Genetic investigation of 100 heart genes in sudden unexplained death victims in a forensic setting

被引:56
作者
Christiansen, Sofie Lindgren [1 ]
Hertz, Christin Loth [1 ]
Ferrero-Miliani, Laura [1 ]
Dahl, Morten [2 ,7 ]
Weeke, Peter Ejvin [3 ]
LuCamp [4 ]
Ottesen, Gyda Lolk [5 ]
Frank-Hansen, Rune [1 ]
Bundgaard, Henning [6 ]
Morling, Niels [1 ]
机构
[1] Univ Copenhagen, Fac Hlth & Med Sci, Sect Forens Genet, Dept Forens Med, Frederik Vs Vej 11, DK-2100 Copenhagen, Denmark
[2] Univ Copenhagen, Copenhagen Univ Hosp, Rigshosp, Dept Clin Biochem, Copenhagen, Denmark
[3] Copenhagen Univ Hosp, Rigshosp, Mol Cardiol Lab, Dept Cardiol, Copenhagen, Denmark
[4] Lundbeck Fdn Ctr Appl Med Genom Personalized Dis, LuCamp, Copenhagen, Denmark
[5] Univ Copenhagen, Fac Hlth & Med Sci, Dept Forens Med, Sect Forens Pathol, Copenhagen, Denmark
[6] Copenhagen Univ Hosp, Rigshosp, Ctr Heart, Unit Inherited Cardiac Dis, Copenhagen, Denmark
[7] Univ Copenhagen, Copenhagen Univ Hosp, Dept Clin Biochem, Copenhagen, Denmark
关键词
UNEXPECTED DEATH; MOLECULAR AUTOPSY; SODIUM CURRENT; CARDIAC DEATH; MUTATIONS; RISK; VARIANTS; EPILEPSY; PLAKOPHILIN-2; NATIONWIDE;
D O I
10.1038/ejhg.2016.118
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
In forensic medicine, one-third of the sudden deaths remain unexplained after medico-legal autopsy. A major proportion of these sudden unexplained deaths (SUD) are considered to be caused by inherited cardiac diseases. Sudden cardiac death (SCD) may be the first manifestation of these diseases. The purpose of this study was to explore the yield of next-generation sequencing of genes associated with SCD in a cohort of SUD victims. We investigated 100 genes associated with cardiac diseases in 61 young (1-50 years) SUD cases. DNA was captured with the Haloplex target enrichment system and sequenced using an Illumina MiSeq. The identified genetic variants were evaluated and classified as likely, unknown or unlikely to have a functional effect. The criteria for this classification were based on the literature, databases, conservation and prediction of the effect of the variant. We found that 21 (34%) individuals carried variants with a likely functional effect. Ten (40%) of these variants were located in genes associated with cardiomyopathies and 15 (60%) of the variants in genes associated with cardiac channelopathies. Nineteen individuals carried variants with unknown functional effect. Our findings indicate that broad genetic investigation of SUD victims increases the diagnostic outcome, and the investigation should comprise genes involved in both cardiomyopathies and cardiac channelopathies.
引用
收藏
页码:1797 / 1802
页数:6
相关论文
共 53 条
  • [1] New population-based exome data are questioning the pathogenicity of previously cardiomyopathy-associated genetic variants
    Andreasen, Charlotte
    Nielsen, Jonas B.
    Refsgaard, Lena
    Holst, Anders G.
    Christensen, Alex H.
    Andreasen, Laura
    Sajadieh, Ahmad
    Haunso, Stig
    Svendsen, Jesper H.
    Olesen, Morten S.
    [J]. EUROPEAN JOURNAL OF HUMAN GENETICS, 2013, 21 (09) : 918 - 928
  • [2] Exome analysis-based molecular autopsy in cases of sudden unexplained death in the young
    Bagnall, Richard D.
    Das, Jipin
    Duflou, Johan
    Semsarian, Christopher
    [J]. HEART RHYTHM, 2014, 11 (04) : 655 - 662
  • [3] Cardiac and Autonomic Mechanisms Contributing to SUDEP
    Bermeo-Ovalle, Adriana C.
    Kennedy, Jeffrey D.
    Schuele, Stephan U.
    [J]. JOURNAL OF CLINICAL NEUROPHYSIOLOGY, 2015, 32 (01) : 21 - 29
  • [4] Sarcomeric gene mutations in sudden infant death syndrome (SIDS)
    Brion, Maria
    Allegue, Catarina
    Santori, Montserrat
    Gil, Rocio
    Blanco-Verea, Alejandro
    Haas, Cordula
    Bartsch, Christine
    Poster, Simone
    Madea, Burkhard
    Campuzano, Oscar
    Brugada, Ramon
    Carracedo, Angel
    [J]. FORENSIC SCIENCE INTERNATIONAL, 2012, 219 (1-3) : 278 - 281
  • [5] Mutations in the cardiac L-type calcium channel associated with inherited J-wave syndromes and sudden cardiac death
    Burashnikov, Elena
    Pfeiffer, Ryan
    Barajas-Martinez, Hector
    Delpon, Eva
    Hu, Dan
    Desai, Mayurika
    Borggrefe, Martin
    Haeissaguerre, Michel
    Kanter, Ronald
    Pollevick, Guido D.
    Guerchicoff, Alejandra
    Laino, Ruben
    Marieb, Mark
    Nademanee, Koonlawee
    Nam, Gi-Byoung
    Robles, Roberto
    Schimpf, Rainer
    Stapleton, Dwight D.
    Viskin, Sami
    Winters, Stephen
    Wolpert, Christian
    Zimmern, Samuel
    Veltmann, Christian
    Antzelevitch, Charles
    [J]. HEART RHYTHM, 2010, 7 (12) : 1872 - 1882
  • [6] Missense Mutations in Plakophilin-2 Cause Sodium Current Deficit and Associate With a Brugada Syndrome Phenotype
    Cerrone, Marina
    Lin, Xianming
    Zhang, Mingliang
    Agullo-Pascual, Esperanza
    Pfenniger, Anna
    Gusky, Halina Chkourko
    Novelli, Valeria
    Kim, Changsung
    Tirasawadichai, Tiara
    Judge, Daniel P.
    Rothenberg, Eli
    Chen, Huei-Sheng Vincent
    Napolitano, Carlo
    Priori, Silvia G.
    Delmar, Mario
    [J]. CIRCULATION, 2014, 129 (10) : 1092 - U88
  • [7] Missense Variants in Plakophilin-2 in Arrhythmogenic Right Ventricular Cardiomyopathy Patients - Disease-Causing or Innocent Bystanders?
    Christensen, Alex Horby
    Benn, Marianne
    Tybjaerg-Hansen, Anne
    Haunso, Stig
    Svendsen, Jesper Hastrup
    [J]. CARDIOLOGY, 2010, 115 (02) : 148 - 154
  • [8] Interpreting Secondary Cardiac Disease Variants in an Exome Cohort
    David Ng
    Johnston, Jennifer J.
    Teer, Jamie K.
    Singh, Larry N.
    Peller, Lindsey C.
    Wynter, Jamila S.
    Lewis, Katie L.
    Cooper, David N.
    Stenson, Peter D.
    Mullikin, James C.
    Biesecker, Leslie G.
    [J]. CIRCULATION-CARDIOVASCULAR GENETICS, 2013, 6 (04): : 337 - 346
  • [9] Actionable, Pathogenic Incidental Findings in 1,000 Participants' Exomes
    Dorschner, Michael O.
    Amendola, Laura M.
    Turner, Emily H.
    Robertson, Peggy D.
    Shirts, Brian H.
    Gallego, Carlos J.
    Bennett, Robin L.
    Jones, Kelly L.
    Tokita, Mari J.
    Bennett, James T.
    Kim, Jerry H.
    Rosenthal, Elisabeth A.
    Kim, Daniel S.
    Tabor, Holly K.
    Bamshad, Michael J.
    Motulsky, Arno G.
    Scott, C. Ronald
    Pritchard, Colin C.
    Walsh, Tom
    Burke, Wylie
    Raskind, Wendy H.
    Byers, Peter
    Hisama, Fuld M.
    Nickerson, Deborah A.
    Jarvik, Gail P.
    [J]. AMERICAN JOURNAL OF HUMAN GENETICS, 2013, 93 (04) : 631 - 640
  • [10] Risk of arrhythmia induced by psychotropic medications: a proposal for clinical management
    Fanoe, Soren
    Kristensen, Diana
    Fink-Jensen, Anders
    Jensen, Henrik Kjaerulf
    Toft, Egon
    Nielsen, Jimmi
    Videbech, Poul
    Pehrson, Steen
    Bundgaard, Henning
    [J]. EUROPEAN HEART JOURNAL, 2014, 35 (20) : 1306 - 1315B