Enpp1 inhibition: a promising oral therapy for later-onset hypophosphatasia

Hypophosphatasia (HPP) is a rare, inherited metabolic bone disorder characterized by deficient activity of tissue-nonspecific alkaline phosphatase (TNAP), an enzyme encoded by the ALPL gene. Loss-of-function mutations in ALPL lead to reduced TNAP activity, resulting in the accumulation of extracellular inorganic pyrophosphate (PPi), a potent inhibitor of hydroxyapatite crystal formation and skeletal mineralization. Clinically, this presents as a broad spectrum of dento-osseous complications, including premature tooth loss, rickets or osteomalacia, and calcific arthropathies.

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Publication Details

Journal
Journal of Bone and Mineral Research
Published
2025-10-23
DOI
https://doi.org/10.1093/jbmr/zjaf154
Primary Topic
Alkaline Phosphatase Research Studies
Type
article
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article

Enpp1 inhibition: a promising oral therapy for later-onset hypophosphatasia

Vicky Elizabeth MacRae
Journal of Bone and Mineral Research
Alkaline Phosphatase Research Studies
article

Enpp1 inhibition: a promising oral therapy for later-onset hypophosphatasia

Vicky Elizabeth MacRae
article en

Abstract

Hypophosphatasia (HPP) is a rare, inherited metabolic bone disorder characterized by deficient activity of tissue-nonspecific alkaline phosphatase (TNAP), an enzyme encoded by the ALPL gene. Loss-of-function mutations in ALPL lead to reduced TNAP activity, resulting in the accumulation of extracellular inorganic pyrophosphate (PPi), a potent inhibitor of hydroxyapatite crystal formation and skeletal mineralization. Clinically, this presents as a broad spectrum of dento-osseous complications, including premature tooth loss, rickets or osteomalacia, and calcific arthropathies.

Journal of Bone and Mineral ResearchVol. 41(3)
Roslin Institute (GB), Anglia Ruskin University (GB)
Openalex Percentile: Top 58%
Alkaline Phosphatase Research Studies
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