TY - JOUR
T1 - Population screening shows risk of inherited cancer and familial hypercholesterolemia in Oregon
AU - O'Brien, Timothy D.
AU - Potter, Amiee B.
AU - Driscoll, Catherine C.
AU - Goh, Gregory
AU - Letaw, John H.
AU - McCabe, Sarah
AU - Thanner, Jane
AU - Kulkarni, Arpita
AU - Wong, Rossana
AU - Medica, Samuel
AU - Week, Tiana
AU - Buitrago, Jacob
AU - Larson, Aaron
AU - Camacho, Katie Johnson
AU - Brown, Kim
AU - Crist, Rachel
AU - Conrad, Casey
AU - Evans-Dutson, Sara
AU - Lutz, Ryan
AU - Mitchell, Asia
AU - Anur, Pavana
AU - Serrato, Vanessa
AU - Shafer, Autumn
AU - Marriott, Lisa K.
AU - Hamman, K. J.
AU - Mulford, Amelia
AU - Wiszniewski, Wojciech
AU - Sampson, Jone E.
AU - Adey, Andrew
AU - O'Roak, Brian J.
AU - Harrington, Christina A.
AU - Shannon, Jackilen
AU - Spellman, Paul T.
AU - Richards, C. Sue
N1 - Publisher Copyright:
© 2023 American Society of Human Genetics
PY - 2023/8/3
Y1 - 2023/8/3
N2 - The Healthy Oregon Project (HOP) is a statewide effort that aims to build a large research repository and influence the health of Oregonians through providing no-cost genetic screening to participants for a next-generation sequencing 32-gene panel comprising genes related to inherited cancers and familial hypercholesterolemia. This type of unbiased population screening can detect at-risk individuals who may otherwise be missed by conventional medical approaches. However, challenges exist for this type of high-throughput testing in an academic setting, including developing a low-cost high-efficiency test and scaling up the clinical laboratory for processing large numbers of samples. Modifications to our academic clinical laboratory including efficient test design, robotics, and a streamlined analysis approach increased our ability to test more than 1,000 samples per month for HOP using only one dedicated HOP laboratory technologist. Additionally, enrollment using a HIPAA-compliant smartphone app and sample collection using mouthwash increased efficiency and reduced cost. Here, we present our experience three years into HOP and discuss the lessons learned, including our successes, challenges, opportunities, and future directions, as well as the genetic screening results for the first 13,670 participants tested. Overall, we have identified 730 pathogenic/likely pathogenic variants in 710 participants in 24 of the 32 genes on the panel. The carrier rate for pathogenic/likely pathogenic variants in the inherited cancer genes on the panel for an unselected population was 5.0% and for familial hypercholesterolemia was 0.3%. Our laboratory experience described here may provide a useful model for population screening projects in other states.
AB - The Healthy Oregon Project (HOP) is a statewide effort that aims to build a large research repository and influence the health of Oregonians through providing no-cost genetic screening to participants for a next-generation sequencing 32-gene panel comprising genes related to inherited cancers and familial hypercholesterolemia. This type of unbiased population screening can detect at-risk individuals who may otherwise be missed by conventional medical approaches. However, challenges exist for this type of high-throughput testing in an academic setting, including developing a low-cost high-efficiency test and scaling up the clinical laboratory for processing large numbers of samples. Modifications to our academic clinical laboratory including efficient test design, robotics, and a streamlined analysis approach increased our ability to test more than 1,000 samples per month for HOP using only one dedicated HOP laboratory technologist. Additionally, enrollment using a HIPAA-compliant smartphone app and sample collection using mouthwash increased efficiency and reduced cost. Here, we present our experience three years into HOP and discuss the lessons learned, including our successes, challenges, opportunities, and future directions, as well as the genetic screening results for the first 13,670 participants tested. Overall, we have identified 730 pathogenic/likely pathogenic variants in 710 participants in 24 of the 32 genes on the panel. The carrier rate for pathogenic/likely pathogenic variants in the inherited cancer genes on the panel for an unselected population was 5.0% and for familial hypercholesterolemia was 0.3%. Our laboratory experience described here may provide a useful model for population screening projects in other states.
KW - clinical genetic testing
KW - familial hypercholesterolemia
KW - inherited cancer
KW - laboratory genetics
KW - population screening
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U2 - 10.1016/j.ajhg.2023.06.014
DO - 10.1016/j.ajhg.2023.06.014
M3 - Article
C2 - 37506692
AN - SCOPUS:85166579416
SN - 0002-9297
VL - 110
SP - 1249
EP - 1265
JO - American Journal of Human Genetics
JF - American Journal of Human Genetics
IS - 8
ER -