Evidence map›Paper›PMID 38703411›Full record

ReviewMolecular genetics and metabolism2024

ALG13-Congenital Disorder of Glycosylation (ALG13-CDG): Updated clinical and molecular review and clinical management guidelines.

Rameen Shah, Erik A Eklund, Silvia Radenkovic, Mustafa Sadek, Ibrahim Shammas, Sanne Verberkmoes, Bobby G Ng, Hudson H Freeze, Andrew C Edmondson, Miao He and 3 more

Abstract readReview
In one paragraph

Review in Molecular genetics and metabolism, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing 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

8 citing papers in PubMed.

  1. Article
  2. Review
  3. Review
  4. Article
  5. NovelHuman mutation · 2026
    Article
  6. Review
  7. Article
  8. 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

13 authors.

Rameen ShahDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA; Department of Biochemistry and Molecular Biology, Mayo Clinic, Rochester, MN, USA.
Erik A EklundDepartment of Clinical Sciences, Lund, Pediatrics, Lund University, Lund, Sweden; Nuffield Department of Clinical Neurosciences, University of Oxford, Oxford, UK.
Silvia RadenkovicDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA.
Mustafa SadekDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA.
Ibrahim ShammasDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA.
Sanne VerberkmoesDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA.
Bobby G NgHuman Genetics Program, Sanford Children's Health Research Center, Sanford Burnham Prebys Medical Discovery Institute, La Jolla, CA 92037, USA.
Hudson H FreezeHuman Genetics Program, Sanford Children's Health Research Center, Sanford Burnham Prebys Medical Discovery Institute, La Jolla, CA 92037, USA.
Andrew C EdmondsonDivision of Human Genetics, Department of Pediatrics, Children's Hospital of Philadelphia, PA, USA.
Miao HeDepartment of Pathology and Laboratory Medicine, The Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Tamas KoziczDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA; Department of Biochemistry and Molecular Biology, Mayo Clinic, Rochester, MN, USA; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, USA; University of Pécs, Medical School, Pécs, Hungary.
Ruqaiah AltassanDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA; Department of Medical Genomics, Centre for Genomics Medicine, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia; College of Medicine, Alfaisal University, Riyadh, Saudi Arabia. Electronic address: raltassan@kfshrc.edu.sa.
Eva MoravaDepartment of Clinical Genomics, Mayo Clinic, Rochester, MN 55905, USA; Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, USA; University of Pécs, Medical School, Pécs, Hungary. Electronic address: Morava-Kozicz.Eva@mayo.edu.

Funding

Pilot and Feasibility CoreU54NS115198 · NINDS · MAYO CLINIC ROCHESTER · PI MORAVA-KOZICZ, EVA · 2019 to 2023
$8.2M
New Congenital Disorders of Glycosylation: Therapy and ModelsR01DK099551 · NIDDK · SANFORD BURNHAM PREBYS MEDICAL DISCOVERY INSTITUTE · PI Hudson H. Freeze · 2014 to 2026
$6.7M
NINDS NIH HHS U54 NS115198
6 · The paper itself

Abstract

ALG13-Congenital Disorder of Glycosylation (CDG), is a rare X-linked CDG caused by pathogenic variants in ALG13 (OMIM 300776) that affects the N-linked glycosylation pathway. Affected individuals present with a predominantly neurological manifestation during infancy. Epileptic spasms are a common presenting symptom of ALG13-CDG. Other common phenotypes include developmental delay, seizures, intellectual disability, microcephaly, and hypotonia. Current management of ALG13-CDG is targeted to address patients' symptoms. To date, less than 100 individuals have been reported with ALG13-CDG. In this article, an international group of experts in CDG reviewed all reported individuals affected with ALG13-CDG and suggested diagnostic and management guidelines for ALG13-CDG. The guidelines are based on the best available data and expert opinion. Neurological symptoms dominate the phenotype of ALG13-CDG where epileptic spasm is confirmed to be the most common presenting symptom of ALG13-CDG in association with hypotonia and developmental delay. We propose that ACTH/prednisolone treatment should be trialed first, followed by vigabatrin, however ketogenic diet has been shown to have promising results in ALG13-CDG. In order to optimize medical management, we also suggest early cardiac, gastrointestinal, skeletal, and behavioral assessments in affected patients.

Indexed as

Congenital Disorders of GlycosylationDevelopmental DisabilitiesGlycosylationHumansInfantIntellectual DisabilityMuscle HypotoniaMutationN-AcetylglucosaminyltransferasesPhenotypePractice Guidelines as TopicSeizuresALG13 protein, humanN-AcetylglucosaminyltransferasesALG13-CDGCongenital disorders of glycosylationEpileptic spasmSeizure disorderX-linked CDG

Identifiers

PMID38703411
PMCPMC11402470

What Socratic holds

Textmetadata
LicenceTDM
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 Socratic graph.