A human genome structural variation sequencing resource reveals insights into mutational mechanisms

Jeffrey M. Kidd, Tina Graves, Tera L. Newman, Robert Fulton, Hillary S. Hayden, Maika Malig, Joelle Kallicki, Rajinder Kaul, Richard K. Wilson, Evan E. Eichler

Research output: Contribution to journalArticlepeer-review

208 Scopus citations

Abstract

Understanding the prevailing mutational mechanisms responsible for human genome structural variation requires uniformity in the discovery of allelic variants and precision in terms of breakpoint delineation. We develop a resource based on capillary end sequencing of 13.8 million fosmid clones from 17 human genomes and characterize the complete sequence of 1054 large structural variants corresponding to 589 deletions, 384 insertions, and 81 inversions. We analyze the 2081 breakpoint junctions and infer potential mechanism of origin. Three mechanisms account for the bulk of germline structural variation: microhomology-mediated processes involving short (2-20 bp) stretches of sequence (28%), nonallelic homologous recombination (22%), and L1 retrotransposition (19%). The high quality and long-range continuity of the sequence reveals more complex mutational mechanisms, including repeat-mediated inversions and gene conversion, that are most often missed by other methods, such as comparative genomic hybridization, single nucleotide polymorphism microarrays, and next-generation sequencing.

Original languageEnglish
Pages (from-to)837-847
Number of pages11
JournalCell
Volume143
Issue number5
DOIs
StatePublished - Nov 24 2010

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