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PMID: 20220176 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Analysis of genetic inheritance in a family quartet by whole-genome sequencing.

Science (New York, N.Y.) ·Vol. 328 ·No. 5978 ·2010-04-30 ·Pages 636-9

Roach JC, Glusman G, Smit AF, Huff CD, Hubley R, Shannon PT, Rowen L, Pant KP, Goodman N, Bamshad M, Shendure J, Drmanac R, Jorde LB, Hood L, Galas DJ

Abstract

We analyzed the whole-genome sequences of a family of four, consisting of two siblings and their parents. Family-based sequencing allowed us to delineate recombination sites precisely, identify 70% of the sequencing errors (resulting in > 99.999% accuracy), and identify very rare single-nucleotide polymorphisms. We also directly estimated a human intergeneration mutation rate of approximately 1.1 x 10(-8) per position per haploid genome. Both offspring in this family have two recessive disorders: Miller syndrome, for which the gene was concurrently identified, and primary ciliary dyskinesia, for which causative genes have been previously identified. Family-based genome analysis enabled us to narrow the candidate genes for both of these Mendelian disorders to only four. Our results demonstrate the value of complete genome sequencing in families.

MeSH Terms
Abnormalities, Multiple/genetics Algorithms Alleles Axonemal Dyneins/genetics Ciliary Motility Disorders/genetics Crossing Over, Genetic Dihydroorotate Dehydrogenase Female Genes, Dominant Genes, Recessive Genetic Association Studies Genome, Human Humans Inheritance Patterns Limb Deformities, Congenital/genetics Male Mandibulofacial Dysostosis/genetics Mutation Nuclear Family Oxidoreductases Acting on CH-CH Group Donors/genetics Pedigree Polymorphism, Single Nucleotide Sequence Analysis, DNA Syndrome
Chemicals
Dihydroorotate Dehydrogenase Oxidoreductases Acting on CH-CH Group Donors Axonemal Dyneins DNAH5 protein, human
Authors & Affiliations
15 authors, click to expand affiliations / ORCID
Roach Jared C
Institute for Systems Biology, Seattle, WA 98103, USA.
Glusman Gustavo
Smit Arian F A
Huff Chad D
Hubley Robert
Shannon Paul T
Rowen Lee
Pant Krishna P
Goodman Nathan
Bamshad Michael
Shendure Jay
Drmanac Radoje
Jorde Lynn B
Hood Leroy
Galas David J
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Article Info
Journal
Science (New York, N.Y.)
Abbr.
Science
ISSN
1095-9203
Published
2010-04-30
Epub
2010-00-10
Pages
636-9
Language
English
Region
United States
NLM ID
0404511
PMCID
PMC3037280
Subset
IM
Grants
NIGMS NIH HHS · GM076547 · United States
NHGRI NIH HHS · R01 HG002939-08 · United States
NIGMS NIH HHS · R01GM081083 · United States
NHGRI NIH HHS · RC2 HG005608 · United States
NHLBI NIH HHS · R01 HL094976 · United States
NHGRI NIH HHS · RZ1HG004749 · United States
NICHD NIH HHS · R01HD048895 · United States
NHGRI NIH HHS · RC2HG005608 · United States
NIGMS NIH HHS · P50 GM076547-05 · United States
NHGRI NIH HHS · R01 HG002939 · United States
NHGRI NIH HHS · R21 HG004749 · United States
NHLBI NIH HHS · R01HL094976 · United States
NIGMS NIH HHS · P50 GM076547 · United States
NICHD NIH HHS · R01 HD048895 · United States
NIGMS NIH HHS · R01 GM081083 · United States
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