Evidence map›Paper›PMID 29795570›Full record

ArticlePLoS genetics2018

Insights into the genetic epidemiology of Crohn's and rare diseases in the Ashkenazi Jewish population.

Manuel A Rivas, Brandon E Avila, Jukka Koskela, Hailiang Huang, Christine Stevens, Matti Pirinen, Talin Haritunians, Benjamin M Neale, Mitja Kurki, Andrea Ganna and 50 more

Erratum issuedAbstract read
In one paragraph

Article in PLoS genetics, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 54 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
54citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

54 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Exome sequencing directly implicates 68 genes in inflammatory bowel disease.medRxiv : the preprint server for health sciences · 2026
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  10. A lifesaving revolution delayed.Nature genetics · 2024
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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

60 authors.

Manuel A RivasMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Brandon E AvilaMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Jukka KoskelaMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Hailiang HuangMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.ORCID http://orcid.org/0000-0003-1461-5762
Christine StevensMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Matti PirinenInstitute for Molecular Medicine Finland (FIMM), University of Helsinki, Helsinki, Finland.
Talin HarituniansTranslational Genomics Unit, F. Widjaja Foundation Inflammatory Bowel and Immunobiology Research Institute, Cedars-Sinai Medical Center, Los Angeles, CA, United States of America.ORCID http://orcid.org/0000-0002-9005-7750
Benjamin M NealeMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Mitja KurkiMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.ORCID http://orcid.org/0000-0002-6010-8617
Andrea GannaMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Daniel GrahamMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Benjamin GlaserHadassah-Hebrew University Medical Center, Endocrinology and Metabolism Service Department of Internal Medicine, Jerusalem, Israel.
Inga PeterDepartment of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY, United States of America.
Gil AtzmonDepartment of Genetics and Medicine, Albert Einstein College of Medicine, Bronx, NY, United States of America.
Nir BarzilaiDepartment of Genetics and Medicine, Albert Einstein College of Medicine, Bronx, NY, United States of America.
Adam P LevineDivision of Medicine, University College London, London, United Kingdom.
Elena SchiffDivision of Medicine, University College London, London, United Kingdom.
Nikolas PontikosDivision of Medicine, University College London, London, United Kingdom.ORCID http://orcid.org/0000-0003-1782-4711
Ben WeisburdMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Monkol LekMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Konrad J KarczewskiMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.ORCID http://orcid.org/0000-0003-2878-4671
Jonathan BloomMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Eric V MinikelMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Britt-Sabina PetersenInstitute of Clinical Molecular Biology, Christian-Albrechts-University of Kiel, Kiel, Germany.
Laurent BeaugerieGastroenterology Department, Saint-Antoine Hospital, AP-HP, UPMC Univ Paris, Paris, France.
Philippe SeksikGastroenterology Department, Saint-Antoine Hospital, AP-HP, UPMC Univ Paris, Paris, France.
Jacques CosnesGastroenterology Department, Saint-Antoine Hospital, AP-HP, UPMC Univ Paris, Paris, France.
Stefan SchreiberDepartment of Internal Medicine, University Hospital Schleswig-Holstein, Kiel, Germany.
Bernd BokemeyerGastroenterology Practice, Minden, Germany.
Johannes BethgeDepartment of Internal Medicine, University Hospital Schleswig-Holstein, Kiel, Germany.
International IBD Genetics Consortium
NIDDK IBD Genetics Consortium
T2D-GENES Consortium
Graham HeapIBD Pharmacogenetics, Royal Devon and Exeter NHS Trust, Exeter, United Kingdom.ORCID http://orcid.org/0000-0003-4131-6792
Tariq AhmadPeninsula College of Medicine and Dentistry, Exeter, United Kingdom.
Vincent PlagnolUCL Genetics Institute, University College London, London, United Kingdom.
Anthony W SegalDivision of Medicine, University College London, London, United Kingdom.
Stephan TarganTranslational Genomics Unit, F. Widjaja Foundation Inflammatory Bowel and Immunobiology Research Institute, Cedars-Sinai Medical Center, Los Angeles, CA, United States of America.
Dan TurnerJuliet Keidan Institute of Pediatric Gastroenterology and Nutrition, Shaare Zedek Medical Center, The Hebrew University of Jerusalem, Jerusalem, Israel.
Paivi SaavalainenResearch Programs Unit, Immunobiology, and Department of Medical and Clinical Genetics, University of Helsinki, Helsinki, Finland.
Martti FarkkilaDepartment of Medicine, Division of Gastroenterology, Helsinki University Hospital, Helsinki, Finland.
Kimmo KontulaDepartment of Medicine, University of Helsinki and Helsinki University Central Hospital, Helsinki, Finland.
Aarno PalotieMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Steven R BrantMeyerhoff Inflammatory Bowel Disease Center, Department of Medicine, School of Medicine, Johns Hopkins University, Baltimore, MD, United States of America.
Richard H DuerrDivision of Gastroenterology, Hepatology and Nutrition, Department of Medicine, University of Pittsburgh School of Medicine, Pittsburgh, PA, United States of America.
Mark S SilverbergInflammatory Bowel Disease Centre, Mount Sinai Hospital, Toronto, Ontario, Canada.
John D RiouxResearch Center, Montreal Heart Institute, Montréal, Québec, Canada.ORCID http://orcid.org/0000-0001-7560-8326
Rinse K WeersmaDepartment of Gastroenterology and Hepatology, University Medical Center Groningen, Groningen, The Netherlands.
Andre FrankeInstitute of Clinical Molecular Biology, Christian-Albrechts-University of Kiel, Kiel, Germany.
Luke JostinsWellcome Trust Centre for Human Genetics, Oxford University, Oxford, United Kingdom.
Carl A AndersonWellcome Trust Sanger Institute, Wellcome Trust Genome Campus, Hinxton, United Kingdom.ORCID http://orcid.org/0000-0003-1719-7009
Jeffrey C BarrettWellcome Trust Sanger Institute, Wellcome Trust Genome Campus, Hinxton, United Kingdom.
Daniel G MacArthurMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Chaim JalasBonei Olam, Center for Rare Jewish Genetic Disorders, Brooklyn, NY, United States of America.
Harry SokolGastroenterology Department, Saint-Antoine Hospital, AP-HP, UPMC Univ Paris, Paris, France.
Ramnik J XavierMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.
Ann PulverDepartment of Psychiatry and Behavioral Sciences, Johns Hopkins University School of Medicine, Baltimore, MD, United States of America.
Judy H ChoIcahn School of Medicine at Mount Sinai, Dr Henry D. Janowitz Division of Gastroenterology, New York, NY, United States of America.
Dermot P B McGovernTranslational Genomics Unit, F. Widjaja Foundation Inflammatory Bowel and Immunobiology Research Institute, Cedars-Sinai Medical Center, Los Angeles, CA, United States of America.
Mark J DalyMedical and Population Genetics, Broad Institute, Cambridge, MA, United States of America.

Funding

Large Scale Sequencing and Analysis of GenomesU54HG003067 · NHGRI · MASSACHUSETTS INSTITUTE OF TECHNOLOGY · PI GABRIEL, STACEY, LANDER, ERIC S · 2004 to 2015
$568.6M
Pilot & Feasibility ProgramP30DK043351 · NIDDK · MASSACHUSETTS GENERAL HOSPITAL · PI RAYMOND T CHUNG · 1991 to 2026
$35.3M
Using polygenic scores to enhance both variant discovery, and understanding of functional consequences of genetic variation in IBD.P01DK046763 · NIDDK · CEDARS-SINAI MEDICAL CENTER · PI TARGAN, STEPHAN R. · 1992 to 2020
$31.1M
Precision IBD via genetics and genomics: integrating International and multi-omic datasets, expanding studies in diverse populations, and defining mechanisms of unmet clinical needs in IBDU24DK062429 · NIDDK · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI Ling-shiang Chuang, Ron Do · 2017 to 2026
$13.9M
Translational Research Enhancement CoreP30DK089502 · NIDDK · JOHNS HOPKINS UNIVERSITY · PI ZACHOS, NICHOLAS CONSTANTINE · 2011 to 2020
$12.0M
Yale University Inflammatory Bowel Disease Genetics Research CenterU01DK062422 · NIDDK · YALE UNIVERSITY · PI Ling-shiang Chuang · 2002 to 2026
$10.8M
IBD Gene Mapping by Clinical and Population SubsetsU01DK062431 · NIDDK · RUTGERS BIOMEDICAL AND HEALTH SCIENCES · PI Steven R Brant · 2002 to 2026
$10.7M
Universite de Montreal IBD Genetics Research CenterU01DK062432 · NIDDK · WHITEHEAD INSTITUTE FOR BIOMEDICAL RES · PI John D. Rioux · 2002 to 2026
$9.6M
NIDDK IBD Genetics Consortium Genetic Research CenterU01DK062420 · NIDDK · UNIVERSITY OF PITTSBURGH AT PITTSBURGH · PI DUERR, RICHARD H · 2002 to 2021
$7.8M
Impact of Genetic Variability on Cellular Pathways and Host-Microbiome InteractioU01DK062423 · NIDDK · SINAI HEALTH SYSTEM · PI MARK S. SILVERBERG · 2002 to 2026
$7.5M
ATG16L1 T300A: genetics to biologyR01DK097485 · NIDDK · MASSACHUSETTS GENERAL HOSPITAL · PI XAVIER, RAMNIK J · 2013 to 2021
$4.1M
Defining the genetic architecture of IBD in Ashkenazi Jewish populationsR01DK092235 · NIDDK · YALE UNIVERSITY · PI CHO, JUDY H. · 2012 to 2016
$2.5M
NHGRI NIH HHS U54 HG003067NIDDK NIH HHS K01 DK114379NIDDK NIH HHS P01 DK046763NIDDK NIH HHS P30 DK043351NIDDK NIH HHS P30 DK089502NIDDK NIH HHS R01 DK092235NIDDK NIH HHS R01 DK097485NIDDK NIH HHS U01 DK062420NIDDK NIH HHS U01 DK062422NIDDK NIH HHS U01 DK062423NIDDK NIH HHS U01 DK062431NIDDK NIH HHS U01 DK062432NIDDK NIH HHS U24 DK062429NIH HHS 5 U54 HG003067-13
6 · The paper itself

Abstract

As part of a broader collaborative network of exome sequencing studies, we developed a jointly called data set of 5,685 Ashkenazi Jewish exomes. We make publicly available a resource of site and allele frequencies, which should serve as a reference for medical genetics in the Ashkenazim (hosted in part at https://ibd.broadinstitute.org, also available in gnomAD at http://gnomad.broadinstitute.org). We estimate that 34% of protein-coding alleles present in the Ashkenazi Jewish population at frequencies greater than 0.2% are significantly more frequent (mean 15-fold) than their maximum frequency observed in other reference populations. Arising via a well-described founder effect approximately 30 generations ago, this catalog of enriched alleles can contribute to differences in genetic risk and overall prevalence of diseases between populations. As validation we document 148 AJ enriched protein-altering alleles that overlap with "pathogenic" ClinVar alleles (table available at https://github.com/macarthur-lab/clinvar/blob/master/output/clinvar.tsv), including those that account for 10-100 fold differences in prevalence between AJ and non-AJ populations of some rare diseases, especially recessive conditions, including Gaucher disease (GBA, p.Asn409Ser, 8-fold enrichment); Canavan disease (ASPA, p.Glu285Ala, 12-fold enrichment); and Tay-Sachs disease (HEXA, c.1421+1G>C, 27-fold enrichment; p.Tyr427IlefsTer5, 12-fold enrichment). We next sought to use this catalog, of well-established relevance to Mendelian disease, to explore Crohn's disease, a common disease with an estimated two to four-fold excess prevalence in AJ. We specifically attempt to evaluate whether strong acting rare alleles, particularly protein-truncating or otherwise large effect-size alleles, enriched by the same founder-effect, contribute excess genetic risk to Crohn's disease in AJ, and find that ten rare genetic risk factors in NOD2 and LRRK2 are enriched in AJ (p < 0.005), including several novel contributing alleles, show evidence of association to CD. Independently, we find that genomewide common variant risk defined by GWAS shows a strong difference between AJ and non-AJ European control population samples (0.97 s.d. higher, p<10-16). Taken together, the results suggest coordinated selection in AJ population for higher CD risk alleles in general. The results and approach illustrate the value of exome sequencing data in case-control studies along with reference data sets like ExAC (sites VCF available via FTP at ftp.broadinstitute.org/pub/ExAC_release/release0.3/) to pinpoint genetic variation that contributes to variable disease predisposition across populations.

Indexed as

AlgorithmsCrohn DiseaseGenetic Predisposition to DiseaseGenetics, PopulationGenome-Wide Association StudyHaplotypesHumansJewsModels, GeneticMolecular EpidemiologyPolymorphism, Single NucleotideRare Diseases

Identifiers

PMID29795570
PMCPMC5967709

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.