Evidence map›Paper›PMID 36309498›Full record

ArticleNature communications2022

Genome-wide rare variant score associates with morphological subtypes of autism spectrum disorder.

Ada J S Chan, Worrawat Engchuan, Miriam S Reuter, Zhuozhi Wang, Bhooma Thiruvahindrapuram, Brett Trost, Thomas Nalpathamkalam, Carol Negrijn, Sylvia Lamoureux, Giovanna Pellecchia and 16 more

Abstract read
In one paragraph

Article in Nature communications, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.

0numbers the graph read from it
0cells of the map it votes in
17citing papers in PubMed
–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

17 citing papers in PubMed.

  1. Article
  2. Article
  3. Review
  4. Article
  5. Trio exome sequencing identifies de novo variants in novel candidate genes in 19.62% of CAKUT families.Genetics in medicine : official journal of the American College of Medical Genetics · 2025
    Article
  6. Article
  7. Article
  8. Article
  9. mAutism research : official journal of the International Society for Autism Research · 2025
    Article
  10. Article
  11. Article
  12. Article
  13. Contribution of autosomal rare andmedRxiv : the preprint server for health sciences · 2024
    Article
  14. Review
  15. The genetics and pathogenesis of CAKUT.Nature reviews. Nephrology · 2023
    Review
  16. Article
  17. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

26 authors.

Ada J S ChanThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Worrawat EngchuanThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Miriam S ReuterThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0001-8662-6685
Zhuozhi WangThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Bhooma ThiruvahindrapuramThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0003-1128-008X
Brett TrostThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0003-4863-7273
Thomas NalpathamkalamThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Carol NegrijnProvincial Medical Genetics Program, Eastern Health, St. John's, NL, Canada.
Sylvia LamoureuxThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Giovanna PellecchiaThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0003-4747-3473
Rohan V PatelThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0001-8674-9706
Wilson W L SungThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0002-4703-5118
Jeffrey R MacDonaldThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0001-5058-2393
Jennifer L HoweThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Jacob VorstmanThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0002-1677-3126
Neal SondheimerDepartment of Molecular Genetics, University of Toronto, Toronto, ON, Canada.ORCID 0000-0001-9982-5712
Nicole TakahashiThompson Center for Autism and Neurodevelopmental Disorders, University of Missouri, Columbia, MO, USA.
Judith H MilesThompson Center for Autism and Neurodevelopmental Disorders, University of Missouri, Columbia, MO, USA.
Evdokia AnagnostouDepartment of Pediatrics, University of Toronto, Toronto, ON, Canada.
Kristiina TammimiesDepartment of Women's and Children's Health, Karolinska Institutet, Stockholm, Sweden.ORCID 0000-0002-8324-4697
Mehdi ZarreiThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.
Daniele MericoThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0002-3728-4401
Dimitri J StavropoulosDepartment of Paediatric Laboratory Medicine, Genome Diagnostics, The Hospital for Sick Children, Toronto, ON, Canada.
Ryan K C YuenThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada.ORCID 0000-0001-7273-4968
Bridget A FernandezProvincial Medical Genetics Program, Eastern Health, St. John's, NL, Canada. bfernandez@chla.usc.edu.ORCID 0000-0003-1265-8259
Stephen W SchererThe Centre for Applied Genomics, Genetics and Genome Biology, The Hospital for Sick Children, Toronto, ON, Canada. stephen.scherer@sickkids.ca.ORCID 0000-0002-8326-1999

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Defining different genetic subtypes of autism spectrum disorder (ASD) can enable the prediction of developmental outcomes. Based on minor physical and major congenital anomalies, we categorize 325 Canadian children with ASD into dysmorphic and nondysmorphic subgroups. We develop a method for calculating a patient-level, genome-wide rare variant score (GRVS) from whole-genome sequencing (WGS) data. GRVS is a sum of the number of variants in morphology-associated coding and non-coding regions, weighted by their effect sizes. Probands with dysmorphic ASD have a significantly higher GRVS compared to those with nondysmorphic ASD (P = 0.03). Using the polygenic transmission disequilibrium test, we observe an over-transmission of ASD-associated common variants in nondysmorphic ASD probands (P = 2.9 × 10

Indexed as

Autism Spectrum DisorderCanadaChildGenetic Predisposition to DiseaseGenomeHumansMultifactorial InheritanceWhole Genome Sequencing

Identifiers

PMID36309498
PMCPMC9617891

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Registered trials

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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.