Lesch-Nyhan disease: I. Construction of expression vectors for hypoxanthine-guanine phosphoribosyltransferase (HGprt) enzyme and amyloid precursor protein (APP).


Journal

Nucleosides, nucleotides & nucleic acids
ISSN: 1532-2335
Titre abrégé: Nucleosides Nucleotides Nucleic Acids
Pays: United States
ID NLM: 100892832

Informations de publication

Date de publication:
2020
Historique:
pubmed: 22 4 2020
medline: 19 12 2020
entrez: 22 4 2020
Statut: ppublish

Résumé

Lesch-Nyhan disease (LND) is a rare X-linked inherited neurogenetic disorder of purine metabolism in which the enzyme, hypoxanthine-guanine phosphoribosyltransferase (HGprt) is defective. Despite having been characterized over 50 years ago, it remains unclear precisely how deficits in HGprt enzyme activity can lead to the neurological syndrome, especially the self-injury of LND. Several studies have proposed different hypotheses regarding the etiology of this disease, and several treatments have been tried in patients. However, up to now, there is no satisfactory explanation of the disease and for many LND patients, efficacious treatment for persistent self-injurious behavior remains unreachable. A role for epistasis between mutated hypoxanthine phosphoribosyltransferase 1 (

Identifiants

pubmed: 32312153
doi: 10.1080/15257770.2020.1714653
doi:

Substances chimiques

Amyloid beta-Protein Precursor 0
Purines 0
Reduced Folate Carrier Protein 0
SLC19A1 protein, human 0
Hypoxanthine Phosphoribosyltransferase EC 2.4.2.8
purine W60KTZ3IZY

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

905-922

Auteurs

Khue Vu Nguyen (KV)

Department of Medicine, Biochemical Genetics and Metabolism, The Mitochondrial and Metabolic Disease Center, School of Medicine, University of California, San Diego, San Diego, California, USA.
Department of Pediatrics, School of Medicine, University of California, San Diego, California, USA.

Robert K Naviaux (RK)

Department of Medicine, Biochemical Genetics and Metabolism, The Mitochondrial and Metabolic Disease Center, School of Medicine, University of California, San Diego, San Diego, California, USA.
Department of Pediatrics, School of Medicine, University of California, San Diego, California, USA.
Department of Pathology, School of Medicine, University of California, San Diego, California, USA.

William L Nyhan (WL)

Department of Pediatrics, School of Medicine, University of California, San Diego, California, USA.

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Classifications MeSH