Comparative metabolomics in the Pah


Journal

Molecular genetics and metabolism
ISSN: 1096-7206
Titre abrégé: Mol Genet Metab
Pays: United States
ID NLM: 9805456

Informations de publication

Date de publication:
05 2022
Historique:
received: 07 03 2022
accepted: 08 03 2022
pubmed: 4 4 2022
medline: 18 5 2022
entrez: 3 4 2022
Statut: ppublish

Résumé

Classical phenylketonuria (PKU, OMIM 261600) owes to hepatic deficiency of phenylalanine hydroxylase (PAH) that enzymatically converts phenylalanine (Phe) to tyrosine (Tyr). PKU neurologic phenotypes include impaired brain development, decreased myelination, early onset mental retardation, seizures, and late-onset features (neuropsychiatric, Parkinsonism). Phe over-representation is systemic; however, tissue response to hyperphenylalaninemia is not consistent. To characterize hyperphenylalaninemia tissue response, metabolomics was applied to Pah

Identifiants

pubmed: 35367142
pii: S1096-7192(22)00155-X
doi: 10.1016/j.ymgme.2022.03.004
pmc: PMC9759961
mid: NIHMS1857163
pii:
doi:

Substances chimiques

Reactive Oxygen Species 0
Phenylalanine 47E5O17Y3R
Phenylalanine Hydroxylase EC 1.14.16.1

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

38-45

Subventions

Organisme : NIDDK NIH HHS
ID : R01 DK090242
Pays : United States
Organisme : NIDDK NIH HHS
ID : R01 DK117916
Pays : United States
Organisme : NICHD NIH HHS
ID : R01 HD103602
Pays : United States

Informations de copyright

Copyright © 2022 Elsevier Inc. All rights reserved.

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Auteurs

Steven F Dobrowolski (SF)

Department of Pathology, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America. Electronic address: dobrowolskis@upmc.edu.

Yu Leng Phua (YL)

Division of Medical Genetics, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Cayla Sudano (C)

Department of Pathology, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Kayla Spridik (K)

Department of Pathology, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Pascal O Zinn (PO)

Department of Neurological Surgery, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Yudong Wang (Y)

Division of Medical Genetics, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Sivakama Bharathi (S)

Division of Medical Genetics, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Jerry Vockley (J)

Division of Medical Genetics, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

Eric Goetzman (E)

Division of Medical Genetics, School of Medicine, University of Pittsburgh, Pittsburgh, PA 15224, United States of America.

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