| Adrenal Cushing's syndrome due to bilateral macronodular adrenal hyperplasia: prediction of the efficacy of beta-blockade therapy and interest of unilateral adrenalectomy. | |
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MedLine Citation:
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PMID: 19564707 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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Bilateral adrenalectomy is the standard treatment for Cushing's syndrome (CS) related to ACTH-independent bilateral macronodular hyperplasia (AIMAH), although it imposes life-long adrenal insufficiency. This study reports a clinical case in order to discuss the clinical interest of pharmacological beta-blockade of illegitimate membrane receptors and unilateral adrenalectomy as alternatives to bilateral adrenalectomy for treatment of CS due to AIMAH. Evidence for cortisol stimulation by upright posture and insulin-induced hypoglycemia in a patient with CS related to AIMAH led us to try beta-blockers as a therapeutic test and then as a first line treatment. Thus, a 3-day beta-blocker test (320 mg/d propranolol) induced normalization of cortisol secretion, with return of hypercortisolism at the end of the test. A long term treatment with 320 mg/d propranolol allowed sustained normalization of cortisol secretion and progressive disappearance of Cushingoid features but after 8 months the patient complained of Raynaud's syndrome and fatigue. Lowering propranolol dosage or switching to atenolol was less efficient to reduce cortisol levels. Unilateral adrenalectomy was then performed as a second line treatment, leading to normalisation of the 24h urinary cortisol without adrenal insufficiency. Long term control of blood pressure and glycemia were observed during a 7-year follow-up without beta-blocker. In conclusion, a 3-day propranolol test may identify patients with AIMAH who can benefit from a long term beta-blocker treatment. In case of intolerance to beta-blocking agents, unilateral adrenalectomy may allow for long term control of Cushing's syndrome related to AIMAH without adrenal insufficiency. |
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Authors:
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T??nia L Mazzuco; Philippe Chaffanjon; Monique Martinie; Nathalie Sturm; Olivier Chabre |
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Publication Detail:
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Type: Case Reports; Journal Article; Research Support, Non-U.S. Gov't Date: 2009-06-30 |
Journal Detail:
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Title: Endocrine journal Volume: 56 ISSN: 1348-4540 ISO Abbreviation: Endocr. J. Publication Date: 2009 |
Date Detail:
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Created Date: 2009-10-26 Completed Date: 2010-01-18 Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 9313485 Medline TA: Endocr J Country: Japan |
Other Details:
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Languages: eng Pagination: 867-77 Citation Subset: IM |
Affiliation:
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Service d'Endocrinologie, Centre Hospitalier Universitaire A. Michallon, Grenoble, France. |
Export Citation:
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| MeSH Terms | |
Descriptor/Qualifier:
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Adrenal Glands
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pathology* Adrenalectomy / methods* Adrenergic beta-Antagonists / therapeutic use* Atenolol Cushing Syndrome / drug therapy*, surgery* Female Humans Hydrocortisone / blood, urine Hyperplasia Middle Aged Posture Propranolol / diagnostic use, therapeutic use* Treatment Outcome Vasopressins / blood |
| Chemical | |
Reg. No./Substance:
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0/Adrenergic beta-Antagonists; 11000-17-2/Vasopressins; 29122-68-7/Atenolol; 50-23-7/Hydrocortisone; 525-66-6/Propranolol |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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