Summary

Hypercalcemia as a result of ectopic 1, 25-dihydroxyvitamin D (1,25-(OH)₂ D) production has been well-described in sarcoidosis and other granulomatous diseases. The 1-alpha-hydroxylase enzyme in activated macrophages is not subject to physiologic regulations, resulting in hypercalcemia with inappropriately normal or elevated 1,25-(OH) ₂ D. Particle disease is the local inflammatory response provoked by an overwhelming production of wear debris from a failed joint prosthesis. Enhanced focal bone resorption in particle disease has been described due to local production of inflammatory cytokines. However, this process previously has not been reported to cause hypercalcemia. We describe a patient with hypercalcemia, low parathyroid hormone levels, and elevated 1,25-(OH)₂ D whose failed prosthetic joint generated a large amount of inflammatory debris, forming a soft tissue mass with lymphadenopathy. Biopsy of the mass demonstrated activated macrophages and foreign body granuloma, resulting in unregulated production of 1,25-(OH)₂ D and hypercalcemia. We present the first case of hypercalcemia associated with elevated 1,25-(OH)₂ D in particle disease due to a failed prosthetic hip.

中文翻译：

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颗粒疾病：高钙血症的独特原因。

概要

由于结节病1，25-二羟基维生素D（1,25-（OH）₂ D）产生而引起的高钙血症已在结节病和其他肉芽肿性疾病中得到了很好的描述。活化的巨噬细胞中的1-α-羟化酶不受生理调节的影响，导致高钙血症，正常或升高的1,25-（OH）₂ D不合适。颗粒疾病是由大量产生的磨损碎片引起的局部炎症反应从失败的关节假体。由于炎症细胞因子的局部产生，已经描述了颗粒疾病中增强的局部骨吸收。但是，以前尚未报道过该过程会引起高钙血症。我们描述了高钙血症，甲状旁腺激素水平低和1,25-（OH）₂升高的患者D假体关节衰竭产生大量炎症碎片，形成软组织肿块并伴有淋巴结肿大。肿块的活检显示巨噬细胞和异物肉芽肿被激活，导致1,25-（OH）₂ D的产生不受调节和高钙血症。我们提出了第一例高钙血症，由于假体髋关节衰竭而导致颗粒疾病中1,25-（OH）₂ D升高。