Evidence map›Paper›PMID 34573906›Full record

ArticleDiagnostics (Basel, Switzerland)2021

Glycosaminoglycans as Biomarkers for Mucopolysaccharidoses and Other Disorders.

Paige C Amendum, Shaukat Khan, Seiji Yamaguchi, Hironori Kobayashi, Yasuhiko Ago, Yasuyuki Suzuki, Betul Celik, Estera Rintz, Jobayer Hossain, Wendi Xiao and 1 more

Open access · goldAbstract read
In one paragraph

Article in Diagnostics (Basel, Switzerland), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
0.8field-weighted citation impact, top 28% 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

6 citing papers in PubMed, 9 citations in OpenAlex.

  1. Article
  2. Review
  3. Detection of Glycosaminoglycans in Biological Specimens.Methods in molecular biology (Clifton, N.J.) · 2023
    Article
  4. Review
  5. Review
  6. MPSI Manifestations and Treatment Outcome: Skeletal Focus.International journal of molecular sciences · 2022
    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

11 authors at 3 institutions in 2 countries.

Paige C AmendumDepartment of Biological Sciences, University of Delaware, Newark, DE 19716, USA.
Shaukat KhanDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.
Seiji YamaguchiDepartment of Pediatrics, Shimane University, Izumo 693-8501, Japan.
Hironori KobayashiDepartment of Pediatrics, Shimane University, Izumo 693-8501, Japan.
Yasuhiko AgoDepartment of Pediatrics, Graduate School of Medicine, Gifu University, Gifu 501-1193, Japan.
Yasuyuki SuzukiMedical Education Development Center, Graduate School of Medicine, Gifu University, Gifu 501-1193, Japan.ORCID 0000-0003-1281-2834
Betul CelikDepartment of Biological Sciences, University of Delaware, Newark, DE 19716, USA.ORCID 0000-0002-0379-8076
Estera RintzDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.
Jobayer HossainDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.ORCID 0000-0003-2609-6605
Wendi XiaoDepartment of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.
Shunji TomatsuDepartment of Biological Sciences, University of Delaware, Newark, DE 19716, USA.ORCID 0000-0002-0673-2160
Alfred I. duPont Hospital for Children · USGifu University · JPShimane University · JP

Funding

Center for Pediatric Research Pilot ProgramP30GM114736 · NIGMS · NEMOURS CHILDREN'S HOSPITAL, DELAWARE · PI SHAFFER, THOMAS H · 2015 to 2019
$5.1M
Non-invasive functional assessment and pathogenesis of Morquio AR01HD102545 · NICHD · NEMOURS CHILDREN'S HOSPITAL, DELAWARE · PI TOMATSU, SHUNJI · 2021 to 2025
$2.9M
Newborn Screening and Biomarkers for MucopolysaccharidosesR01HD065767 · NICHD · SAINT LOUIS UNIVERSITY · PI MONTANO, ADRIANA MARIA, TOMATSU, SHUNJI · 2011 to 2015
$2.4M
NICHD NIH HHS R01 HD065767NICHD NIH HHS R01 HD102545NIGMS NIH HHS P30 GM114736NIH HHS 1R01HD065767NIH HHS 1R01HD102545-01A1NIH HHS P30GM114736
6 · The paper itself

Abstract

Glycosaminoglycans (GAGs) are present in proteoglycans, which play critical physiological roles in various tissues. They are known to be elevated in mucopolysaccharidoses (MPS), a group of rare inherited metabolic diseases in which the lysosomal enzyme required to break down one or more GAG is deficient. In a previous study, we found elevation of GAGs in a subset of patients without MPS. In the current study, we aim to investigate serum GAG levels in patients with conditions beyond MPS. In our investigated samples, the largest group of patients had a clinical diagnosis of viral or non-viral encephalopathy. Clinical diagnoses and conditions also included epilepsy, fatty acid metabolism disorders, respiratory and renal disorders, liver disorders, hypoglycemia, developmental disorders, hyperCKemia, myopathy, acidosis, and vomiting disorders. While there was no conclusive evidence across all ages for any disease, serum GAG levels were elevated in patients with encephalopathy and some patients with other conditions. These preliminary findings suggest that serum GAGs are potential biomarkers in MPS and other disorders. In conclusion, we propose that GAGs elevated in blood can be used as biomarkers in the diagnosis and prognosis of various diseases in childhood; however, further designed experiments with larger sample sizes are required.

Indexed as

encephalopathyglycosaminoglycanLC-MS/MSmucopolysaccharidosesserum

Identifiers

PMID34573906
PMCPMC8468223
OpenAlexW3197694632

What OpenQuestion holds

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LicenceCC BY
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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.