Cosecretion of Estrogen and Inhibin B by a Feminizing Adrenocortical Adenoma: Impact on Gonadotropin Secretion

Kuhn, J M

doi:10.1210/jc.87.5.2367

Cited by 17 publications

(16 citation statements)

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“…The proaC form of inhibin thus constitutes a novel and specific serum tumor marker for ACC. In contrast, serum inhibin A and B levels did not differ between patient groups, although three ACC patients did have increased serum levels of inhibin B, as was previously described in two case reports (38,39).…”

Section: Discussionsupporting

confidence: 72%

Serum inhibin pro-αC is a tumor marker for adrenocortical carcinomas

Hofland

Feelders

Wal

et al. 2012

European Journal of Endocrinology

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Objective: The insufficient diagnostic accuracy for differentiation between benign and malignant adrenocortical disease and lack of sensitive markers reflecting tumor load emphasize the need for novel biomarkers for diagnosis and follow-up of adrenocortical carcinoma (ACC). Design: Since the inhibin a-subunit is expressed within the adrenal cortex, the role of serum inhibin pro-aC as a tumor marker for ACC was studied in patients. Methods: Regulation of adrenal pro-aC secretion was investigated by adrenocortical function tests. Serum inhibin pro-aC levels were measured in controls (nZ181) and patients with adrenocortical hyperplasia (nZ45), adrenocortical adenoma (ADA, nZ32), ACC (nZ32), or non-cortical tumors (nZ12). Steroid hormone, ACTH, and inhibin A and B levels were also estimated in patient subsets. Results: Serum inhibin pro-aC levels increased by 16% after stimulation with ACTH (PZ0.043). ACC patients had higher serum inhibin pro-aC levels than controls (medians 733 vs 307 ng/l, P!0.0001) and patients with adrenocortical hyperplasia, ADA, or non-adrenocortical adrenal tumors (148, 208, and 131 ng/l, respectively, PZ0.0003). Inhibin pro-aC measurement in ACC patients had a sensitivity of 59% and specificity of 84% for differentiation from ADA patients. Receiver operating characteristic analysis displayed areas under the curve of 0.87 for ACC vs controls and 0.81 for ACC vs ADA (P!0.0001). Surgery or mitotane therapy was followed by a decrease of inhibin pro-aC levels in 10/10 ACC patients tested during follow-up (PZ0.0065). Conclusions: Inhibin pro-aC is produced by the adrenal gland. Differentiation between ADA and ACC by serum inhibin pro-aC is limited, but its levels may constitute a novel tumor marker for ACC.

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Section: Discussionsupporting

confidence: 72%

Serum inhibin pro-αC is a tumor marker for adrenocortical carcinomas

Hofland

Feelders

Wal

et al. 2012

European Journal of Endocrinology

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“…To our knowledge, there is a single adrenal adenoma described in the literature in which E 2 and inhibin B cosecretion was determined [10]. The patient described in that study presentedafeminizing adrenocortical tumor and the pathological examination confirmed an adenoma.…”

Section: Discussionmentioning

confidence: 89%

“…Gonadotropin secretion may be modified in some adrenocortical carcinoma cases with functioning lesions, in the presence of excessive tumor testosterone or estrogen production. In the rare adrenocortical tumor cases described with excessive tumor estrogen production, gonadotropin levels were reported as being suppressed [9, 10, 18]. Bouraïma et al [18] described a patient with a feminizing adrenocortical adenoma, wherein LH and FSH pulsatility was completely abolished, indicating the fundamental participation of E 2 in the control of gonadotropin secretion, although the inhibin was not investigated in this case.…”

Section: Discussionmentioning

confidence: 92%

“…Virilization occurs in 20–30% of adults with functional adrenal neoplasms, whereas it is the most common hormonal syndrome in children with adrenocortical tumors [6,7,8]. Feminization, as a pure hormonal syndrome, is a quite rare manifestation of adrenocortical neoplasms [5,9,10]. In this report, we describe the first inhibin B and estrogen-secreting adrenocortical carcinoma leading to a selective FSH suppression in a male.…”

Section: Introductionmentioning

confidence: 99%

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An Inhibin B and Estrogen-Secreting Adrenocortical Carcinoma Leading to Selective FSH Suppression

et al. 2006

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Objective: Hormone-secreting adrenocortical tumors are frequently associated with endocrine syndromes. We describe a 30-year-old man who had abdominal pain, a nodule in the right breast and loss of libido. Abdominal magnetic resonance imaging revealed a very large tumor in the right adrenal gland. Methods: Hormonal profile disclosed increased levels of estradiol and slightly low testosterone levels. The basal and stimulated LH levels were normal, whereas basal and stimulated FSH levels were totally suppressed. Cortisol and adrenal androgen levels were normal. The unusual finding of selective FSH suppression suggested secretion of inhibin B by the adrenocortical tumor. A very high level of serum inhibin B (405 pg/ml) was demonstrated by ELISA assay. Right adrenalectomy and nephrectomy were performed and the tumor was classified as a malignant tumor (Weiss score: 7.0) and unilateral mastectomy disclosed a lipoma. Results: One week after surgery, a GnRH-stimulation test disclosed normal basal and stimulated FSH levels and low levels of inhibin B and estradiol. Immunohistochemical analysis with anti-B-inhibin antibody revealed intense staining in the adrenocortical tumor cells. One month after surgery, an abdominal magnetic resonance imaging revealed a local recurrence of the tumor and a second surgery was performed with partial resection of the tumor and the patient died 1 year after the first surgery. Conclusion: We herein report the first inhibin B and estradiol-secreting adrenocortical carcinoma. The unusual selective inhibition of FSH secretion should be considered a valuable hormonal finding for the diagnosis of inhibin B-secreting adrenocortical tumors.

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“…Inhibin production by adrenocortical cortisol-or estrogen-secreting tumors has been observed in vivo (219,220,221). The main endocrine consequence of this process is inhibition of pituitary FSH release (219,220). It is not known whether inhibin may also affect tumor steroidogenesis through paracrine mechanisms or not.…”

Section: Factorsmentioning

confidence: 99%

MECHANISMS IN ENDOCRINOLOGY: Autocrine/paracrine regulatory mechanisms in adrenocortical neoplasms responsible for primary adrenal hypercorticism

Lefebvre

Prévost

Louiset

2013

European Journal of Endocrinology

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A wide variety of autocrine/paracrine bioactive signals are able to modulate corticosteroid secretion in the human adrenal gland. These regulatory factors, released in the vicinity of adrenocortical cells by diverse cell types comprising chromaffin cells, nerve terminals, cells of the immune system, endothelial cells, and adipocytes, include neuropeptides, biogenic amines, and cytokines. A growing body of evidence now suggests that paracrine mechanisms may also play an important role in the physiopathology of adrenocortical hyperplasias and tumors responsible for primary adrenal steroid excess. These intra-adrenal regulatory systems, although globally involving the same actors as those observed in the normal gland, display alterations at different levels, which reinforce the capacity of paracrine factors to stimulate the activity of adrenocortical cells. The main modifications in the adrenal local control systems reported by now include hyperplasia of cells producing the paracrine factors and abnormal expression of the latter and their receptors. Because steroid-secreting adrenal neoplasms are independent of the classical endocrine regulatory factors angiotensin II and ACTH, which are respectively suppressed by hyperaldosteronism and hypercortisolism, these lesions have long been considered as autonomous tissues. However, the presence of stimulatory substances within the neoplastic tissues suggests that steroid hypersecretion is driven by autocrine/paracrine loops that should be regarded as promising targets for pharmacological treatments of primary adrenal disorders. This new potential therapeutic approach may constitute an alternative to surgical removal of the lesions that is classically recommended in order to cure steroid excess.European Journal of Endocrinology 169 R115-R138

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Cosecretion of Estrogen and Inhibin B by a Feminizing Adrenocortical Adenoma: Impact on Gonadotropin Secretion

Cited by 17 publications

References 0 publications

Serum inhibin pro-αC is a tumor marker for adrenocortical carcinomas

Serum inhibin pro-αC is a tumor marker for adrenocortical carcinomas

An Inhibin B and Estrogen-Secreting Adrenocortical Carcinoma Leading to Selective FSH Suppression

MECHANISMS IN ENDOCRINOLOGY: Autocrine/paracrine regulatory mechanisms in adrenocortical neoplasms responsible for primary adrenal hypercorticism

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