Literature DB >> 32299846

Pilot Study of Return of Genetic Results to Patients in Adult Nephrology.

Jordan G Nestor1, Maddalena Marasa1, Hila Milo-Rasouly1, Emily E Groopman1, S Ali Husain1, Sumit Mohan1,2, Hilda Fernandez1, Vimla S Aggarwal3, Dina F Ahram1, Natalie Vena1,4, Kelsie Bogyo1,3, Andrew S Bomback1, Jai Radhakrishnan1, Gerald B Appel1, Wooin Ahn1, David J Cohen1, Pietro A Canetta1, Geoffrey K Dube1, Maya K Rao1, Heather K Morris1, Russell J Crew1, Simone Sanna-Cherchi1, Krzysztof Kiryluk1, Ali G Gharavi5,4.   

Abstract

BACKGROUND AND OBJECTIVES: Actionable genetic findings have implications for care of patients with kidney disease, and genetic testing is an emerging tool in nephrology practice. However, there are scarce data regarding best practices for return of results and clinical application of actionable genetic findings for kidney patients. DESIGN, SETTING, PARTICIPANTS, & MEASUREMENTS: We developed a return of results workflow in collaborations with clinicians for the retrospective recontact of adult nephrology patients who had been recruited into a biobank research study for exome sequencing and were identified to have medically actionable genetic findings.
RESULTS: Using this workflow, we attempted to recontact a diverse pilot cohort of 104 nephrology research participants with actionable genetic findings, encompassing 34 different monogenic etiologies of nephropathy and five single-gene disorders recommended by the American College of Medical Genetics and Genomics for return as medically actionable secondary findings. We successfully recontacted 64 (62%) participants and returned results to 41 (39%) individuals. In each case, the genetic diagnosis had meaningful implications for the patients' nephrology care. Through implementation efforts and qualitative interviews with providers, we identified over 20 key challenges associated with returning results to study participants, and found that physician knowledge gaps in genomics was a recurrent theme. We iteratively addressed these challenges to yield an optimized workflow, which included standardized consultation notes with tailored management recommendations, monthly educational conferences on core topics in genomics, and a curated list of expert clinicians for patients requiring extranephrologic referrals.
CONCLUSIONS: Developing the infrastructure to support return of genetic results in nephrology was resource-intensive, but presented potential opportunities for improving patient care. PODCAST: This article contains a podcast at https://www.asn-online.org/media/podcast/CJASN/2020_04_16_12481019.mp3.
Copyright © 2020 by the American Society of Nephrology.

Entities:  

Keywords:  adult; biological specimen banks; chronic kidney disease; cohort studies; exome; familial nephropathy; genetic renal disease; genetic testing; genomics; human genetics; humans; kidney diseases; medical genetics; nephrology; patient care; pilot projects; referral and consultation; retrospective studies; whole exome sequencing; workflow

Mesh:

Year:  2020        PMID: 32299846      PMCID: PMC7269209          DOI: 10.2215/CJN.12481019

Source DB:  PubMed          Journal:  Clin J Am Soc Nephrol        ISSN: 1555-9041            Impact factor:   10.614


  51 in total

1.  Research electronic data capture (REDCap)--a metadata-driven methodology and workflow process for providing translational research informatics support.

Authors:  Paul A Harris; Robert Taylor; Robert Thielke; Jonathon Payne; Nathaniel Gonzalez; Jose G Conde
Journal:  J Biomed Inform       Date:  2008-09-30       Impact factor: 6.317

2.  Whole-Exome Sequencing Enables a Precision Medicine Approach for Kidney Transplant Recipients.

Authors:  Nina Mann; Daniela A Braun; Kassaundra Amann; Weizhen Tan; Shirlee Shril; Dervla M Connaughton; Makiko Nakayama; Ronen Schneider; Thomas M Kitzler; Amelie T van der Ven; Jing Chen; Hadas Ityel; Asaf Vivante; Amar J Majmundar; Ankana Daga; Jillian K Warejko; Svjetlana Lovric; Shazia Ashraf; Tilman Jobst-Schwan; Eugen Widmeier; Hannah Hugo; Shrikant M Mane; Leslie Spaneas; Michael J G Somers; Michael A Ferguson; Avram Z Traum; Deborah R Stein; Michelle A Baum; Ghaleb H Daouk; Richard P Lifton; Shannon Manzi; Khashayar Vakili; Heung Bae Kim; Nancy M Rodig; Friedhelm Hildebrandt
Journal:  J Am Soc Nephrol       Date:  2019-01-17       Impact factor: 10.121

3.  Common Elements in Rare Kidney Diseases: Conclusions from a Kidney Disease: Improving Global Outcomes (KDIGO) Controversies Conference.

Authors:  Ségolène Aymé; Detlef Bockenhauer; Simon Day; Olivier Devuyst; Lisa M Guay-Woodford; Julie R Ingelfinger; Jon B Klein; Nine V A M Knoers; Ronald D Perrone; Julia Roberts; Franz Schaefer; Vicente E Torres; Michael Cheung; David C Wheeler; Wolfgang C Winkelmayer
Journal:  Kidney Int       Date:  2017-10       Impact factor: 10.612

4.  Reanalysis of Clinical Exome Sequencing Data.

Authors:  Pengfei Liu; Linyan Meng; Elizabeth A Normand; Fan Xia; Xiaofei Song; Andrew Ghazi; Jill Rosenfeld; Pilar L Magoulas; Alicia Braxton; Patricia Ward; Hongzheng Dai; Bo Yuan; Weimin Bi; Rui Xiao; Xia Wang; Theodore Chiang; Francesco Vetrini; Weimin He; Hanyin Cheng; Jie Dong; Charul Gijavanekar; Paul J Benke; Jonathan A Bernstein; Tanya Eble; Yasemen Eroglu; Deanna Erwin; Luis Escobar; James B Gibson; Karen Gripp; Soledad Kleppe; Mary K Koenig; Andrea M Lewis; Marvin Natowicz; Pedro Mancias; LaKeesha Minor; Fernando Scaglia; Christian P Schaaf; Haley Streff; Hilary Vernon; Crescenda L Uhles; Elaine H Zackai; Nan Wu; V Reid Sutton; Arthur L Beaudet; Donna Muzny; Richard A Gibbs; Jennifer E Posey; Seema Lalani; Chad Shaw; Christine M Eng; James R Lupski; Yaping Yang
Journal:  N Engl J Med       Date:  2019-06-20       Impact factor: 91.245

5.  Return of genomic results to research participants: the floor, the ceiling, and the choices in between.

Authors:  Gail P Jarvik; Laura M Amendola; Jonathan S Berg; Kyle Brothers; Ellen W Clayton; Wendy Chung; Barbara J Evans; James P Evans; Stephanie M Fullerton; Carlos J Gallego; Nanibaa' A Garrison; Stacy W Gray; Ingrid A Holm; Iftikhar J Kullo; Lisa Soleymani Lehmann; Cathy McCarty; Cynthia A Prows; Heidi L Rehm; Richard R Sharp; Joseph Salama; Saskia Sanderson; Sara L Van Driest; Marc S Williams; Susan M Wolf; Wendy A Wolf; Wylie Burke
Journal:  Am J Hum Genet       Date:  2014-05-08       Impact factor: 11.025

6.  A survey-based evaluation of self-perceived competency after nephrology fellowship training.

Authors:  Jeffrey S Berns
Journal:  Clin J Am Soc Nephrol       Date:  2010-01-14       Impact factor: 8.237

7.  Deep Phenotyping on Electronic Health Records Facilitates Genetic Diagnosis by Clinical Exomes.

Authors:  Jung Hoon Son; Gangcai Xie; Chi Yuan; Lyudmila Ena; Ziran Li; Andrew Goldstein; Lulin Huang; Liwei Wang; Feichen Shen; Hongfang Liu; Karla Mehl; Emily E Groopman; Maddalena Marasa; Krzysztof Kiryluk; Ali G Gharavi; Wendy K Chung; George Hripcsak; Carol Friedman; Chunhua Weng; Kai Wang
Journal:  Am J Hum Genet       Date:  2018-06-28       Impact factor: 11.025

Review 8.  Expert consensus guidelines for the genetic diagnosis of Alport syndrome.

Authors:  Judy Savige; Francesca Ariani; Francesca Mari; Mirella Bruttini; Alessandra Renieri; Oliver Gross; Constantinos Deltas; Frances Flinter; Jie Ding; Daniel P Gale; Mato Nagel; Michael Yau; Lev Shagam; Roser Torra; Elisabet Ars; Julia Hoefele; Guido Garosi; Helen Storey
Journal:  Pediatr Nephrol       Date:  2018-07-09       Impact factor: 3.714

9.  Recommendations for reporting of secondary findings in clinical exome and genome sequencing, 2016 update (ACMG SF v2.0): a policy statement of the American College of Medical Genetics and Genomics.

Authors:  Sarah S Kalia; Kathy Adelman; Sherri J Bale; Wendy K Chung; Christine Eng; James P Evans; Gail E Herman; Sophia B Hufnagel; Teri E Klein; Bruce R Korf; Kent D McKelvey; Kelly E Ormond; C Sue Richards; Christopher N Vlangos; Michael Watson; Christa L Martin; David T Miller
Journal:  Genet Med       Date:  2016-11-17       Impact factor: 8.822

10.  The Burden of Candidate Pathogenic Variants for Kidney and Genitourinary Disorders Emerging From Exome Sequencing.

Authors:  Hila Milo Rasouly; Emily E Groopman; Reuben Heyman-Kantor; David A Fasel; Adele Mitrotti; Rik Westland; Louise Bier; Chunhua Weng; Zhong Ren; Brett Copeland; Priya Krithivasan; Wendy K Chung; Simone Sanna-Cherchi; David B Goldstein; Ali G Gharavi
Journal:  Ann Intern Med       Date:  2018-11-27       Impact factor: 51.598

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  9 in total

1.  Clinical Genetic Screening in Adult Patients with Kidney Disease.

Authors:  Enrico Cocchi; Jordan Gabriela Nestor; Ali G Gharavi
Journal:  Clin J Am Soc Nephrol       Date:  2020-07-09       Impact factor: 8.237

2.  Evaluation of Genetic Kidney Diseases in Living Donor Kidney Transplantation: Towards Precision Genomic Medicine in Donor Risk Assessment.

Authors:  Yasar Caliskan; Brian Lee; Adrian Whelan; Fadee Abualrub; Krista L Lentine; Arksarapuk Jittirat
Journal:  Curr Transplant Rep       Date:  2022-03-16

3.  Impact of education on APOL1 testing attitudes among prospective living kidney donors.

Authors:  Jordan G Nestor; Amber J Li; Kristen L King; S Ali Husain; Tristan J McIntosh; Deirdre Sawinski; Ana S Iltis; Melody S Goodman; Heidi A Walsh; James M DuBois; Sumit Mohan
Journal:  Clin Transplant       Date:  2021-10-28       Impact factor: 3.456

4.  A systematic literature review of disclosure practices and reported outcomes for medically actionable genomic secondary findings.

Authors:  Julie C Sapp; Flavia M Facio; Diane Cooper; Katie L Lewis; Emily Modlin; Philip van der Wees; Leslie G Biesecker
Journal:  Genet Med       Date:  2021-08-26       Impact factor: 8.864

5.  Disclosure of clinically actionable genetic variants to thoracic aortic dissection biobank participants.

Authors:  Adelyn Beil; Whitney Hornsby; Cristen Willer; J Scott Roberts; Wendy R Uhlmann; Rajani Aatre; Patricia Arscott; Brooke Wolford; Kim A Eagle; Bo Yang; Jennifer McNamara
Journal:  BMC Med Genomics       Date:  2021-03-01       Impact factor: 3.063

6.  Genomics Integration Into Nephrology Practice.

Authors:  Filippo Pinto E Vairo; Carri Prochnow; Jennifer L Kemppainen; Emily C Lisi; Joan M Steyermark; Teresa M Kruisselbrink; Pavel N Pichurin; Rhadika Dhamija; Megan M Hager; Sam Albadri; Lynn D Cornell; Konstantinos N Lazaridis; Eric W Klee; Sarah R Senum; Mireille El Ters; Hatem Amer; Linnea M Baudhuin; Ann M Moyer; Mira T Keddis; Ladan Zand; David J Sas; Stephen B Erickson; Fernando C Fervenza; John C Lieske; Peter C Harris; Marie C Hogan
Journal:  Kidney Med       Date:  2021-06-29

7.  Mainstreaming Genetic Testing for Adult Patients With Autosomal Dominant Polycystic Kidney Disease.

Authors:  Mark D Elliott; Leslie C James; Emily L Simms; Priyana Sharma; Louis P Girard; Kim Cheema; Meghan J Elliott; Julie L Lauzon; Justin Chun
Journal:  Can J Kidney Health Dis       Date:  2021-10-29

8.  Assessing Physician Needs for the Implementation of Personalized Care.

Authors:  Jordan G Nestor
Journal:  Kidney Int Rep       Date:  2020-12-17

9.  Clinical impact of genomic testing in patients with suspected monogenic kidney disease.

Authors:  Andrew J Mallett; Catherine Quinlan; Kushani Jayasinghe; Zornitza Stark; Peter G Kerr; Clara Gaff; Melissa Martyn; John Whitlam; Belinda Creighton; Elizabeth Donaldson; Matthew Hunter; Anna Jarmolowicz; Lilian Johnstone; Emma Krzesinski; Sebastian Lunke; Elly Lynch; Kathleen Nicholls; Chirag Patel; Yael Prawer; Jessica Ryan; Emily J See; Andrew Talbot; Alison Trainer; Rigan Tytherleigh; Giulia Valente; Mathew Wallis; Louise Wardrop; Kirsty H West; Susan M White; Ella Wilkins
Journal:  Genet Med       Date:  2020-09-17       Impact factor: 8.822

  9 in total

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