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Record W1843990841 · doi:10.1126/science.1261877

Effect of predicted protein-truncating genetic variants on the human transcriptome

2015· article· en· W1843990841 on OpenAlexfundno aff
Manuel A. Rivas, Matti Pirinen, Donald F. Conrad, Monkol Lek, Emily K. Tsang, Konrad J. Karczewski, Julian Maller, Kimberly R. Kukurba, David S. DeLuca, Menachem Fromer, Pedro G. Ferreira, Kevin S. Smith, Rui Zhang, Fengmei Zhao, Eric Banks, Ryan Poplin, Douglas M. Ruderfer, Shaun Purcell, Taru Tukiainen, Eric Vallabh Minikel, Peter D. Stenson, D.N. Cooper, Katharine H. Huang, Timothy J. Sullivan, Jared L. Nedzel, Carlos D. Bustamante, Jin Billy Li, Mark J. Daly, Roderic Guigó, Peter Donnelly, Kristin Ardlie, Michael Sammeth, Emmanouil T. Dermitzakis, Mark I. McCarthy, Stephen B. Montgomery, Tuuli Lappalainen, Daniel G. MacArthur, Ayellet V. Segrè, Taylor Young, Ellen Gelfand, Casandra A. Trowbridge, Lucas D. Ward, Pouya Kheradpour, Benjamin Iriarte, Yan Meng, Cameron D. Palmer, Tõnu Esko, Wendy Winckler, Joel N. Hirschhorn, Manolis Kellis, Gad Getz, Andrey A. Shablin, Gen Li, Yi‐Hui Zhou, Andrew B. Nobel, Ivan Rusyn, Fred A. Wright, Alexis Battle, Sara Mostafavi, Marta Melé, Ferrán Reverter, Jakob M. Goldmann, Daphne Koller, Eric R. Gamazon, Hae Kyung Im, Anuar Konkashbaev, Dan L. Nicolae, Nancy J. Cox, Timothe Flutre, Xiaoquan Wen, Matthew Stephens, Jonathan K. Pritchard, Zhidong Tu, Bin Zhang, Tao Huang, Quan Long, Luan Lin, Jialiang Yang, Jun Zhu, Jun S. Liu, Amanda Brown, Bernadette Mestichelli, Denee Tidwell, Edmund Lo, Mike Salvatore, Saboor Shad, Jeffrey A. Thomas, John T. Lonsdale, Roswell Christopher Choi, Ellen Karasik, Kimberly Ramsey, Michael T. Moser, Barbara A. Foster, Bryan M. Gillard, John Syron, Johnelle Fleming, Harold I. Magazine, Rick Hasz, Gary Walters, Jason Bridge, Mark Miklos, Susan Sullivan, Laura K. Barker, Heather M. Traino, Magboeba Mosavel, Laura A. Siminoff, Dana R. Valley, Daniel C. Rohrer, Scott Jewel, Philip A. Branton, Leslie H. Sobin, Mary E. Barcus, Liqun Qi, Pushpa Hariharan, Shenpei Wu, David E. Tabor, Charles Shive, Anna M. Smith, Stephen A. Buia, Anita H. Undale, Karna Robinson, Nancy Roche, Kimberly M. Valentino, Angela Britton, Robin Burges, Debra Bradbury, Kenneth W. Hambright, John Seleski, Greg E. Korzeniewski, Kenyon Erickson, Yvonne Marcus, Jorge Tejada, Mehran Taherian, Chunrong Lu, Barnaby E. Robles, Margaret J. Basile, Deborah C. Mash, Simona Volpi, Jeffery P. Struewing, Gary F. Temple, Joy T Boyer, Deborah Colantuoni, Susan E. Koester, Latarsha J. Carithers, Helen M. Moore, Ping Guan, Carolyn C. Compton, Sherilyn J. Sawyer, Joanne P. Demchok, Jimmie B. Vaught, Chana A. Rabiner, Nicole C. Lockhart, Marc R. Friedländer, Peter A.C. ’t Hoen, Jean Monlong, Mar Gonzàlez-Porta, Natalja Kurbatova, Thasso Griebel, Matthias Barann, Thomas Wieland, Liliana Greger, Maarten van Iterson, Jonas Carlsson Almlöf, Paolo Ribeca, Irina Pulyakhina, Daniela Esser, Thomas Giger, Andrew Tikhonov, Marc Sultan, Gabrielle Bertier, Esther Lizano, Henk P.J. Buermans, Ismaël Padioleau, Thomas Schwarzmayr, Olof Karlberg, Halit Ongen, Helena Kilpinen, Sergi Beltrán, Katja Kahlem, Vyacheslav Amstislavskiy, Oliver Stegle, Paul Flicek, Tim M. Strom, Hans Lehrach, Stefan Schreiber, Ralf Sudbrak, Ángel Carracedo, Stylianos E. Antonarakis, Robert Häsler, Ann‐Christine Syvänen, Gert‐Jan B. van Ommen, Alvis Brāzma, Thomas Meitinger, Philip Rosenstiel, Marta Gut, Xavier Estivill

Bibliographic record

VenueScience · 2015
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenomics and Chromatin Dynamics
Canadian institutionsnot available
FundersU.S. National Library of MedicineNational Institute of General Medical SciencesNational Cancer InstituteNational Institute on Drug AbuseNational Institute of Diabetes and Digestive and Kidney DiseasesBiotechnology and Biological Sciences Research CouncilNatural Sciences and Engineering Research Council of CanadaSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungNational Institutes of HealthNational Institute of Mental HealthNational Heart, Lung, and Blood InstituteU.S. Department of DefenseLouis-Jeantet FoundationAcademy of FinlandH2020 European Research CouncilUniversity of OxfordNational Human Genome Research InstituteWellcome TrustWellcome
KeywordsBiologyGenetic variationGeneGeneticsTranscriptomeGene expressionExpression quantitative trait lociGenotypeRegulation of gene expressionProtein expressionPhenotypeSingle-nucleotide polymorphism

Abstract

fetched live from OpenAlex

Accurate prediction of the functional effect of genetic variation is critical for clinical genome interpretation. We systematically characterized the transcriptome effects of protein-truncating variants, a class of variants expected to have profound effects on gene function, using data from the Genotype-Tissue Expression (GTEx) and Geuvadis projects. We quantitated tissue-specific and positional effects on nonsense-mediated transcript decay and present an improved predictive model for this decay. We directly measured the effect of variants both proximal and distal to splice junctions. Furthermore, we found that robustness to heterozygous gene inactivation is not due to dosage compensation. Our results illustrate the value of transcriptome data in the functional interpretation of genetic variants.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.002
metaresearch head score (Gemma)0.002
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.002
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0020.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0000.001
Insufficient payload (model declined to judge)0.0010.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.009
GPT teacher head0.247
Teacher spread0.238 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations345
Published2015
Admission routes1
Has abstractyes

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