Janet Malek1, Stacey Pereira2, Jill O Robinson2, Amanda M Gutierrez2, Melody J Slashinski3, D Williams Parsons4,5, Sharon E Plon4,5, Amy L McGuire2. 1. Center for Medical Ethics and Health Policy, Baylor College of Medicine, Houston, TX, USA. janet.malek@bcm.edu. 2. Center for Medical Ethics and Health Policy, Baylor College of Medicine, Houston, TX, USA. 3. School of Public Health & Health Sciences, University of Massachusetts, Amherst, MA, USA. 4. Department of Pediatrics, Baylor College of Medicine, Houston, TX, USA. 5. Texas Children's Cancer Center, Texas Children's Hospital, Houston, TX, USA.
Abstract
PURPOSE: We describe parental perceptions of and experiences with genomic sequencing (GS) in the care of seriously ill children. Understanding parents' perspectives is vital for clinicians caring for children, given the uptake of genomic technologies into clinical practice. METHODS: Longitudinal, semistructured interviews were conducted with parents of pediatric cancer patients who underwent exome sequencing (ES) as a part of the BASIC3 study. Interviews were conducted at baseline, one to eight months after results disclosure, and approximately one year after disclosure. Using thematic qualitative analysis, parent interviews were coded with both inductive and deductive approaches. RESULTS: Before receiving genomic information, parents indicated that they saw ES as something responsible parents would agree to if their child had cancer. Some parents talked about the possibility of sequencing affecting feelings of culpability for their child's cancer, worrying that they passed on a cancer-causing gene or made parenting decisions that caused the disease. However, after receiving their child's ES results many reported feeling relieved of guilt and worry, and felt they had fulfilled parental duties by agreeing to ES for their child. CONCLUSION: These results reveal a layer of meaning that parents associate with GS that may inform clinicians' approach to care.
PURPOSE: We describe parental perceptions of and experiences with genomic sequencing (GS) in the care of seriously ill children. Understanding parents' perspectives is vital for clinicians caring for children, given the uptake of genomic technologies into clinical practice. METHODS: Longitudinal, semistructured interviews were conducted with parents of pediatric cancer patients who underwent exome sequencing (ES) as a part of the BASIC3 study. Interviews were conducted at baseline, one to eight months after results disclosure, and approximately one year after disclosure. Using thematic qualitative analysis, parent interviews were coded with both inductive and deductive approaches. RESULTS: Before receiving genomic information, parents indicated that they saw ES as something responsible parents would agree to if their child had cancer. Some parents talked about the possibility of sequencing affecting feelings of culpability for their child's cancer, worrying that they passed on a cancer-causing gene or made parenting decisions that caused the disease. However, after receiving their child's ES results many reported feeling relieved of guilt and worry, and felt they had fulfilled parental duties by agreeing to ES for their child. CONCLUSION: These results reveal a layer of meaning that parents associate with GS that may inform clinicians' approach to care.
Authors: J A Anderson; M S Meyn; C Shuman; R Zlotnik Shaul; L E Mantella; M J Szego; S Bowdin; N Monfared; R Z Hayeems Journal: J Med Ethics Date: 2016-11-25 Impact factor: 2.903
Authors: Holly K Tabor; Tracy Brazg; Julia Crouch; Emily E Namey; Stephanie M Fullerton; Laura M Beskow; Benjamin S Wilfond Journal: J Empir Res Hum Res Ethics Date: 2011-12 Impact factor: 1.742
Authors: Vasiliki Rahimzadeh; Christoph Schickhardt; Bartha M Knoppers; Karine Sénécal; Danya F Vears; Conrad V Fernandez; Stefan Pfister; Sharon Plon; Sharon Terry; Janet Williams; Marc S Williams; Martina Cornel; Jan M Friedman Journal: JAMA Pediatr Date: 2018-05-01 Impact factor: 16.193
Authors: Laurence B McCullough; Melody J Slashinski; Amy L McGuire; Richard L Street; Christine M Eng; Richard A Gibbs; D William Parsons; Sharon E Plon Journal: Pediatr Blood Cancer Date: 2015-10-27 Impact factor: 3.167
Authors: Lotte Krabbenborg; L E L M Vissers; J Schieving; T Kleefstra; E J Kamsteeg; J A Veltman; M A Willemsen; S Van der Burg Journal: J Genet Couns Date: 2016-04-20 Impact factor: 2.537
Authors: Hadley Stevens Smith; Kyle B Brothers; Sara J Knight; Sara L Ackerman; Christine Rini; David L Veenstra; Amy L McGuire; Benjamin S Wilfond; Janet Malek Journal: Am J Hum Genet Date: 2021-10-22 Impact factor: 11.025
Authors: Julie A Cakici; David P Dimmock; Sara A Caylor; Mary Gaughran; Christina Clarke; Cynthia Triplett; Michelle M Clark; Stephen F Kingsmore; Cinnamon S Bloss Journal: Am J Hum Genet Date: 2020-11-05 Impact factor: 11.025
Authors: Kandamurugu Manickam; Monica R McClain; Laurie A Demmer; Sawona Biswas; Hutton M Kearney; Jennifer Malinowski; Lauren J Massingham; Danny Miller; Timothy W Yu; Fuki M Hisama Journal: Genet Med Date: 2021-07-01 Impact factor: 8.822
Authors: Katherine E Donohue; Siobhan M Dolan; Dana Watnick; Katie M Gallagher; Jacqueline A Odgis; Sabrina A Suckiel; Nehama Teitelman; Bruce D Gelb; Eimear E Kenny; Melissa P Wasserstein; Carol R Horowitz; Laurie J Bauman Journal: Patient Educ Couns Date: 2021-01-29
Authors: Amanda M Gutierrez; Jill O Robinson; Simon M Outram; Hadley S Smith; Stephanie A Kraft; Katherine E Donohue; Barbara B Biesecker; Kyle B Brothers; Flavia Chen; Benyam Hailu; Lucia A Hindorff; Hannah Hoban; Rebecca L Hsu; Sara J Knight; Barbara A Koenig; Katie L Lewis; Kristen Hassmiller Lich; Julianne M O'Daniel; Sonia Okuyama; Gail E Tomlinson; Margaret Waltz; Benjamin S Wilfond; Sara L Ackerman; Mary A Majumder Journal: J Clin Transl Sci Date: 2021-09-14
Authors: Simon M Outram; Julia E H Brown; Astrid N Zamora; Nuriye Sahin-Hodoglugil; Sara L Ackerman Journal: Front Genet Date: 2022-07-18 Impact factor: 4.772
Authors: Samantha Pollard; Deirdre Weymann; Jessica Dunne; Fatemeh Mayanloo; John Buckell; James Buchanan; Sarah Wordsworth; Jan M Friedman; Sylvia Stockler-Ipsiroglu; Nick Dragojlovic; Alison M Elliott; Mark Harrison; Larry D Lynd; Dean A Regier Journal: Eur J Hum Genet Date: 2021-04-26 Impact factor: 4.246