Evidence map›Paper›PMID 36212622›Full record

ArticleBiomarker insights2022

Glycerophosphoinositol is Elevated in Blood Samples From

Jon J Brudvig, Vicki J Swier, Tyler B Johnson, Jacob C Cain, Melissa Pratt, Mitch Rechtzigel, Hannah Leppert, An N Dang Do, Forbes D Porter, Jill M Weimer

Open access · goldAbstract read
In one paragraph

Article in Biomarker insights, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed
1.9field-weighted citation impact, top 14% 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

14 citing papers in PubMed, 17 citations in OpenAlex.

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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 at 4 institutions in 1 country.

Jon J BrudvigPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Vicki J SwierPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Tyler B JohnsonPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Jacob C CainPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Melissa PrattPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Mitch RechtzigelPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Hannah LeppertPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
An N Dang DoDivision of Translational Medicine, Eunice Kennedy Shriver National Institute of Child Health and Human Development, Bethesda, MD, USA.
Forbes D PorterDivision of Translational Medicine, Eunice Kennedy Shriver National Institute of Child Health and Human Development, Bethesda, MD, USA.
Jill M WeimerPediatrics & Rare Diseases, Sanford Research, Sioux Falls, SD, USA.
Sanford Research · USAmicus Therapeutics (United States) · USEunice Kennedy Shriver National Institute of Child Health and Human Development · USUniversity of South Dakota · US

Funding

Clinical Investigations of Inborn Errors of MetabolismZIAHD008989 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI PORTER, FORBES · 2020 to 2025
$5.1M
Intramural NIH HHS ZIA HD008989
6 · The paper itself

Abstract

Introduction: CLN3 Batten disease is a rare pediatric neurodegenerative lysosomal disorder caused by biallelic disease-associated variants in Methods: We performed an exhaustive metabolomic screen using serum samples from a novel minipig model of CLN3 Batten disease and validated findings in Results: CLN3 pig serum samples from 4 ages exhibited large elevations in 4 glycerophosphodiester species: glycerophosphoinositol (GPI), glycerophosphoethanolamine (GPE), glycerophosphocholine (GPC), and glycerophosphoserine (GPS). GPI and GPE exhibited the largest elevations, with similar elevations found in Conclusions: GPE and GPI could have utility as biomarkers of CLN3 disease status. GPI, in particular, shows consistent elevations across a diverse cohort of individuals with CLN3. This raises the potential to use these biomarkers as a blood-based diagnostic test or as an efficacy measure for disease-modifying therapies.

Indexed as

Biomarkerslipid metabolismmetabolomicsneuronal ceroid-lipofuscinoses

Identifiers

PMID36212622
PMCPMC9535353
OpenAlexW4285178172

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
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.