Spontaneous Remission of Cushing's Syndrome in a Patient with an Adrenal Adenoma

Blau, Nenad; Miller, William E.; Miller, Edgar R.; Cervi-Skinner, Sergio J.

doi:10.1210/jcem-40-4-659

Cited by 19 publications

(6 citation statements)

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“…Since then other patients have been reported (Blau et al, 1975;Liberman et al, 1976). The basic mechanism underlying the periodicity of cortisol over production is unclear.…”

Section: Introductionmentioning

confidence: 96%

Cushing's Disease Cycling over Ten Years

Popović¹,

Micić²,

Nesović³

et al. 2009

Exp Clin Endocrinol Diabetes

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Cycles of excessive cortisol secretion have been reported interposed with phases of remission lasting from few days to several months. In this study we report a ten year follow up on a patient who had four episodes of hypercortisolism and Cushing's syndrome associated with hypokalemia and edema and three long lasting spontaneous remissions. This was all caused by corticotroph pituitary adenoma. She demonstrated the longest phase of remission yet recorded in this condition, 4.5 years. The precipitating factors for the recurrence in cyclical Cushing's syndrome are not well defined, but the last recurrence in our patient appeared two months after delivery. We have discussed the continuing uncertainty of the standard dynamic tests in the differential diagnosis of Cushing's syndrome.

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“…Since then other patients have been reported (Blau et al, 1975;Liberman et al, 1976). The basic mechanism underlying the periodicity of cortisol over production is unclear.…”

Section: Introductionmentioning

confidence: 96%

Cushing's Disease Cycling over Ten Years

Popović¹,

Micić²,

Nesović³

et al. 2009

Exp Clin Endocrinol Diabetes

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“…Cyclicity has also been reported in patients with adrenal tumours (13) and ectopic ACTH syndrome (14), but it is most described in patients with Cushing's disease. Owing to the lack of any comprehensive data on the prevalence of the phenomenon, the aim of the present study was to investigate the presence of 'cyclicity' (defined as the presence of clinical and biochemical peaks and troughs) and of 'variability' (biochemical fluctuations of cortisol secretion) in a large cohort of patients with Cushing's disease.…”

Section: Introductionmentioning

confidence: 99%

The prevalence and characteristic features of cyclicity and variability in Cushing's disease

Alexandraki¹,

Kaltsas²,

Isidori

et al. 2009

European Journal of Endocrinology

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Objective: Cyclical Cushing's syndrome may render the diagnosis and management of Cushing's disease difficult. The aim of the present study was to investigate the prevalence of cyclicity and variability in patients with Cushing's disease, and to identify putative distinctive features. Design: Retrospective case-note study. Methods: We analysed the case records of 201 patients with Cushing's disease in a retrospective casenote study. Cyclicity was considered as the presence of at least one cycle, defined as a clinical and/or biochemical hypercortisolaemic peak followed by clinical and biochemical remission, followed by a new clinical and/or biochemical hypercortisolaemic peak. The fluctuations of mean serum cortisol levels, as assessed by a 5-point cortisol day curve, defined the variability. Results: Thirty (14.9%; 26 females) patients had evidence of cyclicity/variability. 'Cycling' patients were older but no difference in sex or paediatric distribution was revealed between 'cycling' and 'noncycling' patients. The median number of cycles was two for each patient, and 4 years was the median intercyclic period. A trend to lower cure rate post-neurosurgery and lower adenoma identification was observed in 'cycling' compared with 'non-cycling' patients. In multivariate analysis, older patients, longer follow-up, female sex and no histological identification of the adenoma were associated with an increased risk of cyclic disease. Conclusions: This large population study reveals that cyclicity/variability is not an infrequent phenomenon in patients with Cushing's disease, with a minimum prevalence of 15%. Physicians should be alert since it can lead to frequent problems in diagnosis and management, and no specific features can be used as markers.

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“…Although spontaneous remission of Cushing's syndrome is unusual it has been described in a few patients with either pituitary-dependent disease (Pasqualini & Gurevich, 1956;Hayslett & Cohn, 1967;Mornex et al, 1972;Putnam et al, 1972;Scott et a[., 1979;Bochner et al, 1979) or a primary adrenal tumour (Crooke, 1947;Ovlisen & Anderson, 1966;Blau et a[., 1975). Recently, Kammer & Barter (1979) described an elderly man who presented with Cushing's syndrome, the clinical picture being strongly suggestive of ectopic ACTH secretion.…”

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confidence: 99%

Prolonged Remission in Florid Cushing's Syndrome Following Metyrapone Treatment

Beardwell

Adamson

Shalet

1981

Clinical Endocrinology

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Two patients presenting with diabetes mellitus and hypokalaemia resulting from markedly increased ACTH and cortisol secretion are described. Neither patient showed any evidence of a tumour and both responded dramatically to treatment with metyrapone in that all abnormal clinical features disappeared, ACTH concentrations returned to normal and both patients showed prolonged remission after metyrapone treatment was stopped. One patient relapsed after a severe viral illness and the administration of dexamethasone and cortisone. It is suggested that these cases may represent an unusual form of Cushing's syndrome in which ACTH secretion is stimulated by increasing concentrations of cortisol. When these are reduced by metyrapone administration ACTH secretion falls in parallel and prolonged remission of disease may result.

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Spontaneous Remission of Cushing's Syndrome in a Patient with an Adrenal Adenoma

Cited by 19 publications

References 0 publications

Cushing's Disease Cycling over Ten Years

Cushing's Disease Cycling over Ten Years

The prevalence and characteristic features of cyclicity and variability in Cushing's disease

Prolonged Remission in Florid Cushing's Syndrome Following Metyrapone Treatment

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