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Robust identification of deletions in exome and genome sequence data based on clustering of Mendelian errors.
Manheimer, Kathryn B; Patel, Nihir; Richter, Felix; Gorham, Joshua; Tai, Angela C; Homsy, Jason; Boskovski, Marko T; Parfenov, Michael; Goldmuntz, Elizabeth; Chung, Wendy K; Brueckner, Martina; Tristani-Firouzi, Martin; Srivastava, Deepak; Seidman, Jonathan G; Seidman, Christine E; Gelb, Bruce D; Sharp, Andrew J.
Afiliación
  • Manheimer KB; Mindich Child Health and Development Institute, Icahn School of Medicine at Mount Sinai, New York, New York.
  • Patel N; Mindich Child Health and Development Institute, Icahn School of Medicine at Mount Sinai, New York, New York.
  • Richter F; Mindich Child Health and Development Institute, Icahn School of Medicine at Mount Sinai, New York, New York.
  • Gorham J; Department of Genetics, Harvard Medical School, Boston, Massachusetts.
  • Tai AC; Department of Genetics, Harvard Medical School, Boston, Massachusetts.
  • Homsy J; Department of Genetics, Harvard Medical School, Boston, Massachusetts.
  • Boskovski MT; Cardiovascular Research Center, Massachusetts General Hospital, Boston, Massachusetts.
  • Parfenov M; Division of Cardiac Surgery, The Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts.
  • Goldmuntz E; Department of Genetics, Harvard Medical School, Boston, Massachusetts.
  • Chung WK; Department of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania.
  • Brueckner M; Division of Cardiology, The Children's Hospital of Philadelphia, The University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania.
  • Tristani-Firouzi M; Department of Pediatrics, Columbia University Medical Center, New York, New York.
  • Srivastava D; Department of Medicine, Columbia University Medical Center, New York, New York.
  • Seidman JG; Genetics, Yale University School of Medicine, New Haven, Connecticut.
  • Seidman CE; Pediatrics, Yale University School of Medicine, New Haven, Connecticut.
  • Gelb BD; Department of Pediatric Cardiology, University of Utah, Salt Lake City, Utah.
  • Sharp AJ; Department of Pediatrics, UCSF, San Francisco, California.
Hum Mutat ; 39(6): 870-881, 2018 06.
Article en En | MEDLINE | ID: mdl-29527824
Multiple tools have been developed to identify copy number variants (CNVs) from whole exome (WES) and whole genome sequencing (WGS) data. Current tools such as XHMM for WES and CNVnator for WGS identify CNVs based on changes in read depth. For WGS, other methods to identify CNVs include utilizing discordant read pairs and split reads and genome-wide local assembly with tools such as Lumpy and SvABA, respectively. Here, we introduce a new method to identify deletion CNVs from WES and WGS trio data based on the clustering of Mendelian errors (MEs). Using our Mendelian Error Method (MEM), we identified 127 deletions (inherited and de novo) in 2,601 WES trios from the Pediatric Cardiac Genomics Consortium, with a validation rate of 88% by digital droplet PCR. MEM identified additional de novo deletions compared with XHMM, and a significant enrichment of 15q11.2 deletions compared with controls. In addition, MEM identified eight cases of uniparental disomy, sample switches, and DNA contamination. We applied MEM to WGS data from the Genome In A Bottle Ashkenazi trio and identified deletions with 97% specificity. MEM provides a robust, computationally inexpensive method for identifying deletions, and an orthogonal approach for verifying deletions called by other tools.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Análisis Mutacional de ADN / Genoma Humano / Eliminación de Secuencia / Variaciones en el Número de Copia de ADN Tipo de estudio: Diagnostic_studies Límite: Female / Humans / Male Idioma: En Revista: Hum Mutat Asunto de la revista: GENETICA MEDICA Año: 2018 Tipo del documento: Article Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Análisis Mutacional de ADN / Genoma Humano / Eliminación de Secuencia / Variaciones en el Número de Copia de ADN Tipo de estudio: Diagnostic_studies Límite: Female / Humans / Male Idioma: En Revista: Hum Mutat Asunto de la revista: GENETICA MEDICA Año: 2018 Tipo del documento: Article Pais de publicación: Estados Unidos