SUN-785 Silicosis Induced Hypercalcemia Treated With Ketoconazole

Abstract Disclosure: B. AlHamer: None. M. Al Mukaddam: None. A 43-year-old transgender woman with a history of illicit soft tissue filler (ISTF) injections was admitted for hypoxic respiratory failure. Endocrinology was consulted for severe hypercalcemia. She notably underwent ISTF injections around 11 years prior to presentation to the cheek, jawline and buttocks. She later went on to develop severe diffuse parenchymal lung disease (DPLD); this was felt to likely be secondary to silicone emboli as liver biopsies demonstrated lipogranulomas consistent with silicone emboli. Initial evaluation showed a 1,25-Dihydroxyvitamin D (Calcitriol) mediated hypercalcemia. Serum calcium was 14.6 mg/dL, PTH was <0.6 pmol/L, PTHrp was 4.5 pmol/L (normal 0.0-2.3 pmol/L, likely due to renal impairment with an eGFR of 27ml/min), 25-Hydroxyvitamin D was 6ng/mL and Calcitriol was elevated to 111 pg/mL (normal 9.9-79.3 pg/mL). FDG PET CT showed gluteal and thigh low level uptake associated with heterogeneity and not distinct musculature on CT. Given prior biopsies negative for other granulomatous diseases, she was presumed to have severe hypercalcemia secondary to silicosis. She was treated with isotonic fluids, calcitonin and pamidronate without significant improvement in calcium levels. High dose methylprednisolone was started as treatment for her DPLD with improvement of calcium to 11.9 mg/dL. Ketoconazole was then started at an initial dose of 200mg twice daily. Calcium improved further to 8.1 mg/dL. Calcitriol was later remeasured to be 62.5mg/mL. Over her hospitalization, ketoconazole was variably held, restarted and adjusted in dose. At the time of writing, she has been maintained on ketoconazole 200mg twice daily with stably improved serum calcium measurements. The use of ISTF injections, including with silicone, has been reported in literature. Injections vary in both site and volume. Complications are often delayed and range from infection to formation of granulomas. Granulomas can induce hypercalcemia through macrophage mediated extra-renal 1 alpha hydroxylase activity, increasing conversion of 25-Hydroxyvitamin D into activated Calcitriol. Evaluation of granuloma mediated hypercalcemia reveals a PTH independent process, often but not always with elevated calcitriol levels. Localization of granulomas can be challenging, with FDG-PET CT having been shown to be useful in identifying areas of hypermetabolic granulomatous disease. Hypercalcemia has been seldom reported in the literature in association with cosmetic injections, commonly of silicone. In these cases, treatment often involves the use of bisphosphonates and steroids, with few reports describing the use of ketoconazole. Ketoconazole inhibits the effects of 1 alpha hydroxylase to reduce calcitriol production. This case represents a rare presentation of hypercalcemia related to silicosis, and the utility of ketoconazole in the treatment of granuloma mediated hypercalcemia. Presentation: Sunday, July 13, 2025

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Journal
Journal of the Endocrine Society
Published
2025-10-01
DOI
https://doi.org/10.1210/jendso/bvaf149.733
Primary Topic
Bone health and treatments
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article
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article

SUN-785 Silicosis Induced Hypercalcemia Treated With Ketoconazole

Mona Al Mukaddam, Bassam AlHamer
Journal of the Endocrine Society
Bone health and treatments
article

SUN-785 Silicosis Induced Hypercalcemia Treated With Ketoconazole

Mona Al Mukaddam, Bassam AlHamer
article en

Abstract

Abstract Disclosure: B. AlHamer: None. M. Al Mukaddam: None. A 43-year-old transgender woman with a history of illicit soft tissue filler (ISTF) injections was admitted for hypoxic respiratory failure. Endocrinology was consulted for severe hypercalcemia. She notably underwent ISTF injections around 11 years prior to presentation to the cheek, jawline and buttocks. She later went on to develop severe diffuse parenchymal lung disease (DPLD); this was felt to likely be secondary to silicone emboli as liver biopsies demonstrated lipogranulomas consistent with silicone emboli. Initial evaluation showed a 1,25-Dihydroxyvitamin D (Calcitriol) mediated hypercalcemia. Serum calcium was 14.6 mg/dL, PTH was <0.6 pmol/L, PTHrp was 4.5 pmol/L (normal 0.0-2.3 pmol/L, likely due to renal impairment with an eGFR of 27ml/min), 25-Hydroxyvitamin D was 6ng/mL and Calcitriol was elevated to 111 pg/mL (normal 9.9-79.3 pg/mL). FDG PET CT showed gluteal and thigh low level uptake associated with heterogeneity and not distinct musculature on CT. Given prior biopsies negative for other granulomatous diseases, she was presumed to have severe hypercalcemia secondary to silicosis. She was treated with isotonic fluids, calcitonin and pamidronate without significant improvement in calcium levels. High dose methylprednisolone was started as treatment for her DPLD with improvement of calcium to 11.9 mg/dL. Ketoconazole was then started at an initial dose of 200mg twice daily. Calcium improved further to 8.1 mg/dL. Calcitriol was later remeasured to be 62.5mg/mL. Over her hospitalization, ketoconazole was variably held, restarted and adjusted in dose. At the time of writing, she has been maintained on ketoconazole 200mg twice daily with stably improved serum calcium measurements. The use of ISTF injections, including with silicone, has been reported in literature. Injections vary in both site and volume. Complications are often delayed and range from infection to formation of granulomas. Granulomas can induce hypercalcemia through macrophage mediated extra-renal 1 alpha hydroxylase activity, increasing conversion of 25-Hydroxyvitamin D into activated Calcitriol. Evaluation of granuloma mediated hypercalcemia reveals a PTH independent process, often but not always with elevated calcitriol levels. Localization of granulomas can be challenging, with FDG-PET CT having been shown to be useful in identifying areas of hypermetabolic granulomatous disease. Hypercalcemia has been seldom reported in the literature in association with cosmetic injections, commonly of silicone. In these cases, treatment often involves the use of bisphosphonates and steroids, with few reports describing the use of ketoconazole. Ketoconazole inhibits the effects of 1 alpha hydroxylase to reduce calcitriol production. This case represents a rare presentation of hypercalcemia related to silicosis, and the utility of ketoconazole in the treatment of granuloma mediated hypercalcemia. Presentation: Sunday, July 13, 2025

Journal of the Endocrine SocietyVol. 9(Supplement_1)
University of Pennsylvania (US), Philadelphia University (US)
Openalex Percentile: Top 59%
Bone health and treatments
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