Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy
Background/Objective: Reducing severity of Cushing’s syndrome caused by an adrenal adenoma (adrenal Cushing’s syndrome [ACS]) might decrease morbidity and mortality risk in adrenalectomy. We used off-label osilodrostat, approved in the United States for pituitary Cushing’s disease, to reduce cortiso...
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| Format: | Article |
| Language: | English |
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Elsevier
2022-11-01
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| Series: | AACE Clinical Case Reports |
| Subjects: | |
| Online Access: | http://www.sciencedirect.com/science/article/pii/S2376060522000657 |
| _version_ | 1798015918397194240 |
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| author | Risha B. Malik, MD Anat Ben-Shlomo, MD |
| author_facet | Risha B. Malik, MD Anat Ben-Shlomo, MD |
| author_sort | Risha B. Malik, MD |
| collection | DOAJ |
| description | Background/Objective: Reducing severity of Cushing’s syndrome caused by an adrenal adenoma (adrenal Cushing’s syndrome [ACS]) might decrease morbidity and mortality risk in adrenalectomy. We used off-label osilodrostat, approved in the United States for pituitary Cushing’s disease, to reduce cortisol levels and disease severity before adrenalectomy 3 weeks later. Case Report: A 48-year-old woman with a 6-year history of obesity, depression, and anxiety and 3-year history of diabetes and hypertension was admitted with vomiting and lumbar back pain. Facial plethora and hirsutism, posterior cervicothoracic fat pad, and truncal obesity coupled with morning serum cortisol >13 μg/dL after 1 mg oral dexamethasone suppression, urinary free cortisol 1324 μg/24hr (4.0-50.0 μg/24 h), and adrenocorticotropin <5 pg/mL (6-50 pg/mL) confirmed ACS. Computed tomography with contrast revealed a 3.4-cm right adrenal mass. Osilodrostat 2 mg twice daily initiated at discharge was increased to 4 mg twice daily on day 6. Three days later, she reported nausea, vomiting, and fatigue. Despite 7.2 μg/dL morning cortisol, adrenal insufficiency was suspected; osilodrostat was reduced to 2 mg twice daily and maintenance oral hydrocortisone 20 mg daily was added with symptom resolution. Prior to adrenalectomy, morning cortisol was 5.1 μg/dL, fasting glucose was 122 mg/dL, and she self-discontinued diabetes medications. Hypertension remained unchanged (149/100 vs 151/94 mmHg). Adrenalectomy revealed a 3.4-cm focally pigmented adrenocortical adenoma. Discussion: Three-week treatment of overt ACS with off-label osilodrostat reduced cortisol and glucose levels before curative adrenalectomy. Abrupt cortisol reduction led to suspected adrenal insufficiency managed with maintenance hydrocortisone. Conclusion: Osilodrostat might help reduce ACS severity before adrenalectomy. Adrenal insufficiency is a risk but can be safely managed with hydrocortisone. |
| first_indexed | 2024-04-11T15:41:15Z |
| format | Article |
| id | doaj.art-416213b449684fa7b5f4d78ca42cc93e |
| institution | Directory Open Access Journal |
| issn | 2376-0605 |
| language | English |
| last_indexed | 2024-04-11T15:41:15Z |
| publishDate | 2022-11-01 |
| publisher | Elsevier |
| record_format | Article |
| series | AACE Clinical Case Reports |
| spelling | doaj.art-416213b449684fa7b5f4d78ca42cc93e2022-12-22T04:15:46ZengElsevierAACE Clinical Case Reports2376-06052022-11-0186267270Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and AdrenalectomyRisha B. Malik, MD0Anat Ben-Shlomo, MD1Division of Endocrinology, Diabetes & Metabolism, Cedars-Sinai Medical Center, Los Angeles, CaliforniaDivision of Endocrinology, Diabetes & Metabolism, Cedars-Sinai Medical Center, Los Angeles, California; Multidisciplinary Adrenal Program, Department of Medicine, Cedars-Sinai Medical Center, Los Angeles, California; Address correspondence to Dr Anat Ben-Shlomo, Cedars-Sinai Medical Center, 127 S San Vicente Blvd, Suite A6600, Los Angeles, CA 90048.Background/Objective: Reducing severity of Cushing’s syndrome caused by an adrenal adenoma (adrenal Cushing’s syndrome [ACS]) might decrease morbidity and mortality risk in adrenalectomy. We used off-label osilodrostat, approved in the United States for pituitary Cushing’s disease, to reduce cortisol levels and disease severity before adrenalectomy 3 weeks later. Case Report: A 48-year-old woman with a 6-year history of obesity, depression, and anxiety and 3-year history of diabetes and hypertension was admitted with vomiting and lumbar back pain. Facial plethora and hirsutism, posterior cervicothoracic fat pad, and truncal obesity coupled with morning serum cortisol >13 μg/dL after 1 mg oral dexamethasone suppression, urinary free cortisol 1324 μg/24hr (4.0-50.0 μg/24 h), and adrenocorticotropin <5 pg/mL (6-50 pg/mL) confirmed ACS. Computed tomography with contrast revealed a 3.4-cm right adrenal mass. Osilodrostat 2 mg twice daily initiated at discharge was increased to 4 mg twice daily on day 6. Three days later, she reported nausea, vomiting, and fatigue. Despite 7.2 μg/dL morning cortisol, adrenal insufficiency was suspected; osilodrostat was reduced to 2 mg twice daily and maintenance oral hydrocortisone 20 mg daily was added with symptom resolution. Prior to adrenalectomy, morning cortisol was 5.1 μg/dL, fasting glucose was 122 mg/dL, and she self-discontinued diabetes medications. Hypertension remained unchanged (149/100 vs 151/94 mmHg). Adrenalectomy revealed a 3.4-cm focally pigmented adrenocortical adenoma. Discussion: Three-week treatment of overt ACS with off-label osilodrostat reduced cortisol and glucose levels before curative adrenalectomy. Abrupt cortisol reduction led to suspected adrenal insufficiency managed with maintenance hydrocortisone. Conclusion: Osilodrostat might help reduce ACS severity before adrenalectomy. Adrenal insufficiency is a risk but can be safely managed with hydrocortisone.http://www.sciencedirect.com/science/article/pii/S2376060522000657Cushing’s syndromeosilodrostatadrenalectomyadrenal insufficiency |
| spellingShingle | Risha B. Malik, MD Anat Ben-Shlomo, MD Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy AACE Clinical Case Reports Cushing’s syndrome osilodrostat adrenalectomy adrenal insufficiency |
| title | Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy |
| title_full | Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy |
| title_fullStr | Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy |
| title_full_unstemmed | Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy |
| title_short | Adrenal Cushing’s Syndrome Treated With Preoperative Osilodrostat and Adrenalectomy |
| title_sort | adrenal cushing s syndrome treated with preoperative osilodrostat and adrenalectomy |
| topic | Cushing’s syndrome osilodrostat adrenalectomy adrenal insufficiency |
| url | http://www.sciencedirect.com/science/article/pii/S2376060522000657 |
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