Hypophosphatasia: Vitamin B

4-Pyridoxic acid Alkaline phosphatase Cerebrospinal fluid Enzymopathy Hypervitaminosis B(6) Hypovitaminosis B(6) Inborn error Inorganic pyrophosphate L-tryptophan challenge Metabolic bone disease Neuropathy Osteomalacia Pyridoxal Pyridoxal 5′-phosphate Pyridoxamine Pyridoxine Rickets Vitamin B(6)-dependent seizures

Journal

Bone
ISSN: 1873-2763
Titre abrégé: Bone
Pays: United States
ID NLM: 8504048

Informations de publication

Date de publication:
01 2022
Historique:
received: 08 06 2021
revised: 09 09 2021
accepted: 14 09 2021
pubmed: 22 9 2021
medline: 12 3 2022
entrez: 21 9 2021
Statut: ppublish

Résumé

Hypophosphatasia (HPP) is the heritable dento-osseous disease caused by loss-of-function mutation(s) of the gene ALPL that encodes the tissue-nonspecific isoenzyme of alkaline phosphatase (TNSALP). TNSALP is a cell-surface homodimeric phosphomonoester phosphohydrolase expressed in healthy people especially in the skeleton, liver, kidneys, and developing teeth. In HPP, diminished TNSALP activity leads to extracellular accumulation of its natural substrates including inorganic pyrophosphate (PPi), an inhibitor of mineralization, and pyridoxal 5'-phosphate (PLP), the principal circulating form of vitamin B

Identifiants

pubmed: 34547524
pii: S8756-3282(21)00370-7
doi: 10.1016/j.bone.2021.116204
pii:
doi:

Substances chimiques

Vitamins 0
Vitamin B 6 8059-24-3
Alkaline Phosphatase EC 3.1.3.1

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

116204

Informations de copyright

Copyright © 2021 Elsevier Inc. All rights reserved.

Auteurs

Michael P Whyte (MP)

Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children - St. Louis, St. Louis, MO 63110, USA; Division of Bone and Mineral Diseases, Department of Internal Medicine, Washington University School of Medicine at Barnes-Jewish Hospital, St. Louis, MO 63110, USA. Electronic address: mwhyte@shrinenet.org.

Fan Zhang (F)

Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children - St. Louis, St. Louis, MO 63110, USA. Electronic address: fzhang@shrinenet.org.

Deborah Wenkert (D)

Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children - St. Louis, St. Louis, MO 63110, USA. Electronic address: wenkert@i1.net.

Karen E Mack (KE)

Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children - St. Louis, St. Louis, MO 63110, USA. Electronic address: kmack@shrinenet.org.

Vinieth N Bijanki (VN)

Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children - St. Louis, St. Louis, MO 63110, USA. Electronic address: vbijanki@shrinenet.org.

Karen L Ericson (KL)

Department of Chemistry, Purdue University Fort Wayne, Fort Wayne, IN 46805, USA. Electronic address: ericsonk@pfw.edu.

Stephen P Coburn (SP)

Department of Chemistry, Purdue University Fort Wayne, Fort Wayne, IN 46805, USA. Electronic address: Coburn@pfw.edu.

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Classifications MeSH