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Record W2584469054 · doi:10.1038/nature21039

Rare and low-frequency coding variants alter human adult height

2017· article· en· W2584469054 on OpenAlexafffund
Eirini Marouli, Mariaelisa Graff, Carolina Medina‐Gómez, Ken Sin Lo, Andrew R. Wood, Troels R. Kjær, Rebecca S. Fine, Yingchang Lu, Claudia Schurmann, Heather M. Highland, Sina Rüeger, Guðmar Þorleifsson, Anne E. Justice, David Lamparter, Kathleen Stirrups, Valérie Turcot, Kristin L. Young, Thomas W. Winkler, Tõnu Esko, Tugce Karaderi, Adam E. Locke, Nicholas G. D. Masca, Maggie Ng, Poorva Mudgal, Manuel A. Rivas, Sailaja Vedantam, Anubha Mahajan, Xiuqing Guo, Gonçalo R. Abecasis, Katja K.H. Aben, Linda S. Adair, Dewan S Alam, Eva Albrecht, Kristine H. Allin, Matthew Allison, Philippe Amouyel, Emil V. R. Appel, Dominique Arveiler, Folkert W. Asselbergs, Paul L. Auer, Beverley Balkau, Bernhard Banas, Lia E. Bang, Marianne Benn, Sven Bergmann, Lawrence F. Bielak, Matthias Blüher, Heiner Boeing, Eric Boerwinkle, Carsten A. Böger, Lori L. Bonnycastle, Jette Bork‐Jensen, Michiel L. Bots, Erwin P. Böttinger, Donald W. Bowden, Ivan Brandslund, Gerome Breen, Murray H. Brilliant, Linda Broer, Amber Burt, Adam S. Butterworth, David J. Carey, Mark J. Caulfield, John C. Chambers, Daniel I. Chasman, Yii‐Der Ida Chen, Rajiv Chowdhury, Cramer Christensen, Audrey Y. Chu, Massimiliano Cocca, Francis S. Collins, James P. Cook, Janie Corley, Jordi Corominas Galbany, Amanda J. Cox, Gabriel Cuéllar-Partida, John Danesh, Gail Davies, Paul I. W. de Bakker, Gert J. de Borst, Simon de Denus, Mark de Groot, Renée de Mutsert, Ian J. Deary, George Dedoussis, Ellen W. Demerath, Anneke I. den Hollander, Joe Dennis, Emanuele Di Angelantonio, Fotios Drenos, Mengmeng Du, Alison M. Dunning, Douglas F. Easton, Tapani Ebeling, Todd L. Edwards, Patrick T. Ellinor, Paul Elliott, Εvangelos Εvangelou, Aliki‐Eleni Farmaki, Jessica D. Faul, Mary F. Feitosa, Shuang Feng, Ele Ferrannini, Maurizio Ferrario, Jean Ferrières, José C. Florez, Ian Ford, Myriam Fornage, Paul W. Franks, Ruth Frikke‐Schmidt, Tessel E. Galesloot, Wei Gan, Ilaria Gandin, Paolo Gasparini, Vilmantas Giedraitis, Ayush Giri, Giorgia Girotto, Scott D. Gordon, Penny Gordon‐Larsen, Mathias Gorski, Niels Grarup, Megan L. Grove, Vilmundur Guðnason, Stefan Gustafsson, Torben Hansen, Kathleen Mullan Harris, Tamara B. Harris, Andrew T. Hattersley, Caroline Hayward, Liang He, Iris M. Heid, Kauko Heikkilä, Øyvind Helgeland, Jussi Hernesniemi, Alex W. Hewitt, Lynne J. Hocking, Mette Hollensted, Oddgeir L. Holmen, G. Kees Hovingh, Joanna M. M. Howson, Carel B. Hoyng, Paul L. Huang, Kristian Hveem, M. Arfan Ikram, Erik Ingelsson, Anne Jackson, Jan‐Håkan Jansson, Gail P. Jarvik, Gorm Boje Jensen, Min A. Jhun, Yucheng Jia, Xuejuan Jiang, Stefan Johansson, Marit E. Jørgensen, Torben Jørgensen, J. Wouter Jukema, Bratati Kahali, René S. Kahn, Mika Kähönen, Pia R. Kamstrup, Stavroula Kanoni, Jaakko Kaprio, Maria Karaleftheri, Sharon L. R. Kardia, Fredrik Karpe, Frank Kee, Renske Keeman, Lambertus A. Kiemeney, Hidetoshi Kitajima, Kirsten B. Kluivers, Thomas Köcher, Pirjo Komulainen, Jukka Kontto, Jaspal S. Kooner, Charles Kooperberg, Péter Kovács, Jennifer Kriebel, Helena Kuivaniemi, Sébastien Küry, Johanna Kuusisto, Martina La Bianca, Markku Laakso, Timo A. Lakka, Ethan M. Lange, Leslie A. Lange, Carl D. Langefeld, Claudia Langenberg, Eric B. Larson, I‐Te Lee, Terho Lehtimäki, Cora E. Lewis, Huaixing Li, Jin Li, Ruifang Li‐Gao, Honghuang Lin, Li‐An Lin, Lin Xu, Lars Lind, Jaana Lindström, Allan Linneberg, Yeheng Liu, Ching‐Ti Liu, Artitaya Lophatananon, Jian’an Luan, Steven A. Lubitz, Leo‐Pekka Lyytikäinen, David A. Mackey, Pamela A. F. Madden, Alisa K. Manning, Satu Männistö, Gaëlle Marenne, Jonathan Marten, Nicholas G. Martin, Angela L. Mazul, Karina Meidtner, Andres Metspalu, Paul Mitchell, Karen L. Mohlke, Dennis O. Mook‐Kanamori, Anna Morgan, Andrew D. Morris, Andrew P. Morris, Martina Müller‐Nurasyid, Patricia B. Munroe, Mike A. Nalls, Matthias Nauck, Christopher P. Nelson, Matt J. Neville, Sune F. Nielsen, Kjell Nikus, Pål R. Njølstad, Børge G. Nordestgaard, Ιωάννα Ντάλλα, Jeffrey R. O’Connel, Heikki Oksa, Loes M. Olde Loohuis, Roel A. Ophoff, Katharine R. Owen, Chris J. Packard, Sandosh Padmanabhan, Gerard Pasterkamp, Aniruddh P. Patel, Alison Pattie, Oluf Pedersen, Peggy Peissig, Gina M. Peloso, Craig E. Pennell, Markus Perola, James A. Perry, John R. B. Perry, Thomas N. Person, Ailith Pirie, Ozren Polašek, Daniëlle Posthuma, Olli T. Raitakari, Asif Rasheed, Rainer Rauramaa, Dermot F. Reilly, Alex P. Reiner, Frida Renström, Paul M. Ridker, John D. Rioux, Neil Robertson, Antonietta Robino, Olov Rolandsson, Igor Rudan, Katherine S. Ruth, Danish Saleheen, Veikko Salomaa, Nilesh J. Samani, Kevin Sandow, Yadav Sapkota, Naveed Sattar, Marjanka K. Schmidt, Pamela J. Schreiner, Matthias B. Schulze, Robert A. Scott, Marcelo P. Segura-Lepe, Svati H. Shah, Xueling Sim, Suthesh Sivapalaratnam, Kerrin S. Small, Albert V. Smith, Jennifer A. Smith, Lorraine Southam, Timothy D. Spector, Elizabeth K. Speliotes, John M. Starr, Valgerður Steinthórsdóttir, Heather M. Stringham, Michael Stümvoll, Praveen Surendran, Leen M. ‘t Hart, Katherine E. Tansey, Jean‐Claude Tardif, Kent D. Taylor, Alexander Teumer, Deborah J. Thompson, Unnur Þorsteinsdóttir, Betina H. Thuesen, Anke Tönjes, Gerard Tromp, Stella Trompet, Emmanouil Tsafantakis, Jaakko Tuomilehto, Anne Tybjærg‐Hansen, Jonathan P. Tyrer, Rudolf Uher, André G. Uitterlinden, Sheila Ulivi, Sander W. van der Laan, Andries R. van der Leij, Cornelia M. van Duijn, Natasja M. van Schoor, Jessica van Setten, Anette Varbo, Tibor V. Varga, Rohit Varma, Digna R. Velez Edwards, Sita H. Vermeulen, Henrik Vestergaard, Véronique Vitart, Thomas Vogt, Diego Vozzi, Mark Walker, Feijie Wang, Carol A. Wang, Shuai Wang, Yiqin Wang, Nicholas J. Wareham, Helen R. Warren, Jennifer Wessel, Sara M. Willems, James G. Wilson, Daniel R. Witte, Michael O. Woods, Ying Wu, Hanieh Yaghootkar, Jie Yao, Pang Yao, Laura M. Yerges-Armstrong, Robin Young, Eleftheria Zeggini, Xiaowei Zhan, Weihua Zhang, Wei Zhao, Zheng He, Wei Zhou, Jerome I. Rotter, Michael Boehnke, Sekar Kathiresan, Mark I. McCarthy, Cristen J. Willer, Kāri Stefánsson, Ingrid B. Borecki, Dajiang J. Liu, Kari E. North, Nancy L. Heard‐Costa, Tune H. Pers, Cecilia M. Lindgren, Claus Oxvig, Zoltán Kutalik, Fernando Rivadeneira, Ruth J. F. Loos, Timothy M. Frayling, Joel N. Hirschhorn, Panos Deloukas, Guillaume Lettre

Bibliographic record

VenueNature · 2017
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicGenetic Associations and Epidemiology
Canadian institutionsMemorial University of NewfoundlandUniversité de MontréalDalhousie UniversityMontreal Heart Institute
FundersInstitute of GeneticsCarolina Population Center, University of North Carolina at Chapel HillNational Center for Advancing Translational SciencesNational Institute of Diabetes and Digestive and Kidney DiseasesNetherlands Heart InstituteNIHR Oxford Biomedical Research CentreHelmholtz Zentrum MünchenNational Heart, Lung, and Blood InstituteBiotechnology and Biological Sciences Research CouncilSocial Science Research Institute, Duke UniversitySchool of Medicine, Stanford UniversityNational Institutes of HealthHjartaverndSteno Diabetes Center CopenhagenTaysNovo Nordisk FondenNational Institute of Mental HealthSyddansk UniversitetUniversità di PisaTerveyden ja hyvinvoinnin laitosMenzies Institute for Medical ResearchBritish Heart FoundationHáskóli ÍslandsRegion HovedstadenNational Institute of General Medical SciencesLeids Universitair Medisch CentrumUniversitetet i BergenTampereen YliopistoLunds UniversitetNational Cancer InstituteUniversity College LondonLundbeckfondenAalborg UniversitetUniversity of GlasgowUniversiteit LeidenUniversity of North Carolina at Chapel HillHarvard UniversityScience for Life LaboratoryNational Human Genome Research InstituteRigshospitaletUniversity of Texas Health Science Center at HoustonUniversità degli Studi dell'InsubriaMenzies Research Institute TasmaniaUniversity of AberdeenGentofte HospitalUniversity of ExeterUniversity of Southern CaliforniaWellcome TrustSidra MedicineNational Eye InstituteNational Institute for Health and Care ResearchUmeå UniversitetHelsingin YliopistoImperial College LondonUniversity of TasmaniaEunice Kennedy Shriver National Institute of Child Health and Human DevelopmentHaukeland UniversitetssjukehusUnited Kingdom Clinical Research CollaborationErasmus Medisch CentrumEuropean Hematology AssociationUniversité de ToulouseUniversity of OxfordNorges Teknisk-Naturvitenskapelige UniversitetMedical Research CouncilUppsala UniversitetUniversity of California, Los AngelesUniversity of WashingtonDoris Duke Charitable Foundation
KeywordsBiologyGeneticsAlleleGeneQuantitative trait locusGenome-wide association studyGenetic associationPhenotypeMinor allele frequencyAllele frequencyHuman genomeCoding regionGenomeSingle-nucleotide polymorphismGenotype

Abstract

fetched live from OpenAlex

Height is a highly heritable, classic polygenic trait with approximately 700 common associated variants identified through genome-wide association studies so far. Here, we report 83 height-associated coding variants with lower minor-allele frequencies (in the range of 0.1–4.8%) and effects of up to 2 centimetres per allele (such as those in IHH, STC2, AR and CRISPLD2), greater than ten times the average effect of common variants. In functional follow-up studies, rare height-increasing alleles of STC2 (giving an increase of 1–2 centimetres per allele) compromised proteolytic inhibition of PAPP-A and increased cleavage of IGFBP-4 in vitro, resulting in higher bioavailability of insulin-like growth factors. These 83 height-associated variants overlap genes that are mutated in monogenic growth disorders and highlight new biological candidates (such as ADAMTS3, IL11RA and NOX4) and pathways (such as proteoglycan and glycosaminoglycan synthesis) involved in growth. Our results demonstrate that sufficiently large sample sizes can uncover rare and low-frequency variants of moderate-to-large effect associated with polygenic human phenotypes, and that these variants implicate relevant genes and pathways. Data from over 700,000 individuals reveal the identity of 83 sequence variants that affect human height, implicating new candidate genes and pathways as being involved in growth. As a highly heritable polygenic trait, human height has provided a model for the genetic analysis of complex traits. So far about 700 common genetic variants have been linked to height through genome-wide association studies, but the role of low-frequency and rare variants has not been systematically explored. Guillaume Lettre, Joel Hirschhorn and colleagues in the GIANT Consortium now report their analysis of coding regions in the genomes of 711,418 individuals. They identify 120 loci newly associated with height, including 32 rare and 51 low-frequency coding variants. They highlight 83 candidate genes with low-frequency height-associated variants and implicate biological pathways with known roles in growth disorders as well as new candidates. Their analyses provide insights into the genomic architecture of human height.

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)0.000
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: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.004
Threshold uncertainty score0.014

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.002
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0010.001
Science and technology studies0.0000.000
Scholarly communication0.0010.000
Open science0.0000.000
Research integrity0.0010.001
Insufficient payload (model declined to judge)0.0040.000

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 head0.284
Teacher spread0.275 · 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".

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Citations654
Published2017
Admission routes2
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