ArticleInternational journal of molecular medicine2015
Effects of etidronate on the Enpp1⁻/⁻ mouse model of generalized arterial calcification of infancy.
Article in International journal of molecular medicine, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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Who cites it
15 citing papers in PubMed, 19 citations in OpenAlex.
- A Fragile Phosphate/Pyrophosphate Balance: From Essential Mineralization to Rare Calcifying Diseases.Current osteoporosis reports · 2025Review
- Therapeutic approaches for the treatment of genetic and acquired cardiovascular calcification.Frontiers in cardiovascular medicine · 2025Review
- Calcification of the elastic component: the impact on the cardiovascular system.Frontiers in cardiovascular medicine · 2025Review
- Increased bone mass but delayed mineralization: in vivo and in vitro study for zoledronate in bone regeneration.BMC oral health · 2024Article
- Longitudinal assessment of vascular calcification in generalized arterial calcification of infancy.Pediatric radiology · 2022Review
- ABCC6, Pyrophosphate and Ectopic Calcification: Therapeutic Solutions.International journal of molecular sciences · 2021Review
- Mitochondrial Dysfunction: Cause or Consequence of Vascular Calcification?Frontiers in cell and developmental biology · 2021Review
- Therapeutics Development for Pseudoxanthoma Elasticum and Related Ectopic Mineralization Disorders: Update 2020.Journal of clinical medicine · 2020Review
- The membrane protein ANKH is crucial for bone mechanical performance by mediating cellular export of citrate and ATP.PLoS genetics · 2020Article
- X-ray Micro-Computed Tomography: An Emerging Technology to Analyze Vascular Calcification in Animal Models.International journal of molecular sciences · 2020Review
- Novel homozygous ENPP1 mutation causes generalized arterial calcifications of infancy, thrombocytopenia, and cardiovascular and central nervous system syndrome.American journal of medical genetics. Part A · 2019Article
- ENPP1-Fc prevents neointima formation in generalized arterial calcification of infancy through the generation of AMP.Experimental & molecular medicine · 2018Article
- ENPP1 enzyme replacement therapy improves blood pressure and cardiovascular function in a mouse model of generalized arterial calcification of infancy.Disease models & mechanisms · 2018Article
- Ectopic calcification in pseudoxanthoma elasticum responds to inhibition of tissue-nonspecific alkaline phosphatase.Science translational medicine · 2017Article
- Large animal models of cardiovascular disease.Cell biochemistry and function · 2016Review
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Authors and funding
4 authors at 3 institutions in 2 countries.
Funding
Abstract
Generalized arterial calcification of infancy (GACI) is an autosomal recessive disorder of spontaneous infantile arterial and periarticular calcification which is attributed to mutations in the ectonucleotide pyrophosphatase/phosphodiesterase 1 (Enpp1) gene. Whilst the bisphosphonate, etidronate, is currently used off-label for the treatment for GACI, recent studies have highlighted its detrimental effects on bone mineralisation. In the present study, we used the Enpp1-/- mouse model of GACI to examine the effects of etidronate treatment (100 µg/kg), on vascular and skeletal calcification. Micro-computed tomography (µCT) analysis revealed a significant decrease in trabecular bone mass, as reflected by the decrease in trabecular bone volume/tissue volume (BV/TV; %), trabecular thickness, trabecular separation, trabecular number and pattern factor (P<0.05) in the Enpp1-/- mice in comparison to the wild-type (WT) mice. Mechanical testing revealed that in the WT mice, treatment with etidronate significantly improved work to fracture and increased work post-failure (P<0.05, in comparison to the vehicle-treated WT mice). This significant increase, however, was not observed in the Enpp1-/- mice. Treatment with etidronate had no effect on bone parameters in the WT mice; however, the Enpp1-/- mice displayed an increased structural model index (SMI; P<0.05). We used a recently developed 3D µCT protocol to reconstruct and quantify the extensive aortic calcification in Enpp1-/- mice in comparison to the WT mice. However, treatment with etidronate did not prevent de novo calcification, and did not arrest the progression of established calcification of the aorta.
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