MON-087 Glucocorticoid Withdrawal State Presenting Late After Surgical Cure Of Cushing’S Disease With Severe Hypercalcemia

Abstract Disclosure: F. Loo: None. R. Lai: None. Introduction Glucocorticoid withdrawal syndrome (GWS) typically occurs days after successful surgical treatment of Cushing’s disease (CD) and presents with non-specific symptoms. We report a rare presentation of GWS with severe hypercalcemia occurring months after successful surgical treatment of CD, at time of recovery of the hypothalamic-pituitary-adrenal (HPA) axis. Case Report A 54-year-old woman with severe CD underwent transsphenoidal surgery with an early post-operative serum cortisol nadir of 93 nmol/L. After surgery, she received a tapering dose of glucocorticoids. At time of hydrocortisone discontinuation 4 months later, short Synacthen test demonstrated a baseline ACTH of 2 pmol/L, 0-minute cortisol of 297 nmol/L, and robust peak cortisol (60 minutes) of 798 nmol/L. 1 week later, the patient was hospitalized with symptoms of nausea, anorexia, and malaise. Repeat 8am cortisol was 363 nmol/L. Serum calcium (adjusted for albumin) was elevated at 3.83 mmol/L [2.15 - 2.50], phosphate 1.7 mmol/L [0.8 - 1.4], creatinine 114 umol/L [40-75] and 25-hydroxyvitamin D 51 mcg/L [30-100]. Parathyroid hormone (PTH) was suppressed at 1.3 pmol/L, PTH related peptide suppressed at 1.7 pmol/L [<4.2], and 1,25-hydroxyvitamin D also suppressed at 10 pg/mL [18-78]. Free thyroxine was 17 pmol/L [8-16] and thyroid stimulating hormone 5.68 mIU/L [0.45 - 4.50], not accounting for hypercalcemia. A negative myeloma panel and unremarkable 18-fluorodeoxyglucose positron-emission tomography (PET) CT scan excluded other causes of non-PTH-mediated hypercalcemia. Following initial treatment of hypercalcemia with hydration and intravenous pamidronate 30mg, the reinstitution of supraphysiological doses of glucocorticoids resulted in sustained improvement of serum calcium levels to 2.38-2.70 mmol/L in the following eight months to date. Discussion GWS usually develops following a rapid relative reduction of excess circulating glucocorticoid. The risk is heightened in treated CD patients with previously prolonged, severe hypercortisolism. Non-PTH-mediated hypercalcemia is an uncommonly reported presentation, and is characterized by elevated urinary calcium and hydroxyproline excretion, and a response to supraphysiological doses of glucocorticoid. Conclusion Treated CD patients with a recovered HPA axis may still present with GWS owing to a state of relative glucocorticoid insufficiency. Hypercalcemia is an atypical presentation. Presentation: Monday, July 14, 2025

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Journal
Journal of the Endocrine Society
Published
2025-10-01
DOI
https://doi.org/10.1210/jendso/bvaf149.1418
Primary Topic
Hematological disorders and diagnostics
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article
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article

MON-087 Glucocorticoid Withdrawal State Presenting Late After Surgical Cure Of Cushing’S Disease With Severe Hypercalcemia

Ray Lai, Fernando Jia Jing Loo
Journal of the Endocrine Society
Hematological disorders and diagnostics
article

MON-087 Glucocorticoid Withdrawal State Presenting Late After Surgical Cure Of Cushing’S Disease With Severe Hypercalcemia

Ray Lai, Fernando Jia Jing Loo
article en

Abstract

Abstract Disclosure: F. Loo: None. R. Lai: None. Introduction Glucocorticoid withdrawal syndrome (GWS) typically occurs days after successful surgical treatment of Cushing’s disease (CD) and presents with non-specific symptoms. We report a rare presentation of GWS with severe hypercalcemia occurring months after successful surgical treatment of CD, at time of recovery of the hypothalamic-pituitary-adrenal (HPA) axis. Case Report A 54-year-old woman with severe CD underwent transsphenoidal surgery with an early post-operative serum cortisol nadir of 93 nmol/L. After surgery, she received a tapering dose of glucocorticoids. At time of hydrocortisone discontinuation 4 months later, short Synacthen test demonstrated a baseline ACTH of 2 pmol/L, 0-minute cortisol of 297 nmol/L, and robust peak cortisol (60 minutes) of 798 nmol/L. 1 week later, the patient was hospitalized with symptoms of nausea, anorexia, and malaise. Repeat 8am cortisol was 363 nmol/L. Serum calcium (adjusted for albumin) was elevated at 3.83 mmol/L [2.15 - 2.50], phosphate 1.7 mmol/L [0.8 - 1.4], creatinine 114 umol/L [40-75] and 25-hydroxyvitamin D 51 mcg/L [30-100]. Parathyroid hormone (PTH) was suppressed at 1.3 pmol/L, PTH related peptide suppressed at 1.7 pmol/L [<4.2], and 1,25-hydroxyvitamin D also suppressed at 10 pg/mL [18-78]. Free thyroxine was 17 pmol/L [8-16] and thyroid stimulating hormone 5.68 mIU/L [0.45 - 4.50], not accounting for hypercalcemia. A negative myeloma panel and unremarkable 18-fluorodeoxyglucose positron-emission tomography (PET) CT scan excluded other causes of non-PTH-mediated hypercalcemia. Following initial treatment of hypercalcemia with hydration and intravenous pamidronate 30mg, the reinstitution of supraphysiological doses of glucocorticoids resulted in sustained improvement of serum calcium levels to 2.38-2.70 mmol/L in the following eight months to date. Discussion GWS usually develops following a rapid relative reduction of excess circulating glucocorticoid. The risk is heightened in treated CD patients with previously prolonged, severe hypercortisolism. Non-PTH-mediated hypercalcemia is an uncommonly reported presentation, and is characterized by elevated urinary calcium and hydroxyproline excretion, and a response to supraphysiological doses of glucocorticoid. Conclusion Treated CD patients with a recovered HPA axis may still present with GWS owing to a state of relative glucocorticoid insufficiency. Hypercalcemia is an atypical presentation. Presentation: Monday, July 14, 2025

Journal of the Endocrine SocietyVol. 9(Supplement_1)
Tan Tock Seng Hospital (SG)
Openalex Percentile: Top 55%
Hematological disorders and diagnostics
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