Glycosaminoglycans as Biomarkers for Mucopolysaccharidoses and Other Disorders.

LC-MS/MS encephalopathy glycosaminoglycan mucopolysaccharidoses serum

Journal

Diagnostics (Basel, Switzerland)
ISSN: 2075-4418
Titre abrégé: Diagnostics (Basel)
Pays: Switzerland
ID NLM: 101658402

Informations de publication

Date de publication:
28 Aug 2021
Historique:
received: 04 06 2021
revised: 23 08 2021
accepted: 24 08 2021
entrez: 28 9 2021
pubmed: 29 9 2021
medline: 29 9 2021
Statut: epublish

Résumé

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.

Identifiants

pubmed: 34573906
pii: diagnostics11091563
doi: 10.3390/diagnostics11091563
pmc: PMC8468223
pii:
doi:

Types de publication

Journal Article

Langues

eng

Subventions

Organisme : NIH HHS
ID : 1R01HD065767
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD102545
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD065767
Pays : United States
Organisme : NIGMS NIH HHS
ID : P30 GM114736
Pays : United States
Organisme : NIH HHS
ID : 1R01HD102545-01A1
Pays : United States
Organisme : NIH HHS
ID : P30GM114736
Pays : United States

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Auteurs

Paige C Amendum (PC)

Department of Biological Sciences, University of Delaware, Newark, DE 19716, USA.
Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Shaukat Khan (S)

Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Seiji Yamaguchi (S)

Department of Pediatrics, Shimane University, Izumo 693-8501, Japan.

Hironori Kobayashi (H)

Department of Pediatrics, Shimane University, Izumo 693-8501, Japan.

Yasuhiko Ago (Y)

Department of Pediatrics, Graduate School of Medicine, Gifu University, Gifu 501-1193, Japan.

Yasuyuki Suzuki (Y)

Medical Education Development Center, Graduate School of Medicine, Gifu University, Gifu 501-1193, Japan.

Betul Celik (B)

Department of Biological Sciences, University of Delaware, Newark, DE 19716, USA.
Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Estera Rintz (E)

Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Jobayer Hossain (J)

Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Wendi Xiao (W)

Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.

Shunji Tomatsu (S)

Department of Biological Sciences, University of Delaware, Newark, DE 19716, USA.
Department of Biomedical Research, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE 19803, USA.
Department of Pediatrics, Shimane University, Izumo 693-8501, Japan.
Department of Pediatrics, Graduate School of Medicine, Gifu University, Gifu 501-1193, Japan.
Department of Pediatrics, Thomas Jefferson University, Philadelphia, PA 19107, USA.

Classifications MeSH