Virilization caused by an ectopic adrenal tumor located behind the iliopsoas muscle

Mavroudis, K.; Aloumanis, Kyriakos; Papapetrou, Peter D.; Voros, Dionysios; Spanos, Iraklis

doi:10.1016/j.fertnstert.2006.08.106

Cited by 7 publications

(10 citation statements)

References 21 publications

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“…Uncommonly, ectopic adrenal tissue can become clinically apparent. To our knowledge, only 2 cases of an ectopic cortical adrenal mass in the thorax and behind the iliopsoas muscle, producing strictly androgens, have been reported in adults [8,9]. Louiset et al [24] described an ACTH-independent Cushing´s syndrome with bilateral micronodular adrenal hyperplasia and ectopic adrenocortical adenoma.…”

Section: Discussionmentioning

confidence: 99%

“…The localization of the rather small tumors may be difficult, especially by abdominal ultrasound. These extremely rare tumors are exceptionally located bilateral or ectopic, but to our knowledge no concomitant ectopic bilateral adrenal adenomas causing virilization in children are described in the literature [8,9]. To verify that the ectopic lesion is from adrenal origin detecting the expression of the melanocortin 2 (MC2) receptor could be useful [10].…”

Section: Introductionmentioning

confidence: 99%

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Virilization of a Young Girl Caused by Concomitant Ectopic and Intra-Adrenal Adenomas of the Adrenal Cortex

Choukair

Beuschlein²,

Zwermann³

et al. 2013

Horm Res Paediatr

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Background: Adenomas of the adrenal gland are rare causes of virilization in childhood. Case Report: A girl aged 2 years and 4 months presented with pubarche, distinct clitoral hypertrophy, tall stature, and increased height velocity. Plasma testosterone and dehydroepiandrosterone were elevated. Androgens remained unchanged after adrenocorticotropic hormone, and dexamethasone administrations. Ultrasound examination and magnetic resonance imaging indicated an extra-adrenal mass adjacent to the left adrenal gland, which was removed by endoscopic surgery. However, plasma androgens remained elevated and ¹³¹I-iodomethyl-norcholesterol scintigraphy revealed tracer enhancement in the right adrenal gland, which was consecutively removed. Virilization regressed after extirpation of the adenomas and height velocity normalized. Results: Histology revealed a circumscribed adenoma in the right adrenal gland and an epithelial mass with adrenal cortical cells in the left-sided ectopic tumor. In the ectopic tumor, melanocortin 2 receptor expression was augmented threefold compared to the control, indicating adrenal origin. Conclusions: In this young girl, virilization is due to concomitant ectopic and intra-adrenal adenomas of the adrenal cortex. By melanocortin 2 receptor expression, it was confirmed that the ectopic adenoma derived from the adrenal cortex. Specific scintigraphy, if available, assists in allocating the source of androgen hypersecretion.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Virilization of a Young Girl Caused by Concomitant Ectopic and Intra-Adrenal Adenomas of the Adrenal Cortex

Choukair

Beuschlein²,

Zwermann³

et al. 2013

Horm Res Paediatr

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“…Moreover, given that radiologic findings can be nonspecific, false-positive results may occur that can lead to inappropriate clinical decisions. Mavroudis et al 23 have reported a case of persistent hyperandrogenism in a woman with an ectopic virilizing adrenal tumor, who had earlier been treated with an oophorectomy guided by a TVUS that showed a 1.6 cm dense area in the left ovary. Particularly in young women, preoperative localization of an androgen-secreting neoplasm is essential in order to plan adequate surgery.…”

Section: Discussion Of Diagnosismentioning

confidence: 99%

A premenopausal woman with virilization secondary to an ovarian Leydig cell tumor

et al. 2011

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“…At 13.5 dpc, these two primordia have separated completely [15]. Despite this complete separation, evidence that primordial adrenal cell cortex cells migrate together with the coelomic epithelial cells lies both in the incidental finding of ectopic remnants of adrenal cortex tissue along the spermatic cord during inguinoscrotal operations of boys [16,17] and in the identification of corticoadrenal rest tissue and adrenal tumors in the broad ligament and in the ovaries of females [18,19,20,21]. These findings suggest that the difference in adrenal rest tumor incidence between males and females may be due to differences in the ontogenesis of the gonads, and not to differences in the migration of aberrant adrenal tissue.…”

Section: Discussionmentioning

confidence: 99%

Ovarian Adrenal Rest Tumor in a Congenital Adrenal Hyperplasia Patient with Adrenocorticotropin Hypersecretion following Adrenalectomy

Tiosano

Vlodavsky²,

Filmar

et al. 2010

Horm Res Paediatr

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Objective: Ovarian adrenal rest tumors (OARTs) are rare in contrast to testicular adrenal rest tumors. We report a case of OART in a patient with congenital adrenal hyperplasia who developed Nelson’s syndrome after bilateral adrenalectomy. Methods: We describe the clinical, imaging, and laboratory findings of the patient and review the relevant literature regarding OART and the possible interaction between ACTH and brown adipose tissue. Results: An 18-year-old female with congenital adrenal hyperplasia, who had undergone bilateral adrenalectomy at the age of 10 years, presented with severe hyperpigmentation and hirsutism. Rectal ultrasonography showed a mass in the right ovary. ¹⁸F-fluorodeoxyglucose PET/CT revealed intense uptake both in this mass and in brown adipose tissue located in typical supradiaphragmatic sites. Laparoscopic removal of the ovarian mass confirmed the diagnosis of OART. A systematic review revealed 9 documented cases of OART. As in our case, all presented with elevated ACTH levels. Conclusions: Common to all documented cases of OART are sustained high ACTH levels that activate the adrenal anlagen tissue in the ovaries.

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Virilization caused by an ectopic adrenal tumor located behind the iliopsoas muscle

Cited by 7 publications

References 21 publications

Virilization of a Young Girl Caused by Concomitant Ectopic and Intra-Adrenal Adenomas of the Adrenal Cortex

Virilization of a Young Girl Caused by Concomitant Ectopic and Intra-Adrenal Adenomas of the Adrenal Cortex

A premenopausal woman with virilization secondary to an ovarian Leydig cell tumor

Ovarian Adrenal Rest Tumor in a Congenital Adrenal Hyperplasia Patient with Adrenocorticotropin Hypersecretion following Adrenalectomy

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