Fluconazole treatment in severe ectopic Cushing syndrome
Severe Cushing syndrome (SCS) is considered an emergency that requires immediate treatment to lower serum cortisol levels. Fluconazole may be considered an alternative treatment in Cushing syndrome when ketoconazole is not tolerated or unavailable. We report a 39-year-old woman with a history of par...
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| Format: | Article |
| Language: | English |
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Bioscientifica
2019-07-01
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| Series: | Endocrinology, Diabetes & Metabolism Case Reports |
| Online Access: | https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM19-0020.xml |
| _version_ | 1819075922141118464 |
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| author | Teresa M Canteros Valeria De Miguel Patricia Fainstein-Day |
| author_facet | Teresa M Canteros Valeria De Miguel Patricia Fainstein-Day |
| author_sort | Teresa M Canteros |
| collection | DOAJ |
| description | Severe Cushing syndrome (SCS) is considered an emergency that requires immediate treatment to lower serum cortisol levels. Fluconazole may be considered an alternative treatment in Cushing syndrome when ketoconazole is not tolerated or unavailable. We report a 39-year-old woman with a history of partial pancreaticoduodenectomy due to a periampullary neuroendocrine tumor with locoregional extension. Three years after surgery, she developed liver metastases and was started on 120 mg of lanreotide/month, despite which, liver metastases progressed in the following 6 months. The patient showed extreme fatigue, muscle weakness, delirium, moon face, hirsutism and severe proximal weakness. Laboratory tests showed anemia, hyperglycemia and severe hypokalemia. 24-h urinary free cortisol: 2152 nmol/day (reference range (RR): <276), morning serum cortisol 4883.4 nmol/L (RR: 138–690), ACTH 127.3 pmol/L (RR: 2.2–10). She was diagnosed with ectopic ACTH syndrome (EAS). On admission, she presented with acute upper gastrointestinal tract bleeding and hemodynamic instability. Intravenous fluconazole 400 mg/day was started. After 48 h, her mental state improved and morning cortisol decreased by 25%. The dose was titrated to 600 mg/day which resulted in a 55% decrease in cortisol levels in 1 week, but then had to be decreased to 400 mg/day because transaminase levels increased over 3 times the upper normal level. After 18 days of treatment, hemodynamic stability, lower cortisol levels and better overall clinical status enabled successful bilateral adrenalectomy. This case report shows that intravenous fluconazole effectively decreased cortisol levels in SCS due to EAS. |
| first_indexed | 2024-12-21T18:33:06Z |
| format | Article |
| id | doaj.art-db19c2371c644ff193fb5cb08d29adf7 |
| institution | Directory Open Access Journal |
| issn | 2052-0573 2052-0573 |
| language | English |
| last_indexed | 2024-12-21T18:33:06Z |
| publishDate | 2019-07-01 |
| publisher | Bioscientifica |
| record_format | Article |
| series | Endocrinology, Diabetes & Metabolism Case Reports |
| spelling | doaj.art-db19c2371c644ff193fb5cb08d29adf72022-12-21T18:54:14ZengBioscientificaEndocrinology, Diabetes & Metabolism Case Reports2052-05732052-05732019-07-01111410.1530/EDM-19-0020Fluconazole treatment in severe ectopic Cushing syndromeTeresa M Canteros0Valeria De Miguel1Patricia Fainstein-Day2Endocrinology, Metabolism and Nuclear Medicine, Hospital Italinao de Buenos Aires, Buenos Aires, ArgentinaEndocrinology, Metabolism and Nuclear Medicine, Hospital Italinao de Buenos Aires, Buenos Aires, ArgentinaEndocrinology, Metabolism and Nuclear Medicine, Hospital Italinao de Buenos Aires, Buenos Aires, ArgentinaSevere Cushing syndrome (SCS) is considered an emergency that requires immediate treatment to lower serum cortisol levels. Fluconazole may be considered an alternative treatment in Cushing syndrome when ketoconazole is not tolerated or unavailable. We report a 39-year-old woman with a history of partial pancreaticoduodenectomy due to a periampullary neuroendocrine tumor with locoregional extension. Three years after surgery, she developed liver metastases and was started on 120 mg of lanreotide/month, despite which, liver metastases progressed in the following 6 months. The patient showed extreme fatigue, muscle weakness, delirium, moon face, hirsutism and severe proximal weakness. Laboratory tests showed anemia, hyperglycemia and severe hypokalemia. 24-h urinary free cortisol: 2152 nmol/day (reference range (RR): <276), morning serum cortisol 4883.4 nmol/L (RR: 138–690), ACTH 127.3 pmol/L (RR: 2.2–10). She was diagnosed with ectopic ACTH syndrome (EAS). On admission, she presented with acute upper gastrointestinal tract bleeding and hemodynamic instability. Intravenous fluconazole 400 mg/day was started. After 48 h, her mental state improved and morning cortisol decreased by 25%. The dose was titrated to 600 mg/day which resulted in a 55% decrease in cortisol levels in 1 week, but then had to be decreased to 400 mg/day because transaminase levels increased over 3 times the upper normal level. After 18 days of treatment, hemodynamic stability, lower cortisol levels and better overall clinical status enabled successful bilateral adrenalectomy. This case report shows that intravenous fluconazole effectively decreased cortisol levels in SCS due to EAS.https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM19-0020.xml |
| spellingShingle | Teresa M Canteros Valeria De Miguel Patricia Fainstein-Day Fluconazole treatment in severe ectopic Cushing syndrome Endocrinology, Diabetes & Metabolism Case Reports |
| title | Fluconazole treatment in severe ectopic Cushing syndrome |
| title_full | Fluconazole treatment in severe ectopic Cushing syndrome |
| title_fullStr | Fluconazole treatment in severe ectopic Cushing syndrome |
| title_full_unstemmed | Fluconazole treatment in severe ectopic Cushing syndrome |
| title_short | Fluconazole treatment in severe ectopic Cushing syndrome |
| title_sort | fluconazole treatment in severe ectopic cushing syndrome |
| url | https://edm.bioscientifica.com/view/journals/edm/2019/1/EDM19-0020.xml |
| work_keys_str_mv | AT teresamcanteros fluconazoletreatmentinsevereectopiccushingsyndrome AT valeriademiguel fluconazoletreatmentinsevereectopiccushingsyndrome AT patriciafainsteinday fluconazoletreatmentinsevereectopiccushingsyndrome |