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  • Title: A case of severe 1,25-dihydroxyvitamin D-mediated hypercalcemia due to a granulomatous disorder.
    Author: Zhang JT, Chan C, Kwun SY, Benson KA.
    Journal: J Clin Endocrinol Metab; 2012 Aug; 97(8):2579-83. PubMed ID: 22639294.
    Abstract:
    BACKGROUND: Profound hypercalcemia is usually due to underlying malignancy. CASE: We describe a case of granulomatous myositis presenting with extreme hypercalcemia of 20.1 mg/dl and generalized weakness that did not resolve despite rapid correction of serum calcium. The disease process was unmasked by cholecalciferol supplementation. Initial search for a malignant process yielded no diagnosis, but an elevated 1,25-dihydroxyvitamin D level, in the setting of a suppressed PTH and undetectable PTHrP, pointed to the presence of excessive 1α-hydroxylase activity. METHODS AND RESULTS: Biopsy of the vastus lateralis muscle showed extensive granulomatous myositis. Immunohistochemical staining for 1α-hydroxylase was localized to the multinucleated giant cells and histiocytes. Musculoskeletal magnetic resonance imaging showed involvement of proximal muscle groups of both thighs and upper limbs. CONCLUSION: Measurement of vitamin D metabolites is pivotal in diagnosing 1,25-dihydroxyvitamin D-mediated hypercalcemia. Granulomatous disease can occasionally cause profound hypercalcemia and needs to be considered in the differential diagnosis. 1,25-Dihydroxyvitamin D-mediated hypercalcemia is responsive to glucocorticoid therapy.
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