree Phenotype Pilot Projects Sequence Analysis, DNA/methods
Authors & Affiliations
24 authors, click to expand affiliations / ORCID
Biesecker Leslie G
National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland 20892, USA. [email protected]
Mullikin James C
Facio Flavia M
Turner Clesson
Cherukuri Praveen F
Blakesley Robert W
Bouffard Gerard G
Chines Peter S
Cruz Pedro
Hansen Nancy F
Teer Jamie K
Maskeri Baishali
Young Alice C
NISC Comparative Sequencing Program
Manolio Teri A
Wilson Alexander F
Finkel Toren
Hwang Paul
Arai Andrew
Remaley Alan T
Sachdev Vandana
Shamburek Robert
Cannon Richard O
Green Eric D
References (33)
33 references, click to expand
  1. Prediction of coronary heart disease using risk factor categories.
    Circulation. 1998 May 12;97(18):1837-47 PMID: 9603539
  2. Automating sequence-based detection and genotyping of SNPs from diploid samples.
    Nat Genet. 2006 Mar;38(3):375-81 PMID: 16493422
  3. A genome-wide association study for blood lipid phenotypes in the Framingham Heart Study.
    BMC Med Genet. 2007 Sep 19;8 Suppl 1:S17 PMID: 17903299
  4. Consed: a graphical tool for sequence finishing.
    Genome Res. 1998 Mar;8(3):195-202 PMID: 9521923
  5. Massively parallel exon capture and library-free resequencing across 16 genomes.
    Nat Methods. 2009 May;6(5):315-6 PMID: 19349981
  6. The diploid genome sequence of an individual human.
    PLoS Biol. 2007 Sep 4;5(10):e254 PMID: 17803354
  7. Human non-synonymous SNPs: server and survey.
    Nucleic Acids Res. 2002 Sep 1;30(17):3894-900 PMID: 12202775
  8. Angiopoietin-like protein 4 converts lipoprotein lipase to inactive monomers and modulates lipase activity in adipose tissue.
    Proc Natl Acad Sci U S A. 2006 Nov 14;103(46):17450-5 PMID: 17088546
  9. A second generation human haplotype map of over 3.1 million SNPs.
    Nature. 2007 Oct 18;449(7164):851-61 PMID: 17943122
  10. Initial sequencing and analysis of the human genome.
    Nature. 2001 Feb 15;409(6822):860-921 PMID: 11237011
  11. A HapMap harvest of insights into the genetics of common disease.
    J Clin Invest. 2008 May;118(5):1590-605 PMID: 18451988
  12. Rare loss-of-function mutations in ANGPTL family members contribute to plasma triglyceride levels in humans.
    J Clin Invest. 2009 Jan;119(1):70-9 PMID: 19075393
  13. The complete genome of an individual by massively parallel DNA sequencing.
    Nature. 2008 Apr 17;452(7189):872-6 PMID: 18421352
  14. SIFT: Predicting amino acid changes that affect protein function.
    Nucleic Acids Res. 2003 Jul 1;31(13):3812-4 PMID: 12824425
  15. Rare independent mutations in renal salt handling genes contribute to blood pressure variation.
    Nat Genet. 2008 May;40(5):592-599 PMID: 18391953
  16. The impact of next-generation sequencing technology on genetics.
    Trends Genet. 2008 Mar;24(3):133-41 PMID: 18262675
  17. Sequence variations in PCSK9, low LDL, and protection against coronary heart disease.
    N Engl J Med. 2006 Mar 23;354(12):1264-72 PMID: 16554528
  18. Automating resequencing-based detection of insertion-deletion polymorphisms.
    Nat Genet. 2006 Dec;38(12):1457-62 PMID: 17115056
  19. The diploid genome sequence of an Asian individual.
    Nature. 2008 Nov 6;456(7218):60-5 PMID: 18987735
  20. Disclosure of the right of research participants to receive research results: an analysis of consent forms in the Children's Oncology Group.
    Cancer. 2003 Jun 1;97(11):2904-9 PMID: 12767106
  21. Base-calling of automated sequencer traces using phred. II. Error probabilities.
    Genome Res. 1998 Mar;8(3):186-94 PMID: 9521922
  22. Resolving individuals contributing trace amounts of DNA to highly complex mixtures using high-density SNP genotyping microarrays.
    PLoS Genet. 2008 Aug 29;4(8):e1000167 PMID: 18769715
  23. Patterns of single-nucleotide polymorphisms in candidate genes for blood-pressure homeostasis.
    Nat Genet. 1999 Jul;22(3):239-47 PMID: 10391210
  24. Accurate whole human genome sequencing using reversible terminator chemistry.
    Nature. 2008 Nov 6;456(7218):53-9 PMID: 18987734
  25. Association between a specific apolipoprotein B mutation and familial defective apolipoprotein B-100.
    Proc Natl Acad Sci U S A. 1989 Jan;86(2):587-91 PMID: 2563166
  26. Finishing the euchromatic sequence of the human genome.
    Nature. 2004 Oct 21;431(7011):931-45 PMID: 15496913
  27. Base-calling of automated sequencer traces using phred. I. Accuracy assessment.
    Genome Res. 1998 Mar;8(3):175-85 PMID: 9521921
  28. The molecular mechanism for the genetic disorder familial defective apolipoprotein B100.
    J Biol Chem. 2001 Mar 23;276(12):9214-8 PMID: 11115503
  29. Mechanisms of disease: genetic causes of familial hypercholesterolemia.
    Nat Clin Pract Cardiovasc Med. 2007 Apr;4(4):214-25 PMID: 17380167
  30. Disruption of LDL but not VLDL clearance in autosomal recessive hypercholesterolemia.
    J Clin Invest. 2007 Jan;117(1):165-74 PMID: 17200716
  31. Newly identified loci that influence lipid concentrations and risk of coronary artery disease.
    Nat Genet. 2008 Feb;40(2):161-9 PMID: 18193043
  32. Genetic architecture of inter-individual variability in apolipoprotein, lipoprotein and lipid phenotypes.
    Ciba Found Symp. 1987;130:99-127 PMID: 3327665
  33. LRP6 mutation in a family with early coronary disease and metabolic risk factors.
    Science. 2007 Mar 2;315(5816):1278-82 PMID: 17332414
Article Info
Journal
Genome research
Abbr.
Genome Res
ISSN
1549-5469
Published
2009-09-00
Epub
2009-00-14
Pages
1665-74
Language
English
Region
United States
NLM ID
9518021
PMCID
PMC2752125
Subset
IM
Grants
Intramural NIH HHS · United States
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