Particle disease: a unique cause of hypercalcemia
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CASE REPORT
Particle disease: a unique cause of hypercalcemia J. Zhang 1 & D.E. Sellmeyer 1 Received: 22 May 2020 / Accepted: 1 September 2020 # International Osteoporosis Foundation and National Osteoporosis Foundation 2020
Abstract 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. Keywords 1,25-Dihydroxyvitamin D . Granulomatous disease . Hypercalcemia . Particle disease
Introduction Hypercalcemia due to increased 1,25-dihydroxyvitamin D (1,25-(OH)2 D) production can be seen in a variety of granulomatous and neoplastic conditions. The most well-described of these has been in sarcoidosis, where the prevalence of hypercalcemia has been reported to be up to 5–10% [1, 2]. Granulomatous reactions to infections or to foreign body materials have also been reported to cause hypercalcemia [2]. We present a unique cause of 1,25-(OH)2 D-mediated hypercalcemia due to foreign body granuloma associated with particle disease in a failed prosthetic hip.
Case history A 72-year-old female presented to her primary care physician with 1 week of left lower quadrant abdominal pain and nausea. The patient had a left hip total arthroplasty for osteoarthritis 23 years prior to presentation. She had been * D.E. Sellmeyer [email protected] 1
Division of Endocrinology, Gerontology and Metabolism, Stanford University School of Medicine, Stanford, CA 94305, USA
experiencing progressively worsening left hip pain over several months. CT abdomen and pelvis revealed a 10-cm enhancing soft tissue mass, concerning for sarcoma per the radiologist’s impression, which was located adjacent to a failed left hip prosthesis (Fig. 1). This was associated with enlarged peri-aortic and iliac chain lymph nodes. Initial laboratory testing demonstrated serum calcium 12.9 mg/dL (normal 8.6– 10.4 mg/dL), 25-hydroxyvitamin D (25-OHD) 27 ng/mL (normal > 30 ng/mL), PTH-related peptide 0.6 pmol/L (normal < 2.0 pmol/L), and serum
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