Evidence map›Paper›PMID 39277505›Full record

ReviewNeurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics2024

Neurological glycogen storage diseases and emerging therapeutics.

Matthieu Colpaert, Pankaj K Singh, Katherine J Donohue, Natacha T Pires, David D Fuller, Manuela Corti, Barry J Byrne, Ramon C Sun, Craig W Vander Kooi, Matthew S Gentry

Abstract readReview
In one paragraph

Review in Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing 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

10 citing papers in PubMed.

  1. Review
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Spatial Molecular Imaging of the Glycome Using Mass Spectrometry.Journal of visualized experiments : JoVE · 2025
    Article
  8. Article
  9. Review
  10. 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

10 authors.

Matthieu ColpaertDepartment of Biochemistry and Molecular Biology, College of Medicine, University of Florida, Gainesville, FL, USA.
Pankaj K SinghDepartment of Biochemistry and Molecular Biology, College of Medicine, University of Florida, Gainesville, FL, USA.
Katherine J DonohueChelsea's Hope Lafora Children Research Fund, USA.
Natacha T PiresAdult Polyglucosan Body Disease Research Foundation, USA.
David D FullerDepartment of Physical Therapy and Breathing Research and Therapeutics Center, University of Florida, Gainesville, FL, USA.
Manuela CortiDepartment of Pediatrics, Powell Gene Therapy Center, College of Medicine, University of Florida, Gainesville, FL, USA.
Barry J ByrneDepartment of Pediatrics, Powell Gene Therapy Center, College of Medicine, University of Florida, Gainesville, FL, USA.
Ramon C SunDepartment of Biochemistry and Molecular Biology, College of Medicine, University of Florida, Gainesville, FL, USA; Center for Advanced Spatial Biomolecule Research (CASBR), University of Florida, Gainesville, FL, USA.
Craig W Vander KooiDepartment of Biochemistry and Molecular Biology, College of Medicine, University of Florida, Gainesville, FL, USA; Center for Advanced Spatial Biomolecule Research (CASBR), University of Florida, Gainesville, FL, USA.
Matthew S GentryDepartment of Biochemistry and Molecular Biology, College of Medicine, University of Florida, Gainesville, FL, USA; Center for Advanced Spatial Biomolecule Research (CASBR), University of Florida, Gainesville, FL, USA. Electronic address: matthew.gentry@ufl.edu.

Funding

Brain Glycogen-Metabolism,Mechanisms, and Therapeutic PotentialR35NS116824 · NINDS · UNIVERSITY OF KENTUCKY · PI Matthew S. Gentry · 2020 to 2026
$8.2M
Immunomodulation Approaches to Improve Safety And Efficacy of Gene Therapy Treatment in Friedreich’s AtaxiaU01NS116752 · NINDS · UNIVERSITY OF FLORIDA · PI CORTI, MANUELA · 2021 to 2025
$3.8M
Deciphering the Glycan Code in Human Alzheimer’s Disease BrainR01AG078702 · NIA · UNIVERSITY OF KENTUCKY · PI Peggi M Angel, Sean Curtis Bendall · 2022 to 2026
$3.8M
Aberrant Glycogen Modulates Cerebral Glucose Metabolism in Aging and Alzheimer's DiseaseR01AG066653 · NIA · UNIVERSITY OF KENTUCKY · PI SUN, RAMON C. · 2020 to 2024
$1.9M
Ampakines and Respiratory NeuroplasticityR01HL139708 · NHLBI · UNIVERSITY OF FLORIDA · PI FULLER, DAVID D · 2018 to 2021
$1.5M
Treatment of Lafora disease with an antibody-enzyme fusionR33NS111081 · NINDS · UNIVERSITY OF FLORIDA · PI GENTRY, MATTHEW S. · 2023 to 2024
$751k
NHLBI NIH HHS R01 HL139708NIA NIH HHS R01 AG066653NIA NIH HHS R01 AG078702NINDS NIH HHS R33 NS111081NINDS NIH HHS R35 NS116824NINDS NIH HHS U01 NS116752
6 · The paper itself

Abstract

Glycogen storage diseases (GSDs) comprise a group of inherited metabolic disorders characterized by defects in glycogen metabolism, leading to abnormal glycogen accumulation in multiple tissues, most notably affecting the liver, skeletal muscle, and heart. Recent findings have uncovered the importance of glycogen metabolism in the brain, sustaining a myriad of physiological functions and linking its perturbation to central nervous system (CNS) pathology. This link resulted in classification of neurological-GSDs (n-GSDs), a group of diseases with shared deficits in neurological glycogen metabolism. The n-GSD patients exhibit a spectrum of clinical presentations with common etiology while requiring tailored therapeutic approaches from the traditional GSDs. Recent research has elucidated the genetic and biochemical mechanisms and pathophysiological basis underlying different n-GSDs. Further, the last decade has witnessed some promising developments in novel therapeutic approaches, including enzyme replacement therapy (ERT), substrate reduction therapy (SRT), small molecule drugs, and gene therapy targeting key aspects of glycogen metabolism in specific n-GSDs. This preclinical progress has generated noticeable success in potentially modifying disease course and improving clinical outcomes in patients. Herein, we provide an overview of current perspectives on n-GSDs, emphasizing recent advances in understanding their molecular basis, therapeutic developments, underscore key challenges and the need to deepen our understanding of n-GSDs pathogenesis to develop better therapeutic strategies that could offer improved treatment and sustainable benefits to the patients.

Indexed as

Genetic TherapyGlycogen Storage DiseaseAnimalsEnzyme Replacement TherapyGlycogenHumansNervous System DiseasesGlycogenAdult polyglucosan body disease (APBD)Glycogen storage diseases (GSDs)Lafora disease (LD)Pompe diseaseSubstrate reduction therapy (SRT)

Identifiers

PMID39277505
PMCPMC11581880

What OpenQuestion holds

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Registered trials

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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.