A Gonadotrophin Responsive Testosterone Producing Adrenocortical Adenoma and High Gonadotrophin Levels in an Elderly Woman

Lange, W; Pratt, John J.; Doorenbos, H

doi:10.1097/00006254-198007000-00015

Cited by 9 publications

(11 citation statements)

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“…There are also reports about malignant androgen producing LH-dependent adrenal tumors (Givens et al 1975, de Lange et al 1980, Leinonen et al 1991. Hence, LH/hCG dependent mechanisms do exist among human adrenal pathologies, LH/hCG dependent mechanisms exist and they may be clinically important in certain circumstances, e.g.…”

Section: Page 4 Of 24mentioning

confidence: 99%

“…While there are differences between these TG mouse adrenal tumors and human adrenocortical tumors, certain similarities are noteworthy. Even if rare, the human adrenocortical tumors are gonadotropin responsive (Givens et al 1975, de Lange et al 1980, Leinonen et al 1991 (also see above). They may also express some common neuroectodermal and steroidogenic enzymes like in the case of transgenic mice (Kananen et al 1996, Kananen et al 1997 potentially useful for identifying factors critical for adrenocortical tumorigenesis in humans and a very useful model for human adrenocortical cancer research.…”

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confidence: 99%

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Adrenocortical tumorigenesis, luteinizing hormone receptor and transcription factors GATA-4 and GATA-6

Vuorenoja¹,

Rivero-Müller²,

Kiiveri³

et al. 2007

Molecular and Cellular Endocrinology

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Section: Page 4 Of 24mentioning

confidence: 99%

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confidence: 99%

Adrenocortical tumorigenesis, luteinizing hormone receptor and transcription factors GATA-4 and GATA-6

Vuorenoja¹,

Rivero-Müller²,

Kiiveri³

et al. 2007

Molecular and Cellular Endocrinology

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“…Since the luteinizing hormone (LH)/hCG receptor was shown to be expressed in the reticularis zone of adrenal glands (15), an aberrant expression of this receptor has also been suggested in some cases of CS (14,16,17) and in several cases of pure androgen-secreting adrenal adenoma (18)(19)(20)(21)(22)(23)(24) or hyperplasia (25) responding to LH or hCG stimulation in vivo and/or in vitro.…”

Section: Introductionmentioning

confidence: 99%

Food-dependent Cushing's syndrome: from molecular characterization to therapeutical results

Albiger

Occhi²,

Mariniello³

et al. 2007

European Journal of Endocrinology

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Objective: Cortisol secretion in ACTH-independent macronodular adrenal hyperplasia (AIMAH) may be regulated by the aberrant expression of several G-protein-coupled receptors. Bilateral adrenalectomy is the treatment of choice in most cases. We searched for aberrant receptor expression in a patient with AIMAH and evaluated the response to medical and surgical treatment. Patient: A 35-year-old woman with amenorrhea, hirsutism, and hypertension presented ACTHindependent cortisol secretion with high androgen levels. Abdominal computed tomography showed bilateral adrenal macronodules (4.5 cm right and 1.0 cm left). Scintigraphy with I 131 -norcholesterol showed bilateral uptake, prevalent on the right side. Several in vivo stimulation tests were assessed before and after treatment and in vitro studies were performed after unilateral adrenalectomy. Results: Plasma cortisol increased after a standard meal test (60%) and oral glucose loading (147%), and the response was blunted by pretreatment with 100 mg s.c. octreotide. The therapy with longacting release octreotide (octreotide-LAR) showed an improvement in urinary free cortisol (UFC) levels. Unilateral adrenalectomy was performed and histopathology revealed macronodular AIMAH. Cortisol and androgens increased after perifusion of tumoral tissue with glucose-dependent insulinotropic polypeptide (GIP), and GIP and LH-receptor overexpression was found in both the adrenal nodules and the adjacent cortex. After surgery, UFC and androgen levels normalized followed by clinical improvement. Conclusions: GIP and LH-receptor expression may coexist in AIMAH, influencing the functional and morphological phenotype. Aberrant hormone receptor expression enables specific pharmacological treatment, but long-term studies are needed to evaluate its real efficacy. Unilateral adrenalectomy may be a safe initial option, particularly for asymmetric bilateral adrenal enlargements.European Journal of Endocrinology 157 771-778

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“…However dynamic hormonal testing has been challenged as a means of differentiating between the two sources of hormone production in view of varied tumor responsiveness to hormones and subset of gonadotrophin responsive tumors [4,7,8]. The results of dynamic hormonal testing can be misleading, as too many unnecessary oophorectomies have been performed in literature when the real culprit was the adrenal tumour [4,5,[15][16][17]. Currently the best method to differentiate between adrenal and ovarian source is by modern radiologic techniques such as Ultrasonography, CT Scan, MRI and venous sampling [1].…”

Section: Discussionmentioning

confidence: 99%

Pure androgen secreting adrenal adenoma: a case report and review of literature

Gangkak¹,

Verma²,

Agarwal³

et al. 2015

Clin Nephrol Urol Sci

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Pure androgen secreting Adrenal adenoma is exceptionally rare. Till now only around 20 cases of exclusively androgen secreting adrenal adenomas have been reported in English literature. These tumours may present with virilisation, hirsutism, menstrual abnormalities and even infertility. Various hormonal characteristic including rare presentation of responsiveness to gonadotrophins have been reported. We present here our rare case of virilising purely androgen secreting adrenal adenoma in a 32 year old female and review of literature.

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A Gonadotrophin Responsive Testosterone Producing Adrenocortical Adenoma and High Gonadotrophin Levels in an Elderly Woman

Cited by 9 publications

References 0 publications

Adrenocortical tumorigenesis, luteinizing hormone receptor and transcription factors GATA-4 and GATA-6

Adrenocortical tumorigenesis, luteinizing hormone receptor and transcription factors GATA-4 and GATA-6

Food-dependent Cushing's syndrome: from molecular characterization to therapeutical results

Pure androgen secreting adrenal adenoma: a case report and review of literature

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