Tsang R C, Venkataraman P, Ho M, Steichen J J, Whitsett J, Greer F
Am J Dis Child. 1984 Jul;138(7):654-8. doi: 10.1001/archpedi.1984.02140450036011.
The hormonal changes in the development of pseudohypoparathyroidism ( PSH ) have not, to our knowledge, been previously reported. The male sibling of a child with PSH was studied for 2 1/2 years. At 1 year of age he had generalized subcutaneous calcifications that subsequently migrated over his body. At 3 years of age and over a six-month period, serum calcium levels fell; serum phosphorus, parathyroid hormone (PTH), and 1,25-dihydroxyvitamin D (1,25-[OH]2D) concentrations increased. There was no calcemic, phosphaturic, or urinary cyclic adenosine monophosphate response to PTH. The concentration of serum PTH was suppressed by infusion of calcium and doubled with edetic acid infusion, indicating that the parathyroids were sensitive to changes in calcium levels. Thus, increasing PTH and increased 1,25-(OH)2D concentrations occur in the development of PSH . Migratory skin calcifications may occur. We speculate that increasing the serum PTH level reflects increasing compensatory parathyroid production to overcome a progressive PTH receptor defect and serves, with increased 1,25-(OH)2D concentrations, to prevent severe falls in serum calcium concentrations in the early stage of the disease.
据我们所知,此前尚未有关于假性甲状旁腺功能减退症(PSH)发展过程中激素变化的报道。对一名患有PSH儿童的男性同胞进行了为期两年半的研究。1岁时,他全身出现皮下钙化,随后钙化在全身迁移。3岁时,在六个月的时间里,血清钙水平下降;血清磷、甲状旁腺激素(PTH)和1,25-二羟维生素D(1,25-[OH]2D)浓度升高。对PTH没有血钙、尿磷或尿中环磷酸腺苷反应。静脉输注钙可抑制血清PTH浓度,而输注依地酸则使其加倍,这表明甲状旁腺对钙水平的变化敏感。因此,在PSH的发展过程中会出现PTH升高和1,25-(OH)2D浓度增加的情况。可能会出现迁移性皮肤钙化。我们推测,血清PTH水平升高反映了甲状旁腺代偿性分泌增加,以克服逐渐发展的PTH受体缺陷,并与升高的1,25-(OH)2D浓度一起,在疾病早期防止血清钙浓度严重下降。