Evidence map›Paper›PMID 39195519›Full record

ArticleMetabolites2024

Metabolic Deficits in the Retina of a Familial Dysautonomia Mouse Model.

Stephanann M Costello, Anastasia Schultz, Donald Smith, Danielle Horan, Martha Chaverra, Brian Tripet, Lynn George, Brian Bothner, Frances Lefcort, Valérie Copié

Abstract read
In one paragraph

Article in Metabolites, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing 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

2 citing papers in PubMed.

  1. Article
  2. 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

10 authors.

Stephanann M CostelloDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.ORCID 0000-0002-9721-6742
Anastasia SchultzDepartment of Microbiology and Cell Biology, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Donald SmithDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Danielle HoranDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Martha ChaverraDepartment of Microbiology and Cell Biology, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Brian TripetDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Lynn GeorgeDepartment of Biological and Physical Sciences, Montana State University-Billings, Billings, MT 59102, USA.
Brian BothnerDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.ORCID 0000-0003-1295-9609
Frances LefcortDepartment of Microbiology and Cell Biology, Montana State University-Bozeman, Bozeman, MT 59717, USA.
Valérie CopiéDepartment of Chemistry and Biochemistry, Montana State University-Bozeman, Bozeman, MT 59717, USA.ORCID 0000-0002-2778-1463

Funding

Research Core (Developmental Research Project Program)P20GM103474 · NIGMS · MONTANA STATE UNIVERSITY - BOZEMAN · PI Ann Therese Bertagnolli · 2012 to 2026
$60.0M
Metabolic Control of gut-brain axis in Familial dysautonomia Supplement to parent grant 1R01DK117473-01A1 to support a PhD candidate/graduate student from an under-represented minorityR01DK117473 · NIDDK · MONTANA STATE UNIVERSITY - BOZEMAN · PI COPIE, VALERIE, WALK, SETH T · 2018 to 2022
$3.0M
The Nucleolus as a Nexus of Disease in Amyotrophic Lateral SclerosisR15NS090384 · NINDS · MONTANA STATE UNIVERSITY -BILLINGS · PI LYNN D GEORGE · 2015 to 2026
$1.6M
Acquisition of a 600 MHz NMR cryoprobe and 600 MHz spectrometer console upgradeS10RR026659 · NCRR · MONTANA STATE UNIVERSITY - BOZEMAN · PI COPIE, VALERIE · 2011 to 2011
$600k
Ultra Performance Liquid Chromatography Tandem Mass Spectrometer for MetabolomicsS10OD028650 · OD · MONTANA STATE UNIVERSITY - BOZEMAN · PI BOTHNER, BRIAN P · 2020 to 2020
$599k
Murdock Charitable Trust 2015066:MNLNational Science Foundation MRI-DBI-1532078; MRI-CHE-2018388NCRR NIH HHS S10 RR026659NIDDK NIH HHS R01 DK117473NIGMS NIH HHS P20 GM103474NIH HHS R01-DK117473; R15NS080384; P20GM103473; 1S10RR13878; 1S10RR026659; 1S10OD28650NIH HHS S10 OD028650NINDS NIH HHS R15 NS090384
6 · The paper itself

Abstract

Neurodegenerative retinal diseases such as glaucoma, diabetic retinopathy, Leber's hereditary optic neuropathy (LHON), and dominant optic atrophy (DOA) are marked by progressive death of retinal ganglion cells (RGC). This decline is promoted by structural and functional mitochondrial deficits, including electron transport chain (ETC) impairments, increased oxidative stress, and reduced energy (ATP) production. These cellular mechanisms associated with progressive optic nerve atrophy have been similarly observed in familial dysautonomia (FD) patients, who experience gradual loss of visual acuity due to the degeneration of RGCs, which is thought to be caused by a breakdown of mitochondrial structures, and a disruption in ETC function. Retinal metabolism plays a crucial role in meeting the elevated energetic demands of this tissue, and recent characterizations of FD patients' serum and stool metabolomes have indicated alterations in central metabolic processes and potential systemic deficits of taurine, a small molecule essential for retina and overall eye health. The present study sought to elucidate metabolic alterations that contribute to the progressive degeneration of RGCs observed in FD. Additionally, a critical subpopulation of retinal interneurons, the dopaminergic amacrine cells, mediate the integration and modulation of visual information in a time-dependent manner to RGCs. As these cells have been associated with RGC loss in the neurodegenerative disease Parkinson's, which shares hallmarks with FD, a targeted analysis of the dopaminergic amacrine cells and their product, dopamine, was also undertaken. One dimensional (1D) proton (

Indexed as

conditional knockout (CKO) FD mouse modeldopaminergic amacrine cellseye healthfamilial dysautonomia (FD)mass spectrometrymetabolomicsnuclear magnetic resonanceretinal ganglion cellsretinal immunohistologyretinal metabolismunivariate and multivariate statistical analyses

Identifiers

PMID39195519
PMCPMC11356057

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.