Evidence map›Paper›PMID 26912795›Full record

ArticleMolecular biology of the cell2016

trappc11 is required for protein glycosylation in zebrafish and humans.

Charles DeRossi, Ana Vacaru, Ruhina Rafiq, Ayca Cinaroglu, Dru Imrie, Shikha Nayar, Anastasia Baryshnikova, Miroslav P Milev, Daniela Stanga, Dhara Kadakia and 6 more

Abstract read
In one paragraph

Article in Molecular biology of the cell, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
19citing papers in PubMed, 1 pooled it
3.6field-weighted citation impact, top 8% 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

19 citing papers in PubMed, 1 synthesis or guideline pooled it, 43 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Review
  4. Article
  5. Review
  6. Methods to Study Liver Disease Using Zebrafish Larvae.Methods in molecular biology (Clifton, N.J.) · 2024
    Article
  7. Article
  8. Review
  9. A novel homozygous variant inJournal of medical genetics · 2021
    Article
  10. Digenic Variants in theFrontiers in neuroscience · 2021
    Article
  11. Review
  12. Article
  13. Article
  14. Article
  15. Article
  16. Zebrafish as a Model for Obesity and Diabetes.Frontiers in cell and developmental biology · 2018
    Review
  17. NovelCase reports in genetics · 2018
    Article
  18. Article
  19. 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

16 authors at 7 institutions in 2 countries.

Charles DeRossiDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Department of Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Ana VacaruDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Department of Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Ruhina RafiqDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Department of Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Ayca CinarogluDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Department of Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Dru ImrieDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Graduate School of Biomedical Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Shikha NayarDepartment of Pediatrics and Mindich Institute for Child Health, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Anastasia BaryshnikovaLewis-Sigler Institute for Integrative Genomics, Princeton University, Princeton, NJ 08544.
Miroslav P MilevDepartment of Biology, Concordia University, Montreal, QC H4B 1R6, Canada.
Daniela StangaDepartment of Biology, Concordia University, Montreal, QC H4B 1R6, Canada.
Dhara KadakiaDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Ningguo GaoDepartment of Pharmacology, University of Texas Southwestern Medical Center, Dallas, TX 75390.
Jaime ChuDepartment of Pediatrics and Mindich Institute for Child Health, Icahn School of Medicine at Mount Sinai, New York, NY 10029.
Hudson H FreezeSanford Children's Health Research Center, Sanford Burnham Prebys Medical Discovery Institute, La Jolla, CA 92037.
Mark A LehrmanDepartment of Pharmacology, University of Texas Southwestern Medical Center, Dallas, TX 75390.
Michael SacherDepartment of Biology, Concordia University, Montreal, QC H4B 1R6, Canada Department of Anatomy and Cell Biology, McGill University, Montreal, QC H3A 0C7, Canada.
Kirsten C SadlerDepartment of Medicine, Division of Liver Diseases, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Department of Developmental and Regenerative Biology, Icahn School of Medicine at Mount Sinai, New York, NY 10029 Graduate School of Biomedical Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029 kirsten.edepli@nyu.edu.
Icahn School of Medicine at Mount Sinai · USChild Health and Development Institute · USConcordia University · CAThe University of Texas Southwestern Medical Center · USMcGill University · CAPrinceton University · USSanford Burnham Prebys Medical Discovery Institute · US

Funding

New Congenital Disorders of Glycosylation: Therapy and ModelsR01DK099551 · NIDDK · SANFORD BURNHAM PREBYS MEDICAL DISCOVERY INSTITUTE · PI Hudson H. Freeze · 2014 to 2026
$6.7M
The N-Glycosylation Pathway And Signaling By Free Mannose-6-PhosphateR01GM038545 · NIGMS · UT SOUTHWESTERN MEDICAL CENTER · PI LEHRMAN, MARK · 1987 to 2017
$5.7M
Understanding fatty liver disease using the zebrafish mutant, foie grasR01AA018886 · NIAAA · NEW YORK UNIVERSITY · PI SADLER EDEPLI, KIRSTEN C · 2009 to 2017
$2.4M
TRAINING PROGRAM IN INVESTIGATIVE GASTROENTEROLOGYT32DK007792 · NIDDK · MOUNT SINAI SCHOOL OF MEDICINE OF NYU · PI CHO, JUDY H. · 2001 to 2016
$2.3M
Testing Substrate-Flux Therapies for Glycosylation Disorders using ZebrafishRC1HD064159 · NICHD · SANFORD BURNHAM PREBYS MEDICAL DISCOVERY INSTITUTE · PI FREEZE, HUDSON H., LEHRMAN, MARK · 2009 to 2010
$1000k
A novel role for p53 in congenital disorders of glycosylationK08DK101340 · NIDDK · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI CHU, JAIME C · 2013 to 2016
$554k
Canadian Institutes of Health ResearchNIAAA NIH HHS R01 AA018886NIAAA NIH HHS R01AA018886NICHD NIH HHS RC1 HD064159NIDDK NIH HHS K08 DK101340NIDDK NIH HHS R01 DK099551NIDDK NIH HHS T32 DK007792NIGMS NIH HHS R01 GM038545
6 · The paper itself

Abstract

Activation of the unfolded protein response (UPR) can be either adaptive or pathological. We term the pathological UPR that causes fatty liver disease a "stressed UPR." Here we investigate the mechanism of stressed UPR activation in zebrafish bearing a mutation in thetrappc11gene, which encodes a component of the transport protein particle (TRAPP) complex.trappc11mutants are characterized by secretory pathway defects, reflecting disruption of the TRAPP complex. In addition, we uncover a defect in protein glycosylation intrappc11mutants that is associated with reduced levels of lipid-linked oligosaccharides (LLOs) and compensatory up-regulation of genes in the terpenoid biosynthetic pathway that produces the LLO anchor dolichol. Treating wild-type larvae with terpenoid or LLO synthesis inhibitors phenocopies the stressed UPR seen intrappc11mutants and is synthetically lethal withtrappc11mutation. We propose that reduced LLO level causing hypoglycosylation is a mechanism of stressed UPR induction intrappc11mutants. Of importance, in human cells, depletion of TRAPPC11, but not other TRAPP components, causes protein hypoglycosylation, and lipid droplets accumulate in fibroblasts from patients with theTRAPPC11mutation. These data point to a previously unanticipated and conserved role for TRAPPC11 in LLO biosynthesis and protein glycosylation in addition to its established function in vesicle trafficking.

Indexed as

Unfolded Protein ResponseAnimalsAnimals, Genetically ModifiedAtorvastatinDolicholsGlycosylationGolgi ApparatusHumansLarvaLipidsLiverMutationOligosaccharidesTerpenesVesicular Transport ProteinsZebrafishAtorvastatinDolicholsLipidsOligosaccharidesTerpenesTRAPPC11 protein, humanTRAPPC11 protein, zebrafishVesicular Transport ProteinsZebrafish Proteins

Identifiers

PMID26912795
PMCPMC4831877
OpenAlexW2303781241

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-SA
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.