Pituitary Adenoma Showing Intermittent Secretion of High Molecular Weight Adrenocorticotropin without Evidence of Cushing’s Disease

Hashimoto, Kozo; Kaneda, Tadafumi; Nagano, Isao; Asaba, Koichiro; Takeda, Kyoko; Takao, Tosihiro

doi:10.1159/000023431

Cited by 7 publications

(3 citation statements)

References 18 publications

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“…Molecular analysis of circulating ACTH was performed by gel chromatography as previously reported [4,5]. Briefly, the plasma sample was extracted using SEP-PAK C 18 cartridge (Waters, Milford, MA).…”

Section: Analysis Of Preoperative Plasma Acth By Gel Chromatographymentioning

confidence: 99%

Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

et al. 2005

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Abstract. We report the case of 19-year-old woman with cyclical Cushing's disease, in whom plasma adrenocorticotropin (ACTH) was secreted periodically after her first pregnancy. Since the 33rd week of pregnancy, hypertension and proteinuria became clinically remarkable. She gave normal birth at 36th week of pregnancy; however she continued to gain body weight even after delivery and developed typical Cushingoid features. Her ACTH secretion lacked normal daily fluctuation but exhibited periodic change during 1-year observation, showing 119 pg/ml, 34.6 pg/ml and 115 pg/ml at the 4th, 7th and 13th months after delivery. Plasma ACTH levels were increased by corticotropin releasing hormone and metyrapone, while low-dose dexamethasone suppressed cortisol secretion. Gel filtration analysis of the patient's plasma detected big ACTH molecules being eluted with a peak of authentic 1-39 ACTH. Cranial magnetic resonance imaging revealed a 1-cm pituitary mass in right cavernous sinus. The pituitary tumor was removed by transsphenoidal surgery at 13th month after delivery and was pathologically compatible with ACTH-producing pituitary adenoma by immunohistochemistry. This case includes clinically rare subsets of Cushing's syndrome showing periodic ACTH secretion and aberrant ACTH molecules.

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Section: Analysis Of Preoperative Plasma Acth By Gel Chromatographymentioning

confidence: 99%

Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

et al. 2005

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“…1A and B). On her endocrine examinations, plasma ACTH level was markedly increased (196 to 280 pg/ml; normal, 9-52) but the serum cortisol level was not remarkable (23 to 27 mg/dl; normal [8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24][25]. Daily secretion of ACTH and cortisol lacked the normal circadian rhythm (Fig.…”

Section: Case Reportmentioning

confidence: 99%

“…Analysis of molecular size of circulating immunoreactive (IR)-ACTH was performed by gel chromatography using a method similar to the one we previously reported [24]. The patient plasma collected after surgery was used for the ACTH analysis.…”

Section: Analysis Of Molecular Size Of Plasma Acthmentioning

confidence: 99%

An Aberrant ACTH-producing Ectopic Pituitary Adenoma in the Sphenoid Sinus

et al. 2004

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Abstract.A 32-year-old woman with an ectopic adrenocorticotropic hormone (ACTH)-secreting pituitary adenoma (EAPA) is presented. She had rapidly gained weight and suffered recurrent facial acne for a few years but lacked the typical Cushingoid features. Endocrine examinations revealed that her plasma ACTH was markedly high (196 to 280 pg/ml) without showing normal circadian rhythm and failed to respond to corticotropin-releasing hormone stimulation. Her cortisol levels ranged from 22 to 30 mg/dl throughout observation but low doses (1 and 2 mg) of dexamethasone failed to suppress either ACTH or cortisol level. Magnetic resonance imaging study revealed a 3-cm mass occupying the sphenoidal sinus with partial enhancement by gadolinium, which was separated from the normal pituitary in the sella region. The tumor resected by transsphenoidal surgery was histologically diagnosed as an ACTH-producing pituitary adenoma. After surgery her weight gain and acne remitted in accordance with decreases in plasma ACTH. Analysis of patient plasma by gel filtration method revealed the existence of big ACTH molecules eluted with a peak of authentic 1-39 ACTH, suggesting that this biologically less-active ACTH might be the reason why overt features of Cushing's syndrome failed to develop in this case. Although EAPA is clinically rare in parasellar disorders, the presence of ectopic pituitary adenoma should be considered in such cases showing ACTH hypersecretion without typical Cushingoid features.

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Effect of cabergoline treatment on Cushing’s disease caused by aberrant adrenocorticotropin-secreting macroadenoma

Miyoshi

Otsuka

Takeda

et al. 2004

J Endocrinol Invest

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The present case involves a 47-yr-old woman with Cushing's disease due to pituitary macroadenoma. The patient had suffered from hypertension and obesity for two yr. Her serum cortisol levels were moderately elevated throughout the observation period, and dexamethasone failed to suppress the cortisol secretion. Plasma ACTH levels were markedly high (>100 pg/ml) and did not respond to CRH provocation. Gel filtration analysis of the patient's plasma detected the existence of big ACTH molecules, which eluted with a peak of authentic 1-39 ACTH. Cranial magnetic resonance imaging (MRI) revealed a 3 cm pituitary tumor occupying the sellar region and right cavernous sinus with diffuse enhancement by gadolinium. The pituitary mass was removed by transsphenoidal surgery, and was pathologically identified as compatible to ACTH-producing pituitary adenoma by immunohistochemistry. RT-PCR analysis of total cellular RNA extracted from the resected adenoma revealed a relatively high expression level of dopamine D2 receptor (D2R) mRNA. Therefore, a long-acting D2R agonist, cabergoline (0.25 to 0.5 mg/week), was administered for the remnant adenoma, which gradually reduced ACTH levels in 90 days. In addition, cranial MRI exhibited shrinkage of the remnant pituitary mass after a 6-month treatment with cabergoline. This case demonstrates the efficacy of cabergoline to treat Cushing's disease caused by pituitary macroadenoma secreting aberrant ACTH molecules.

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Pituitary Adenoma Showing Intermittent Secretion of High Molecular Weight Adrenocorticotropin without Evidence of Cushing’s Disease

Cited by 7 publications

References 18 publications

Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

Periodic Secretion of Adrenocorticotropin in a Patient with Cushing's Disease Manifested during Pregnancy

An Aberrant ACTH-producing Ectopic Pituitary Adenoma in the Sphenoid Sinus

Effect of cabergoline treatment on Cushing’s disease caused by aberrant adrenocorticotropin-secreting macroadenoma

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