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Our aim was to explore participating parents\u2019 attitudes towards and understanding of genome sequencing in this hybrid context. We conducted in-depth telephone interviews with 20 parents of children with rare diseases participating in the 100,000 Genomes Project. Parents were positive about contributing to research, although some had needed reassurance about data protections. Although most felt positive about secondary findings, some could not recall or misunderstood key aspects. Some were also concerned about potential emotional impact of results and a few raised concerns about life insurance implications, and the impact of future legal changes. Participants were generally positive about consent appointments, but several raised concerns about \u2018information overload\u2019 because of deciding about secondary findings at the same time as about the main diagnostic genome sequencing and data contribution. Additional information resources, particularly online tools, were highlighted as potentially useful ways of supporting the consent process. We conclude that parents offered genome sequencing as part of a national hybrid clinical and research project report many positive attitudes and experiences, but also concerns and misunderstandings. Further research is needed on how best to support informed consent, particularly about secondary findings. Additional resources such as online tools might usefully support future genome sequencing consent processes.<\/jats:p>","DOI":"10.1038\/s41431-020-0575-2","type":"journal-article","created":{"date-parts":[[2020,1,30]],"date-time":"2020-01-30T09:03:59Z","timestamp":1580375039000},"page":"874-884","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":38,"title":["Parents\u2019 motivations, concerns and understanding of genome sequencing: a qualitative interview study"],"prefix":"10.1038","volume":"28","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7169-1521","authenticated-orcid":false,"given":"Celine","family":"Lewis","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Saskia","family":"Sanderson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3900-1425","authenticated-orcid":false,"given":"Melissa","family":"Hill","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4191-0663","authenticated-orcid":false,"given":"Chris","family":"Patch","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Beverly","family":"Searle","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amy","family":"Hunter","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lyn S.","family":"Chitty","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2020,1,30]]},"reference":[{"key":"575_CR1","doi-asserted-by":"publisher","first-page":"253","DOI":"10.1038\/nrg.2017.116","volume":"19","author":"CF Wright","year":"2018","unstructured":"Wright CF, FitzPatrick DR, Firth HV. 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None of the other authors have any conflicts of interest to declare. CL is currently funded by a Health\u00a0Education England Fellowship and has developed decision support aids for children undergoing genome sequencing.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}]}}