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Endocrine Abstracts (2018) 57 025 | DOI: 10.1530/endoabs.57.025

BES2018 BES 2018 Rare cause of 1,25-dihydroxy vitamin D mediated hypercalcemia: A case report and literature study (1 abstracts)

Rare cause of 1,25-dihydroxy vitamin D mediated hypercalcemia: A case report and literature study

J de Bellefroid 1, , A Van den Bruel 1 & S Vandecasteele 2


1Department of Internal Medicine, Endocrinology, AZ Sint Jan Bruges, Belgium; 2Department of Internal Medicine, Nephrology, AZ Sint Jan Bruges, Belgium.


Background: 1,25-dihydroxy vitamin D mediated hypercalcemia resulting from an inflammatory reaction caused by a foreign body such as a textiloma has never been described.

Clinical case: A 61 year-old man with a medical history of Alport syndrome and in need of chronic dialysis after transplant kidney resection because of relapsing severe urinary tract infections, presented with recurrent and progressive hypercalcemia two years after parathyroidectomy (C and B) because of tertiary hyperparathyroidism. There was no intake of calcium supplements or diuretics. Laboratory evaluations showed a normal 25-hydroxyvitamin D but an elevated 1,25-dihydroxyvitamin D and an inadequately normal PTH (Table 1). A 99mTc parathyroid scintigraphy could not show any adenoma. In order to localize a possible inflammatory process, an FDG-PET-CT was performed and revealed an abdominal mass with a high metabolic captive border. Four days later the patient arrived at dialysis and showed signs of fatigue. Inflammatory values were high. He was admitted to the hospital and empirical antibiotics were started. Despite the antibiotics, inflammatory parameters raised and the patient complained of progressive abdominal pain. A diagnostic laparotomy was performed and revealed a textiloma (a surgical gauze). After removal of the textiloma a complete normalization of the hypercalcemia and 1,25-dihydroxyvitamin D was observed.

Table 1 Laboratory results.
LabValue Value after surgeryReference value
Calcium2.88 mmol/l2.50 mmol/l2.2–2.55 mmol/l
Phosphorous1.80 mmol/l1.08 mmol/l0.81–1.45 mmol/l
PTH23 ng/l218 ng/l15–65 ng/l
25-hydroxyvitamin D47 ng/ml53.3 ng/ml>30 ng/ml
1,25-dihydroxyvitamin D172,6 pmol/l45.9 pmol/l<5–42 pmol/l
Serum creatinine13.6 mg/dl8.8 mg/dl0.6–1.2 mg/dl

Conclusion: Hypercalcemia mediated by 1,25-dihydroxy vitamin D is rather uncommon. Well-recognized etiologies are sarcoidosis and lymphomas. Less than 10 case reports were found where the underlying cause was a foreign body exposure, no case was found where the foreign body was a textiloma. The underlying mechanism remains unclear, but the literature mainly suggests that a foreign body reaction results in formation of foreign body granulomas. Activated macrophages express 1-α hydroxylase which transforms 25-hydroxyvitamin D into 1,25-dihydroxyvitamin D, leading to hypercalcemia.

References: 1. A. a. S.-Y. Ren, “Autoregulation of 1,25-dihydroxyvitamin D synthesis in macrophage mitochondria by nitric oxide’, The Endocrine Society, pp. 4514–4517, 1996.

2. M. Kallas, “Rare causes of calcitriol-mediated hypercalcemia: a case report and literature review”, J Cllin Endocrinol Metab, pp. 3111–3117, 2010.

3. P. J. Donovan, “Calcitriol-mediated hypercalcemia: causes and course in 101 patients”, J Clin Endocrinol Metab, pp. 4023–4029, 2013.

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