Ichikawa Shoji, Gray Amie K, Padgett Leah R, Reilly Austin M, Unsicker Tyler R
Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA.
J Bone Miner Res. 2014 Sep;29(9):2017-23. doi: 10.1002/jbmr.2242.
Familial tumoral calcinosis is characterized by ectopic calcifications due to persistent hyperphosphatemia. The most common genetic cause of the disease is mutations in GALNT3, encoding a glycosyltransferase involved in a posttranslational modification of fibroblast growth factor 23 (FGF23). The Galnt3 knockout mouse we developed was hyperphosphatemic due to low intact Fgf23 levels, but did not develop any apparent calcifications on a standard rodent diet. We therefore tested the hypothesis that a further challenge with a high phosphate diet could induce ectopic calcifications in Galnt3 knockout mice. Mice were fed either normal (0.6%) or high (1.65%) phosphate diet for 20 weeks beginning from weaning at 3 weeks. The high phosphate diet did not affect serum phosphorus concentration. However, regardless of the dietary phosphate contents, serum phosphorus levels were consistently elevated in Galnt3 knockout mice. The mice on the high phosphate diet had slightly low serum calcium, but significantly high alkaline phosphatase, parathyroid hormone (PTH), and calcium in the kidney. Although none of Galnt3 knockout mice on the normal phosphate diet developed calcifications, calcifications appeared in approximately one-half of the mice on the high phosphate diet by 12 weeks. Calcified masses were most often found around the neck and on the back and as large as 9.9 mm in length. These data indicate that dietary phosphate load has major impact on the development of ectopic calcifications in tumoral calcinosis.
家族性肿瘤性钙化症的特征是由于持续性高磷血症导致异位钙化。该疾病最常见的遗传原因是GALNT3基因突变,GALNT3编码一种参与成纤维细胞生长因子23(FGF23)翻译后修饰的糖基转移酶。我们培育的Galnt3基因敲除小鼠由于完整Fgf23水平低而出现高磷血症,但在标准啮齿动物饮食条件下未出现任何明显的钙化。因此,我们检验了这样一个假设,即高磷饮食的进一步刺激可能会在Galnt3基因敲除小鼠中诱导异位钙化。从3周龄断奶开始,小鼠被喂食正常(0.6%)或高(1.65%)磷饮食20周。高磷饮食不影响血清磷浓度。然而,无论饮食中的磷含量如何,Galnt3基因敲除小鼠的血清磷水平持续升高。食用高磷饮食的小鼠血清钙略低,但肾脏中的碱性磷酸酶、甲状旁腺激素(PTH)和钙显著升高。虽然食用正常磷饮食的Galnt3基因敲除小鼠均未出现钙化,但到12周时,食用高磷饮食的小鼠中约有一半出现了钙化。钙化团块最常出现在颈部周围和背部,长度可达9.9毫米。这些数据表明,饮食中的磷负荷对肿瘤性钙化症中异位钙化的发展有重大影响。