An unusual cause of Cushing's syndrome with secondary adrenal insufficiency

2013 ◽  
pp. 1-1
Author(s):  
Amalia Iliopoulou ◽  
Emma Ward
Keyword(s):  
Adrenal Insufficiency ◽  
Cushing’S Syndrome ◽  
Cushing's Syndrome ◽  
Secondary Adrenal Insufficiency

scholarly journals Thrombospondin-1 is a glucocorticoid responsive protein in humans

2016 ◽  
Vol 174 (2) ◽  
pp. 193-201 ◽  
Author(s):  
Johanna L Barclay ◽  
Carolyn J Petersons ◽  
Sahar Keshvari ◽  
Jane Sorbello ◽  
Brenda L Mangelsdorf ◽  
...  
Keyword(s):  
Healthy Volunteers ◽  
Adrenal Insufficiency ◽  
Cushing’S Syndrome ◽  
Cushing's Syndrome ◽  
Matricellular Protein ◽  
Mrna Levels ◽  
Secondary Adrenal Insufficiency ◽  
Interventional Study ◽  
Peripheral Blood Mononuclear ◽  
Thrombospondin 1

ObjectiveThrombospondin-1 (TSP1) is a matricellular protein whose gene expression has previously been shown to increase acutely after exposure to dexamethasone in vitro. The aim of this study was to determine if TSP1 is altered by acute and chronic states of glucocorticoid excess in human subjects.Design and methodsThree studies have been undertaken to assess the difference or change in TSP1 in response to altered glucocorticoid activity: i) an acute interventional study assessed the effects of a single 4 mg dose of dexamethasone in 20 healthy volunteers; ii) a cross-sectional study compared plasma TSP1 in 20 healthy volunteers and eight patients with Cushing's syndrome; iii) an interventional study assessed the effect on plasma TSP1 of an increase in hydrocortisone dose from ≤20 mg/day to 30 mg/day for 7 days in 16 patients with secondary adrenal insufficiency.ResultsIn healthy volunteers, 4 mg dexamethasone significantly increased peripheral blood mononuclear cell (PBMC) TSP1 mRNA levels (P<0.0001) and plasma TSP1 concentrations (P<0.0001), peaking at 12 h. Median (interquartile range) plasma TSP1 was higher in Cushing's, 638 (535–756) ng/ml, than in healthy volunteers, 272 (237–336) ng/ml (P<0.0001). Plasma TSP1 >400 ng/ml diagnosed Cushing's syndrome with sensitivity of 100% and specificity of 85%. The higher hydrocortisone dose increased plasma TSP1 from 139 (86–199) to 256 (133–516) ng/ml, (P<0.01) in patients with secondary adrenal insufficiency.ConclusionsTSP1 is a glucocorticoid responsive protein in humans. Further research is required to determine if plasma TSP1 has a role as a glucocorticoid biomarker.

scholarly journals Too little or too much corticosteroid? Coexisting adrenal insufficiency and Cushing's syndrome from chronic, intermittent use of intranasal betamethasone

2013 ◽  
Vol 2013 ◽  
Author(s):  
Adrienne Dow ◽  
Run Yu ◽  
John Carmichael
Keyword(s):  
Adrenal Insufficiency ◽  
Cushing’S Syndrome ◽  
Cushing's Syndrome ◽  
High Dose ◽  
List Type ◽  
Secondary Adrenal Insufficiency ◽  
Severe Fatigue ◽  
Learning Points ◽  
Endocrinology Clinic ◽  
Iatrogenic Cushing’S Syndrome

Summary To report the puzzling, rare occurrence of coexisting adrenal insufficiency and Cushing's syndrome from chronic, intermittent use of intranasal betamethasone spray. A 62-year-old male was referred to our endocrinology clinic for management of adrenal insufficiency. This previously healthy individual began to experience chronic sinus symptoms in 2007, was treated with multiple ensuing sinus surgeries, and received oral glucocorticoid for 6 months. In the following 5 years, he suffered severe fatigue and was diagnosed with secondary adrenal insufficiency. He could not be weaned from corticosteroid and developed clear cushingoid features. In our clinic, careful inquiry on medications revealed chronic, intermittent use of high-dose intranasal betamethasone since 2008, which was not apparent to his other treating physicians. His cushingoid features significantly improved after holding intranasal betamethasone. Learning points Chronic, intermittent intranasal betamethasone can cause secondary adrenal insufficiency and iatrogenic Cushing's syndrome when used in excess. Topical corticosteroid use should be considered in the differential diagnosis of adrenal insufficiency or Cushing's syndrome.

scholarly journals Steroid withdrawal syndrome after successful treatment of Cushing’s syndrome: a reminder

2005 ◽  
Vol 153 (2) ◽  
pp. 207-210 ◽  
Author(s):  
A Bhattacharyya ◽  
K Kaushal ◽  
D J Tymms ◽  
J R E Davis
Keyword(s):  
Adrenal Insufficiency ◽  
Replacement Therapy ◽  
Cushing’S Syndrome ◽  
Withdrawal Syndrome ◽  
Pituitary Surgery ◽  
Cushing's Syndrome ◽  
Temporary Increase ◽  
Steroid Withdrawal ◽  
Glucocorticoid Replacement Therapy ◽  
Cortisol Levels

Steroid withdrawal syndrome (SWS) usually refers to relapse of the disease being treated after withdrawal of glucocorticoid therapy, or the symptoms of adrenal insufficiency which occur when glucocorticoids are rapidly reduced or stopped. A less well-recognised form of SWS is that which develops when patients experience a symptom complex similar to that of adrenal insufficiency despite acceptable cortisol levels. We describe three patients who presented with this form of SWS following surgical treatment for endogenous Cushing’s syndrome. All responded well to a short-term increase in the dose of glucocorticoid replacement therapy, with the median duration of the syndrome being 10 months (range 6–10 months). Trough serum cortisol levels above 100 nmol/l, with peaks between 460 and 750 nmol/l were documented in the first two patients at presentation with SWS. It is thought that the syndrome may result from development of tolerance to glucocorticoids, and mediators considered to be important in its development include interleukin-6, corticotrophin-releasing hormone, vasopressin, and central noradrenergic and dopaminergic systems. The exact underlying mechanism for SWS remains unclear. However, with increasing recommendations for use of lower doses of replacement glucocorticoids, its incidence may increase. Physicians need to be aware of this condition, which is self-limiting and easily treated by a temporary increase in the dose of glucocorticoid replacement therapy. It is possible that a slower glucocorticoid tapering regimen than that used in the standard postoperative management of patients undergoing pituitary surgery may reduce the risk of development of SWS.

scholarly journals Mifepristone (RU 486) in Cushing's syndrome

2007 ◽  
Vol 157 (5) ◽  
pp. 561-569 ◽  
Author(s):  
Sarah Johanssen ◽  
Bruno Allolio
Keyword(s):  
Adrenal Insufficiency ◽  
Cushing’S Syndrome ◽  
Case Reports ◽  
Evidence Synthesis ◽  
Cushing's Syndrome ◽  
Therapeutic Effects ◽  
Receptor Blockade ◽  
Onset Of Action ◽  
Ru 486 ◽  
Long Term Treatment

AbstractContextMifepristone (RU 486) blocks the action of cortisol by binding to the glucocorticoid receptor and, therefore, is of potential therapeutic value in Cushing's syndrome. However, research in endogenous hypercortisolism has been hampered by the controversy related to the use of mifepristone for inducing abortion. Currently, new studies are planned to better define the role of RU 486 in Cushing's syndrome. This paper reviews the available evidence concerning the therapeutic effects and adverse events of RU 486 in Cushing's syndrome.Evidence acquisitionOriginal articles and reviews were identified using a PubMed search strategy covering the time period until February 2007.Evidence synthesisTreatment of Cushing's syndrome with mifepristone has been reported in a total of 18 patients, with daily doses ranging from 5 to 30 mg/kg. Case reports indicate that the mifepristone-induced receptor blockade may lead to significant clinical improvement in patients with Cushing's syndrome in whom surgery and inhibitors of adrenal steroidogenesis fail to control hypercortisolism. Due to its rapid onset of action, mifepristone may be particularly useful in acute crises, e.g. in cortisol-induced psychosis. Side effects include adrenal insufficiency and, as a result of its antiprogestin action, endometrial hyperplasia in long-term treatment. Adrenal insufficiency can be assessed only by careful clinical evaluation, as the hormonal parameters are not reliable during receptor blockade, and is rapidly reversed by exogenous dexamethasone. Well-designed larger clinical trials are needed to better assess the value of this interesting drug in the treatment of Cushing's syndrome.

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