Literature DB >> 31019283

Secondary actionable findings identified by exome sequencing: expected impact on the organisation of care from the study of 700 consecutive tests.

Christel Thauvin-Robinet1,2,3,4, Julien Thevenon5,6,7,8, Sophie Nambot6,7, Julian Delanne7, Paul Kuentz5,6, Ange-Line Bruel5,6,8, Aline Chassagne5,9, Elodie Cretin5,9, Aurore Pelissier5,10, Chritine Peyron5,10, Elodie Gautier5, Daphné Lehalle6,7, Nolwenn Jean-Marçais7, Patrick Callier5,6,8, Anne-Laure Mosca-Boidron5,6,8, Antonio Vitobello5,6,8, Arthur Sorlin5,6,7,8, Frédéric Tran Mau-Them5,6,8, Christophe Philippe5,6,8, Pierre Vabres5,6, Laurent Demougeot11, Charlotte Poé6, Thibaud Jouan6, Martin Chevarin6, Mathilde Lefebvre6,7, Marc Bardou12, Emilie Tisserant6, Maxime Luu12, Christine Binquet12, Jean-François Deleuze13, Céline Verstuyft14, Yannis Duffourd5,6, Laurence Faivre15,16,17,18.   

Abstract

With exome/genome sequencing (ES/GS) integrated into the practice of medicine, there is some potential for reporting incidental/secondary findings (IFs/SFs). The issue of IFs/SFs has been studied extensively over the last 4 years. In order to evaluate their implications in care organisation, we retrospectively evaluated, in a cohort of 700 consecutive probands, the frequency and burden of introducing the search for variants in a maximum list of 244 medically actionable genes (genes that predispose carriers to a preventable or treatable disease in childhood/adulthood and genes for genetic counselling issues). We also focused on the 59 PharmGKB class IA/IB pharmacogenetic variants. We also compared the results in different gene lists. We identified variants (likely) affecting protein function in genes for care in 26 cases (3.7%) and heterozygous variants in genes for genetic counselling in 29 cases (3.8%). Mean time for the 700 patients was about 6.3 min/patient for medically actionable genes and 1.3 min/patient for genes for genetic counselling, and a mean time of 37 min/patients for the reinterpreted variants. These results would lead to all 700 pre-test counselling sessions being longer, to 55 post-test genetic consultations and to 27 secondary specialised medical evaluations. ES also detected 42/59 pharmacogenetic variants or combinations of variants in the majority of cases. An extremely low metabolizer status in genes relevant for neurodevelopmental disorders (CYP2C9 and CYP2C19) was found in 57/700 cases. This study provides information regarding the need to anticipate the implementation of genomic medicine, notably the work overload at various steps of the process.

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Year:  2019        PMID: 31019283      PMCID: PMC6777608          DOI: 10.1038/s41431-019-0384-7

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  39 in total

Review 1.  Whole-Exome Sequencing and Whole-Genome Sequencing in Critically Ill Neonates Suspected to Have Single-Gene Disorders.

Authors:  Laurie D Smith; Laurel K Willig; Stephen F Kingsmore
Journal:  Cold Spring Harb Perspect Med       Date:  2015-12-18       Impact factor: 6.915

2.  Whole-genome sequencing in health care. Recommendations of the European Society of Human Genetics.

Authors:  Carla G van El; Martina C Cornel; Pascal Borry; Ros J Hastings; Florence Fellmann; Shirley V Hodgson; Heidi C Howard; Anne Cambon-Thomsen; Bartha M Knoppers; Hanne Meijers-Heijboer; Hans Scheffer; Lisbeth Tranebjaerg; Wybo Dondorp; Guido M W R de Wert
Journal:  Eur J Hum Genet       Date:  2013-06       Impact factor: 4.246

3.  Performance of ACMG-AMP Variant-Interpretation Guidelines among Nine Laboratories in the Clinical Sequencing Exploratory Research Consortium.

Authors:  Laura M Amendola; Gail P Jarvik; Michael C Leo; Heather M McLaughlin; Yassmine Akkari; Michelle D Amaral; Jonathan S Berg; Sawona Biswas; Kevin M Bowling; Laura K Conlin; Greg M Cooper; Michael O Dorschner; Matthew C Dulik; Arezou A Ghazani; Rajarshi Ghosh; Robert C Green; Ragan Hart; Carrie Horton; Jennifer J Johnston; Matthew S Lebo; Aleksandar Milosavljevic; Jeffrey Ou; Christine M Pak; Ronak Y Patel; Sumit Punj; Carolyn Sue Richards; Joseph Salama; Natasha T Strande; Yaping Yang; Sharon E Plon; Leslie G Biesecker; Heidi L Rehm
Journal:  Am J Hum Genet       Date:  2016-05-12       Impact factor: 11.025

4.  Integrating Pharmacogenomics into Clinical Practice: Promise vs Reality.

Authors:  Jennifer L St Sauver; Suzette J Bielinski; Janet E Olson; Elizabeth J Bell; Michaela E Mc Gree; Debra J Jacobson; Jennifer B McCormick; Pedro J Caraballo; Paul Y Takahashi; Veronique L Roger; Carolyn R Rohrer Vitek
Journal:  Am J Med       Date:  2016-05-05       Impact factor: 4.965

5.  Effectiveness of exome and genome sequencing guided by acuity of illness for diagnosis of neurodevelopmental disorders.

Authors:  Sarah E Soden; Carol J Saunders; Laurel K Willig; Emily G Farrow; Laurie D Smith; Josh E Petrikin; Jean-Baptiste LePichon; Neil A Miller; Isabelle Thiffault; Darrell L Dinwiddie; Greyson Twist; Aaron Noll; Bryce A Heese; Lee Zellmer; Andrea M Atherton; Ahmed T Abdelmoity; Nicole Safina; Sarah S Nyp; Britton Zuccarelli; Ingrid A Larson; Ann Modrcin; Suzanne Herd; Mitchell Creed; Zhaohui Ye; Xuan Yuan; Robert A Brodsky; Stephen F Kingsmore
Journal:  Sci Transl Med       Date:  2014-12-03       Impact factor: 17.956

6.  The very low penetrance of cystic fibrosis for the R117H mutation: a reappraisal for genetic counselling and newborn screening.

Authors:  C Thauvin-Robinet; A Munck; F Huet; E Génin; G Bellis; E Gautier; M-P Audrézet; C Férec; G Lalau; M Des Georges; M Claustres; T Bienvenu; B Gérard; P Boisseau; F Cabet-Bey; D Feldmann; C Clavel; E Bieth; A Iron; B Simon-Bouy; C Costa; R Medina; J Leclerc; D Hubert; R Nové-Josserand; I Sermet-Gaudelus; G Rault; J Flori; S Leroy; N Wizla; G Bellon; A Haloun; S Perez-Martin; G d'Acremont; H Corvol; A Clément; E Houssin; C Binquet; C Bonithon-Kopp; C Alberti-Boulmé; M A Morris; L Faivre; M Goossens; M Roussey; E Girodon
Journal:  J Med Genet       Date:  2009-06-29       Impact factor: 6.318

Review 7.  Clinical Pharmacogenetics Implementation Consortium guidelines for cytochrome P450 2D6 genotype and codeine therapy: 2014 update.

Authors:  K R Crews; A Gaedigk; H M Dunnenberger; J S Leeder; T E Klein; K E Caudle; C E Haidar; D D Shen; J T Callaghan; S Sadhasivam; C A Prows; E D Kharasch; T C Skaar
Journal:  Clin Pharmacol Ther       Date:  2014-01-23       Impact factor: 6.875

8.  Actionable exomic incidental findings in 6503 participants: challenges of variant classification.

Authors:  Laura M Amendola; Michael O Dorschner; Peggy D Robertson; Joseph S Salama; Ragan Hart; Brian H Shirts; Mitzi L Murray; Mari J Tokita; Carlos J Gallego; Daniel Seung Kim; James T Bennett; David R Crosslin; Jane Ranchalis; Kelly L Jones; Elisabeth A Rosenthal; Ella R Jarvik; Andy Itsara; Emily H Turner; Daniel S Herman; Jennifer Schleit; Amber Burt; Seema M Jamal; Jenica L Abrudan; Andrew D Johnson; Laura K Conlin; Matthew C Dulik; Avni Santani; Danielle R Metterville; Melissa Kelly; Ann Katherine M Foreman; Kristy Lee; Kent D Taylor; Xiuqing Guo; Kristy Crooks; Lesli A Kiedrowski; Leslie J Raffel; Ora Gordon; Kalotina Machini; Robert J Desnick; Leslie G Biesecker; Steven A Lubitz; Surabhi Mulchandani; Greg M Cooper; Steven Joffe; C Sue Richards; Yaoping Yang; Jerome I Rotter; Stephen S Rich; Christopher J O'Donnell; Jonathan S Berg; Nancy B Spinner; James P Evans; Stephanie M Fullerton; Kathleen A Leppig; Robin L Bennett; Thomas Bird; Virginia P Sybert; William M Grady; Holly K Tabor; Jerry H Kim; Michael J Bamshad; Benjamin Wilfond; Arno G Motulsky; C Ronald Scott; Colin C Pritchard; Tom D Walsh; Wylie Burke; Wendy H Raskind; Peter Byers; Fuki M Hisama; Heidi Rehm; Debbie A Nickerson; Gail P Jarvik
Journal:  Genome Res       Date:  2015-01-30       Impact factor: 9.043

9.  Patient and interest organizations' views on personalized medicine: a qualitative study.

Authors:  Isabelle Budin-Ljøsne; Jennifer R Harris
Journal:  BMC Med Ethics       Date:  2016-05-13       Impact factor: 2.652

10.  ACMG recommendations for reporting of incidental findings in clinical exome and genome sequencing.

Authors:  Robert C Green; Jonathan S Berg; Wayne W Grody; Sarah S Kalia; Bruce R Korf; Christa L Martin; Amy L McGuire; Robert L Nussbaum; Julianne M O'Daniel; Kelly E Ormond; Heidi L Rehm; Michael S Watson; Marc S Williams; Leslie G Biesecker
Journal:  Genet Med       Date:  2013-06-20       Impact factor: 8.822

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  3 in total

1.  An accessible insight into genetic findings for transplantation recipients with suspected genetic kidney disease.

Authors:  Zhigang Wang; Hongen Xu; Tianchao Xiang; Danhua Liu; Fei Xu; Lixiang Zhao; Yonghua Feng; Linan Xu; Jialu Liu; Ye Fang; Huanfei Liu; Ruijun Li; Xinxin Hu; Jingyuan Guan; Longshan Liu; Guiwen Feng; Qian Shen; Hong Xu; Dmitrij Frishman; Wenxue Tang; Jiancheng Guo; Jia Rao; Wenjun Shang
Journal:  NPJ Genom Med       Date:  2021-07-02       Impact factor: 8.617

2.  Critical assessment of secondary findings in genes linked to primary arrhythmia syndromes.

Authors:  Isabel Diebold; Ulrike Schön; Florentine Scharf; Anna Benet-Pagès; Andreas Laner; Elke Holinski-Feder; Angela Abicht
Journal:  Hum Mutat       Date:  2020-02-18       Impact factor: 4.878

3.  Old Challenges or New Issues? Genetic Health Professionals' Experiences Obtaining Informed Consent in Diagnostic Genomic Sequencing.

Authors:  Danya F Vears; Pascal Borry; Julian Savulescu; Julian J Koplin
Journal:  AJOB Empir Bioeth       Date:  2020-10-05
  3 in total

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