ArticlePloS one2014
Dysregulated gene expression in the primary osteoblasts and osteocytes isolated from hypophosphatemic Hyp mice.
Article in PloS one, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers.
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Who cites it
33 citing papers in PubMed, 56 citations in OpenAlex.
- A bone fragment-based protocol for RNA analysis of osteocyte-associated transcripts in surgically obtained human bone specimens.MethodsX · 2026Article
- Fibroblast growth factor 23: Regulation, signalling and systemic links between bone, metabolism and inflammation.Molecular metabolism · 2026Review
- Current advances of bone homeostasis imbalance in the cause of hereditary metabolic bone diseases.EFORT open reviews · 2026Review
- Genetic reduction or deletion of osteopontin in osteomalacicJBMR plus · 2025Article
- Emerging concepts on the FGF23 regulation and activity.Molecular and cellular biochemistry · 2025Review
- MyD88 in osteoclast and osteoblast lineages differentially controls bone remodeling in homeostasis and malaria.International immunology · 2024Article
- X-linked hypophosphatemia, fibroblast growth factor 23 signaling, and craniosynostosis.Experimental biology and medicine (Maywood, N.J.) · 2023Review
- Osteoclast-derived IGF1 induces RANKL production in osteocytes and contributes to pagetic lesion formation.JCI insight · 2023Article
- Hypochondroplasia gain-of-function mutation in FGFR3 causes defective bone mineralization in mice.JCI insight · 2023Article
- Meclozine ameliorates bone mineralization and growth plate structure in a mouse model of X‑linked hypophosphatemia.Experimental and therapeutic medicine · 2023Article
- Single-cell RNA sequencing identifiesFrontiers in physiology · 2023Article
- Paracrine and endocrine functions of osteocytes.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2023Review
- Calcium‑dependent activation of PHEX, MEPE and DMP1 in osteocytes.Molecular medicine reports · 2022Article
- Spatial metabolomics reveals upregulation of several pyrophosphate-producing pathways in cortical bone of Hyp mice.JCI insight · 2022Article
- Parathyroid Hormone Regulates Circulating Levels of Sclerostin and FGF23 in a Primary Hyperparathyroidism Model.Journal of the Endocrine Society · 2022Article
- Roles of osteocytes in phosphate metabolism.Frontiers in endocrinology · 2022Review
- Osteocytes and the pathogenesis of hypophosphatemic rickets.Frontiers in endocrinology · 2022Review
- Energy Metabolism of Osteocytes.Current osteoporosis reports · 2021Review
- Osteocytes as main responders to low-intensity pulsed ultrasound treatment during fracture healing.Scientific reports · 2021Article
- Osteocyte Vegf-a contributes to myeloma-associated angiogenesis and is regulated by Fgf23.Scientific reports · 2020Article
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Authors and funding
11 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Osteocytes express multiple genes involved in mineral metabolism including PHEX, FGF23, DMP1 and FAM20C. In Hyp mice, a murine model for X-linked hypophosphatemia (XLH), Phex deficiency results in the overproduction of FGF23 in osteocytes, which leads to hypophosphatemia and impaired vitamin D metabolism. In this study, to further clarify the abnormality in osteocytes of Hyp mice, we obtained detailed gene expression profiles in osteoblasts and osteocytes isolated from the long bones of 20-week-old Hyp mice and wild-type (WT) control mice. The expression of Fgf23, Dmp1, and Fam20c was higher in osteocytic cells than in osteoblastic cells in both genotypes, and was up-regulated in Hyp cells. Interestingly, the up-regulation of these genes in Hyp bones began before birth. On the other hand, the expression of Slc20a1 encoding the sodium/phosphate (Na+/Pi) co-transporter Pit1 was increased in osteoblasts and osteocytes from adult Hyp mice, but not in Hyp fetal bones. The direct effects of extracellular Pi and 1,25-dihydroxyvitamin D3 [1,25(OH)2D3] on isolated osteoblastic and osteocytic cells were also investigated. Twenty-four-hour treatment with 10-8 M 1,25(OH)2D3 increased the expression of Fgf23 in WT osteoblastic cells but not in osteocytic cells. Dmp1 expression in osteocytic cells was increased due to the 24-hour treatment with 10 mM Pi and was suppressed by 10-8 M 1,25(OH)2D3 in WT osteocytic cells. We also found the up-regulation of the genes for FGF1, FGF2, their receptors, and Egr-1 which is a target of FGF signaling, in Hyp osteocytic cells, suggesting the activation of FGF/FGFR signaling. These results implicate the complex gene dysregulation in osteoblasts and osteocytes of Hyp mice, which might contribute to the pathogenesis.
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