Spontaneous Remission in a Case of Cushing's Syndrome

Pasqualini, R Q; Гуревич, Н.Л.

doi:10.1210/jcem-16-3-406

Cited by 28 publications

(3 citation statements)

References 3 publications

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“…mal-sized pituitary gland with par¬ tially empty sella. The patient was then treated with1.5 to 4 g/d of mitotane. This successfully lowered plasma cortisol levels to normal with an ame¬ lioration of clinical manifestations of Cushing's disease, although plasma corticotropin levels increased fur¬ ther.…”

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confidence: 99%

Spontaneous Remission in Cushing's Disease

Ishibashi

Shimada²,

Abe³

et al. 1993

Arch Intern Med

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Two patients with Cushing's disease underwent spontaneous clinical and biochemical remission following a period of secondary adrenal insufficiency. One of the patients was untreated, while the other had been treated with partial bilateral adrenalectomy before. Both patients underwent remission shortly after the administration of dexamethasone for diagnostic purposes and remained symptom free for 2 and 3 years, respectively, which was followed by relapse of the disease. We conclude that although resolution of Cushing's disease may occur spontaneously, probably by hemorrhagic infarction of corticotroph microadenomas, careful follow-up studies are required since the relapse of the disease occurs later in some patients.

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confidence: 99%

Spontaneous Remission in Cushing's Disease

Ishibashi

Shimada²,

Abe³

et al. 1993

Arch Intern Med

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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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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 remissions in CS and CD are infrequent events that have been already reported more than fifty years ago (14,15). In the subsequent years, some other cases were published and some of them presented recurrence in the follow-up period (7,8,16).…”

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confidence: 94%

Spontaneous remission of hypercortisolism presumed due to asymptomatic tumor apoplexy in ACTH-producing pituitary macroadenoma

Machado

Gadelha

Bronstein

et al. 2013

Arq Bras Endocrinol Metab

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SUMMARYCushing's disease (CD) is usually caused by secretion of ACTH by a pituitary corticotroph microadenoma. Nevertheless, 7%-20% of patients present with ACTH-secreting macroadenomas. Our aim is to report a 36-year-old female patient with CD due to solid-cystic ACTH-macroadenoma followed up during 34 months. The patient presented spontaneous remission due to presumed asymptomatic tumor apoplexy. She showed typical signs and symptoms of Cushing's syndrome (CS). Initial tests were consistent with ACTH-dependent CS: elevated urinary free cortisol, abnormal serum cortisol after low dose dexamethasone suppression test, and elevated midnight salivary cortisol, associated with high plasma ACTH levels. Pituitary magnetic resonance imaging (MRI) showed a sellar mass of 1.2 x 0.8 x 0.8 cm of diameter with supra-sellar extension leading to slight chiasmatic impingement, and showing hyperintensity on T2-weighted imaging, suggesting a cystic component. She had no visual impairment. After two months, while waiting for pituitary surgery, she presented spontaneous resolution of CS. Tests were consistent with remission of hypercortisolism: normal 24-h total urinary cortisol and normal midnight salivary cortisol. Pituitary MRI showed shrinkage of the tumor with disappearance of the chiasmatic compression. She has been free from the disease for 28 months (without hypercortisolism or hypopituitarism). The hormonal and imaging data suggested that silent apoplexy of pituitary tumor led to spontaneous remission of CS. However, recurrence of CS was described in cases following pituitary apoplexy. Therefore, careful long-term follow-up is required. Arq Bras Endocrinol Metab. 2013;57(6):486-9 SUMÁRIO A doença de Cushing (DC) é usualmente causada por um microadenoma produtor de ACTH. Entretanto, 7%-20% dos pacientes apresentam um macroadenoma. O objetivo deste trabalho é reportar uma paciente de 36 anos, feminina, com diagnóstico de DC devido a macroadenoma hipofisário sólido-cístico com seguimento de 34 meses que apresentou remissão espontânea presumidamente em decorrência de uma apoplexia tumoral assintomática. Inicialmente, ela apresentava sinais e sintomas típicos da síndrome de Cushing (SC). Na admissão, os testes foram consistentes com o diagnóstico de SC ACTH-dependente: cortisol urinário livre de 24h elevado, não supressão do cortisol sérico após dose baixa de dexametasona e cortisol salivar noturno elevado, associado a concentrações elevadas do ACTH plasmático. Ressonância magnética (RM) de hipófise revelou uma massa selar de 1.2 x 0.8 x 0.8 cm com extensão suprasselar levando a uma discreta compressão do quiasma óptico e mostrando região de hipersinal na imagem ponderada em T2 sugerindo um componente cístico. A paciente não apresentava queixas visuais. Após dois meses, enquanto aguardava o tratamento cirúrgico, a paciente apresentou remissão espontânea da SC. A repetição dos exames indicou remissão do hipercortisolismo: normalização do cortisol urinário livre de 24h e normalização do cortisol salivar noturno. Nova RM de hipóf...

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Spontaneous Remission in a Case of Cushing's Syndrome

Cited by 28 publications

References 3 publications

Spontaneous Remission in Cushing's Disease

Spontaneous Remission in Cushing's Disease

Prolonged Remission in Florid Cushing's Syndrome Following Metyrapone Treatment

Spontaneous remission of hypercortisolism presumed due to asymptomatic tumor apoplexy in ACTH-producing pituitary macroadenoma

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