- Dewey, Frederick E;
- Chen, Rong;
- Cordero, Sergio P;
- Ormond, Kelly E;
- Caleshu, Colleen;
- Karczewski, Konrad J;
- Whirl-Carrillo, Michelle;
- Wheeler, Matthew T;
- Dudley, Joel T;
- Byrnes, Jake K;
- Cornejo, Omar E;
- Knowles, Joshua W;
- Woon, Mark;
- Sangkuhl, Katrin;
- Gong, Li;
- Thorn, Caroline F;
- Hebert, Joan M;
- Capriotti, Emidio;
- David, Sean P;
- Pavlovic, Aleksandra;
- West, Anne;
- Thakuria, Joseph V;
- Ball, Madeleine P;
- Zaranek, Alexander W;
- Rehm, Heidi L;
- Church, George M;
- West, John S;
- Bustamante, Carlos D;
- Snyder, Michael;
- Altman, Russ B;
- Klein, Teri E;
- Butte, Atul J;
- Ashley, Euan A
- Editor(s): Copenhaver, Gregory P
Whole-genome sequencing harbors unprecedented potential for characterization of individual and family genetic variation. Here, we develop a novel synthetic human reference sequence that is ethnically concordant and use it for the analysis of genomes from a nuclear family with history of familial thrombophilia. We demonstrate that the use of the major allele reference sequence results in improved genotype accuracy for disease-associated variant loci. We infer recombination sites to the lowest median resolution demonstrated to date (< 1,000 base pairs). We use family inheritance state analysis to control sequencing error and inform family-wide haplotype phasing, allowing quantification of genome-wide compound heterozygosity. We develop a sequence-based methodology for Human Leukocyte Antigen typing that contributes to disease risk prediction. Finally, we advance methods for analysis of disease and pharmacogenomic risk across the coding and non-coding genome that incorporate phased variant data. We show these methods are capable of identifying multigenic risk for inherited thrombophilia and informing the appropriate pharmacological therapy. These ethnicity-specific, family-based approaches to interpretation of genetic variation are emblematic of the next generation of genetic risk assessment using whole-genome sequencing.