PURPOSE: To explore how parents of pediatric cancer patients perceived the utility of clinical tumor and germline whole-exome sequencing (WES) results. PATIENTS AND METHODS: We conducted longitudinal interviews with parents of a diverse pediatric cancer population before disclosure of WES results (n=64), then one to eight months (n=33) after disclosure. Interview transcripts were analyzed using a thematic qualitative approach. RESULTS: Parents identified a broad range of types of utility for their child's WES results. Even when results did not affect their child's current treatment, they expressed optimism about future clinical utility for their child, themselves, and other family members. Parents also reported experiencing psychological utility including peace of mind, relief of guilt, and satisfaction of curiosity. Pragmatic utility, such as the ability to plan for the future and make better reproductive decisions, was also described. CONCLUSION: Parents of pediatric cancer patients perceive WES to have broad utility, including psychological and pragmatic utility, even if there is no direct impact on clinical care. Further work will need to consider how the value of genomic information should be characterized, how risks and benefits should be described, and how these results should inform recommendations and decisions about using WES.
PURPOSE: To explore how parents of pediatric cancer patients perceived the utility of clinical tumor and germline whole-exome sequencing (WES) results. PATIENTS AND METHODS: We conducted longitudinal interviews with parents of a diverse pediatric cancer population before disclosure of WES results (n=64), then one to eight months (n=33) after disclosure. Interview transcripts were analyzed using a thematic qualitative approach. RESULTS: Parents identified a broad range of types of utility for their child's WES results. Even when results did not affect their child's current treatment, they expressed optimism about future clinical utility for their child, themselves, and other family members. Parents also reported experiencing psychological utility including peace of mind, relief of guilt, and satisfaction of curiosity. Pragmatic utility, such as the ability to plan for the future and make better reproductive decisions, was also described. CONCLUSION: Parents of pediatric cancer patients perceive WES to have broad utility, including psychological and pragmatic utility, even if there is no direct impact on clinical care. Further work will need to consider how the value of genomic information should be characterized, how risks and benefits should be described, and how these results should inform recommendations and decisions about using WES.
Authors: Marian Reiff; Ellen Giarelli; Barbara A Bernhardt; Ebony Easley; Nancy B Spinner; Pamela L Sankar; Surabhi Mulchandani Journal: J Autism Dev Disord Date: 2015-10
Authors: Jeffrey R Botkin; John W Belmont; Jonathan S Berg; Benjamin E Berkman; Yvonne Bombard; Ingrid A Holm; Howard P Levy; Kelly E Ormond; Howard M Saal; Nancy B Spinner; Benjamin S Wilfond; Joseph D McInerney Journal: Am J Hum Genet Date: 2015-07-02 Impact factor: 11.025
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: Susan E Waisbren; Danielle K Bäck; Christina Liu; Sarah S Kalia; Steven A Ringer; Ingrid A Holm; Robert C Green Journal: Genet Med Date: 2014-12-04 Impact factor: 8.822
Authors: D Williams Parsons; Angshumoy Roy; Yaping Yang; Tao Wang; Sarah Scollon; Katie Bergstrom; Robin A Kerstein; Stephanie Gutierrez; Andrea K Petersen; Abhishek Bavle; Frank Y Lin; Dolores H López-Terrada; Federico A Monzon; M John Hicks; Karen W Eldin; Norma M Quintanilla; Adekunle M Adesina; Carrie A Mohila; William Whitehead; Andrew Jea; Sanjeev A Vasudevan; Jed G Nuchtern; Uma Ramamurthy; Amy L McGuire; Susan G Hilsenbeck; Jeffrey G Reid; Donna M Muzny; David A Wheeler; Stacey L Berg; Murali M Chintagumpala; Christine M Eng; Richard A Gibbs; Sharon E Plon Journal: JAMA Oncol Date: 2016-05-01 Impact factor: 31.777
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: Hadley Stevens Smith; Stephanie R Morain; Jill Oliver Robinson; Isabel Canfield; Janet Malek; Caryn Kseniya Rubanovich; Cinnamon S Bloss; Sara L Ackerman; Barbara Biesecker; Kyle B Brothers; Crispin N Goytia; Carol R Horowitz; Sara J Knight; Barbara Koenig; Stephanie A Kraft; Simon Outram; Christine Rini; Kelly J Shipman; Margaret Waltz; Benjamin Wilfond; Amy L McGuire Journal: Patient Date: 2021-10-18 Impact factor: 3.481
Authors: Jill O Robinson; Julia Wynn; Barbara Biesecker; Leslie G Biesecker; Barbara Bernhardt; Kyle B Brothers; Wendy K Chung; Kurt D Christensen; Robert C Green; Amy L McGuire; M Ragan Hart; Ida Griesemer; Donald L Patrick; Christine Rini; David Veenstra; Angel M Cronin; Stacy W Gray Journal: Genet Med Date: 2019-06-13 Impact factor: 8.822
Authors: Janet Malek; Stacey Pereira; Jill O Robinson; Amanda M Gutierrez; Melody J Slashinski; D Williams Parsons; Sharon E Plon; Amy L McGuire Journal: Genet Med Date: 2019-06-12 Impact factor: 8.864
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: Katianne M Howard Sharp; Niki Jurbergs; Annastasia Ouma; Lynn Harrison; Elsie Gerhardt; Leslie Taylor; Kayla Hamilton; Rose B McGee; Regina Nuccio; Emily Quinn; Stacy Hines-Dowell; Chimene Kesserwan; Anusha Sunkara; Jami S Gattuso; Michelle Pritchard; Belinda Mandrell; Mary V Relling; Cyrine E Haidar; Guolian Kang; Liza M Johnson; Kim E Nichols Journal: JCO Precis Oncol Date: 2020-03-24