Evidence map›Paper›PMID 31398317›Full record

ArticleDevelopmental biology2019

Alkylglycerol monooxygenase, a heterotaxy candidate gene, regulates left-right patterning via Wnt signaling.

Anna R Duncan, Delfina P González, Florencia Del Viso, Andrew Robson, Mustafa K Khokha, John N Griffin

Open access · greenAbstract read
In one paragraph

Article in Developmental biology, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
0.6field-weighted citation impact, top 33% of its field
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

5 citing papers in PubMed, 11 citations in OpenAlex.

  1. Article
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  4. Review
  5. Review
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

6 authors at 1 institution in 1 country.

Anna R DuncanPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA.
Delfina P GonzálezPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA.
Florencia Del VisoPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA.
Andrew RobsonPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA.
Mustafa K KhokhaPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA. Electronic address: mustafa.khokha@yale.edu.
John N GriffinPediatric Genomics Discovery Program, Department of Pediatrics and Genetics, Yale University School of Medicine, 333 Cedar Street, New Haven, CT, 06510, USA.
Yale University · US

Funding

A system approach to the analysis of Heterotaxy Candidate GenesR01HD102186 · NICHD · YALE UNIVERSITY · PI KHOKHA, MUSTAFA K · 2020 to 2024
$3.1M
A system approach to the analysis of Heterotaxy Candiate GenesR01HD081379 · NICHD · YALE UNIVERSITY · PI KHOKHA, MUSTAFA K · 2014 to 2018
$2.7M
NICHD NIH HHS R01 HD081379NICHD NIH HHS R01 HD102186
6 · The paper itself

Abstract

Congenital heart disease (CHD) is a major cause of morbidity in the pediatric population yet its genetic and molecular causes remain poorly defined. Previously, we identified AGMO as a candidate heterotaxy disease gene, a disorder of left-right (LR) patterning that can have a profound effect on cardiac function. AGMO is the only known alkylglycerol monooxygenase, an orphan tetrahydrobiopterin dependent enzyme that cleaves the ether linkage in alkylglycerols. However, whether AGMO plays a role in LR patterning was unexplored. Here we reveal that Agmo is required for correct development of the embryonic LR axis in Xenopus embryos recapitulating the patient's heterotaxy phenotype. Mechanistically, we demonstrate that Agmo is a regulator of canonical Wnt signaling, required during gastrulation for normal formation of the left - right organizer. Mutational analysis demonstrates that this function is dependent on Agmo's alkylglycerol monooxygenase activity. Together, our findings identify Agmo as a regulator of canonical Wnt signaling, demonstrate a role for Agmo in embryonic axis formation, and provide insight into the poorly understood developmental requirements for ether lipid cleavage.

Indexed as

AnimalsBody PatterningDNA Mutational AnalysisGastrulaGene Expression Regulation, DevelopmentalLipid MetabolismLipidsMixed Function OxygenasesPhospholipid EthersWnt Signaling PathwayXenopusXenopus Proteinsglyceryl-ether monooxygenaseLipidsMixed Function OxygenasesPhospholipid EthersXenopus ProteinsAGMOAlkylglycerol monooxygenaseCongenital heart diseaseEther lipidsHeterotaxyTMEM195WntXenopus

Identifiers

PMID31398317
PMCPMC7784330
OpenAlexW2964553057

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

None linked

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.