Evidence map›Paper›PMID 37776278›Full record

ArticleJournal of inherited metabolic disease2023

Grip strength in patients with galactosemia and in a galactose-1-phosphate uridylyltransferase (GALT)-null rat model.

Jared J Druss, Josephine Rudd Zhong Manis, Nancy L Potter, Judith L Fridovich-Keil

Open access · greenAbstract read
In one paragraph

Article in Journal of inherited metabolic disease, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.2field-weighted citation impact, top 45% 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

1 citing paper in PubMed, 1 citations in OpenAlex.

  1. 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

4 authors at 3 institutions in 1 country.

Jared J DrussDepartment of Human Genetics, Emory University School of Medicine, Atlanta, Georgia, USA.
Josephine Rudd Zhong ManisMedical College of Georgia, Augusta University, Augusta, Georgia, USA.
Nancy L PotterDepartment of Speech and Hearing Sciences, Elson S. Floyd College of Medicine, Washington State University, Spokane, Washington, DC, USA.
Judith L Fridovich-KeilDepartment of Human Genetics, Emory University School of Medicine, Atlanta, Georgia, USA.ORCID 0000-0001-7947-5807
Emory University · USAugusta University · USWashington State University Spokane · US

Funding

A rat model for studies of galactosemiaR21HD092785 · NICHD · EMORY UNIVERSITY · PI FRIDOVICH-KEIL, JUDITH L. · 2017 to 2018
$553k
NICHD NIH HHS R21 HD092785
6 · The paper itself

Abstract

Classic galactosemia (CG) and clinical variant galactosemia (CVG) are allelic inborn errors of metabolism that result from profound deficiency, and near-profound deficiency, respectively, of galactose-1-P uridylyltransferase (GALT). Despite early detection and lifelong dietary restriction of galactose, which is the current standard of care, most patients with CG/CVG grow to experience a range of long-term developmental and other complications. One of the less well-understood complications of CG/CVG is decreased hand grip strength, as reported by Potter et al. (2013). Here, we confirm this phenotype in an independent cohort of 36 cases (4-18 years) and 19 controls (4-17 years), and further demonstrate that the grip strength deficit observed in cases may be secondary to growth delay. Specifically, we found that when grip strength of cases and controls in a new cohort recruited in 2022 was plotted by weight, rather than age, the difference between cases and controls for both sexes disappeared. Reanalyzing data from the original 2013 cohort, we found that differences in weight accounted for grip strength differences between cases and controls in girls and young women, but not in boys and young men. Finally, we tested whether a GALT-null rat model of CG also showed a grip strength deficit-it did-and again the difference between GALT-null and wild-type rats associated with differences in body mass. Combined, these results confirm that GALT deficiency is associated with a grip strength deficit in both young patients with CG/CVG and GALT-null rats, and further demonstrate that this phenotype may be secondary to growth delay, and therefore not evidence of a muscle abnormality.

Indexed as

GalactosemiasAnimalsFemaleGalactoseGalactosephosphatesHand StrengthHumansMaleRatsUTP-Hexose-1-Phosphate UridylyltransferaseGalactosegalactose-1-phosphateGalactosephosphatesUTP-Hexose-1-Phosphate Uridylyltransferasegalactosemiagrip strengthgrowth delayrat model

Identifiers

PMID37776278
PMCPMC10842421
OpenAlexW4387217486

What OpenQuestion holds

Textmetadata
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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.