Razali Nurul Nadirah, Hwu Ting Tzer, Thilakavathy Karuppiah
J Pediatr Endocrinol Metab. 2015 Sep;28(9-10):1009-17. doi: 10.1515/jpem-2014-0366.
Hypophosphatemic rickets (HR) is a syndrome of hypophosphatemia and rickets that resembles vitamin D deficiency, which is caused by malfunction of renal tubules in phosphate reabsorption. Phosphate is an essential mineral, which is important for bone and tooth structure. It is regulated by parathyroid hormone, 1,25-dihydroxyvitamin D and fibroblast-growth-factor 23 (FGF23). X-linked hypophosphatemia (XLH), autosomal dominant HR (ADHR), and autosomal recessive HR (ARHR) are examples of hereditary forms of HR, which are mainly caused by mutations in the phosphate regulating endopeptidase homolog, X-linked (PHEX), FGF23, and, dentin matrix protein-1 (DMP1) and ecto-nucleotide pyro phosphatase/phosphodiesterase 1 (ENPP1) genes, respectively. Mutations in these genes are believed to cause elevation of circulating FGF23 protein. Increase in FGF23 disrupts phosphate homeostasis, leading to HR. This review aims to summarize phosphate homeostasis and focuses on the genes and mutations related to XLH, ADHR, and ARHR. A compilation of XLH mutation hotspots based on the PHEX gene database and mutations found in the FGF23, DMP1, and ENPP1 genes are also made available in this review.
低磷性佝偻病(HR)是一种低磷血症和佝偻病综合征,类似于维生素D缺乏症,由肾小管磷酸盐重吸收功能障碍引起。磷酸盐是一种必需矿物质,对骨骼和牙齿结构很重要。它受甲状旁腺激素、1,25-二羟基维生素D和成纤维细胞生长因子23(FGF23)调节。X连锁低磷血症(XLH)、常染色体显性HR(ADHR)和常染色体隐性HR(ARHR)是HR的遗传性形式的例子,它们分别主要由磷酸盐调节内肽酶同源物X连锁(PHEX)、FGF23、牙本质基质蛋白1(DMP1)和胞外核苷酸焦磷酸酶/磷酸二酯酶1(ENPP1)基因的突变引起。这些基因的突变被认为会导致循环FGF23蛋白升高。FGF23增加会破坏磷酸盐稳态,导致HR。本综述旨在总结磷酸盐稳态,并重点关注与XLH、ADHR和ARHR相关的基因和突变。本综述还提供了基于PHEX基因数据库的XLH突变热点汇编以及在FGF23、DMP1和ENPP1基因中发现的突变。