Disclosure of genetic research results to members of a founder population.
There is currently extensive discussion and debate in the literature on how, when, and to whom genetic research results should be returned (see Genetics in Medicine, April 2012 issue). Here, we describe our experience in disclosing genetic information on Mendelian disorders discovered during the course of our research in the Hutterites. We first assessed attitudes toward the disclosure of carrier results, which revealed that many individuals wanted carrier information and that many intended to use the information in family planning. Based on this information, we developed a pilot study to test and disclose cystic fibrosis (CF) carrier status. Next, a larger scale project was developed in order to disclose genetic research results for 14 diseases to those interested in receiving the information. We developed brochures, offered a live interactive educational program, conducted a consent process, and disclosed results in letters mailed to the consented individuals. Overall, ~80% of individuals who participated in the educational program signed consent forms for the release of their results for 14 diseases. We describe our experience with returning individual genetic research results to participants in a population-based research study.
Department of Human Genetics, University of Chicago, 5841 S. Maryland Ave. M/C 0077, Chicago, IL, 60637, USA.
Rebecca L
RL
Anderson
Kathleen
K
Murray
Jessica X
JX
Chong
Rebecca
R
Ouwenga
Marina
M
Antillon
Peixian
P
Chen
Lorena
L
Diaz de Leon
Kathryn J
KJ
Swoboda
Lucille A
LA
Lester
Soma
S
Das
Carole
C
Ober
Darrel J
DJ
Waggoner
D032921:Consensus; D003550:Cystic Fibrosis / Q000175:diagnosis* / Q000235:genetics*; D030881:Disclosure; D005260:Female; D018703:Founder Effect; D006580:Genetic Carrier Screening; D005817:Genetic Counseling / Q000592:standards*; D036281:Genetic Research; D005820:Genetic Testing / Q000592:standards*; D006801:Humans; D008297:Male; D010353:Patient Education as Topic; D010865:Pilot Projects