Literature DB >> 33754291

Preconception expanded carrier screening: a focus group study with relatives of mucopolysaccharidosis type III patients and the general population.

Thirsa Conijn1,2, Ivy van Dijke3,4, Lotte Haverman2, Phillis Lakeman5, Frits A Wijburg1, Lidewij Henneman6.   

Abstract

Preconception expanded carrier screening (ECS) enables prospective parents to assess their risk of having a child with an autosomal recessive disorder. Knowledge on motivations, feelings, and considerations people have towards the offer and use of ECS is limited. To enrich the public and professional discussion on ECS implementation, this study explored the perspectives towards various aspects of ECS in seven focus groups compromising first- and second-degree relatives of MPS III patients (N=9, N=4, N=5, N=5) and members of the general Dutch population (N=6, N=7, N=5). The focus groups were audio recorded and the transcripts were qualitatively analyzed to identify themes. Both relatives of MPS III patients and participants from the general population supported offering ECS, in particular for severe, childhood-onset disorders. Important barriers identified for ECS were a lack of genetic knowledge and a perceived lack of personal relevance and awareness, as well as out-of-pocket costs of testing. The majority of participants would prefer full disclosure of individual test results instead of couple-based test results. Moreover, offering people a choice for the way of reporting was proposed. All participants agreed that more efforts, for example by governmental campaigns, should be made to increase awareness on the availability, potentials, and limitations of ECS. Educating prospective parents about ECS is essential for increasing awareness and informed decision making. This study provides valuable insights that can be used by governments and public health authorities when considering implementation of preconception ECS.

Entities:  

Keywords:  Expanded carrier screening; Focus groups; Genetic screening; Mucopolysaccharidosis type III; Perspectives; Qualitative study

Year:  2021        PMID: 33754291     DOI: 10.1007/s12687-021-00519-2

Source DB:  PubMed          Journal:  J Community Genet        ISSN: 1868-310X


  31 in total

1.  Preconceptional couple screening for cystic fibrosis carrier status: couples prefer full disclosure of test results.

Authors:  L Henneman; L P Ten Kate
Journal:  J Med Genet       Date:  2002-05       Impact factor: 6.318

2.  Expanded carrier screening in reproductive medicine-points to consider: a joint statement of the American College of Medical Genetics and Genomics, American College of Obstetricians and Gynecologists, National Society of Genetic Counselors, Perinatal Quality Foundation, and Society for Maternal-Fetal Medicine.

Authors:  Janice G Edwards; Gerald Feldman; James Goldberg; Anthony R Gregg; Mary E Norton; Nancy C Rose; Adele Schneider; Katie Stoll; Ronald Wapner; Michael S Watson
Journal:  Obstet Gynecol       Date:  2015-03       Impact factor: 7.661

Review 3.  Expanded carrier screening for monogenic disorders: where are we now?

Authors:  Davit Chokoshvili; Danya Vears; Pascal Borry
Journal:  Prenat Diagn       Date:  2017-07-27       Impact factor: 3.050

4.  Reasons for Declining Preconception Expanded Carrier Screening Using Genome Sequencing.

Authors:  Marian J Gilmore; Jennifer Schneider; James V Davis; Tia L Kauffman; Michael C Leo; Kellene Bergen; Jacob A Reiss; Patricia Himes; Elissa Morris; Carol Young; Carmit McMullen; Benjamin S Wilfond; Katrina A B Goddard
Journal:  J Genet Couns       Date:  2017-03-17       Impact factor: 2.537

Review 5.  International perspectives on the implementation of reproductive carrier screening.

Authors:  Martin B Delatycki; Fowzan Alkuraya; Alison Archibald; Carlo Castellani; Martina Cornel; Wayne W Grody; Lidewij Henneman; Adonis S Ioannides; Edwin Kirk; Nigel Laing; Anneke Lucassen; John Massie; Juliette Schuurmans; Meow-Keong Thong; Irene van Langen; Joël Zlotogora
Journal:  Prenat Diagn       Date:  2019-11-29       Impact factor: 3.050

6.  Population screening for spinal muscular atrophy: A mixed methods study of the views of affected families.

Authors:  Felicity K Boardman; Philip J Young; Frances E Griffiths
Journal:  Am J Med Genet A       Date:  2016-10-28       Impact factor: 2.802

7.  Arrays in postnatal and prenatal diagnosis: An exploration of the ethics of consent.

Authors:  Wybo Dondorp; Birgit Sikkema-Raddatz; Christine de Die-Smulders; Guido de Wert
Journal:  Hum Mutat       Date:  2012-04-04       Impact factor: 4.878

Review 8.  Carrier screening for recessive disorders.

Authors:  Stylianos E Antonarakis
Journal:  Nat Rev Genet       Date:  2019-09       Impact factor: 53.242

9.  Public Views on Genetics and Genetic Testing: A Survey of the General Public in Belgium.

Authors:  Davit Chokoshvili; Carmen Belmans; Roxanne Poncelet; Sofie Sanders; Deborah Vaes; Danya Vears; Sandra Janssens; Isabelle Huys; Pascal Borry
Journal:  Genet Test Mol Biomarkers       Date:  2017-03

10.  "It's something I need to consider": decisions about carrier screening for fragile X syndrome in a population of non-pregnant women.

Authors:  Alison D Archibald; Alice M Jaques; Samantha Wake; Veronica R Collins; Jonathan Cohen; Sylvia A Metcalfe
Journal:  Am J Med Genet A       Date:  2009-12       Impact factor: 2.802

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  1 in total

Review 1.  Societal implications of expanded universal carrier screening: a scoping review.

Authors:  Lieke M van den Heuvel; Nina van den Berg; A Cecile J W Janssens; Erwin Birnie; Lidewij Henneman; Wybo J Dondorp; Mirjam Plantinga; Irene M van Langen
Journal:  Eur J Hum Genet       Date:  2022-09-12       Impact factor: 5.351

  1 in total

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