Pure Androgen-Secreting Adrenal Adenoma Associated with Resistant Hypertension

Rodríguez-Gutiérrez, René; Bautista-Medina, Mario Arturo; Teniente-Sanchez, Ana Eugenia; Zapata-Rivera, Maria Azucena; Montes-Villarreal, Juan

doi:10.1155/2013/356086

Cited by 9 publications

(6 citation statements)

References 18 publications

(23 reference statements)

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“…Rodríguez‐Gutiérrez et al . also reported that an 18‐year‐old woman with an adrenal tumor that produced only androgen presented with hirsutism, amenorrhea, hypertension, and elevated testosterone levels, and surgery on the adrenal tumor led to an improvement in symptoms 5 . Likewise, in the present case, hypertestosteronemia may potentially have been the cause of hypertension and heart failure in the patient.…”

Section: Discussionsupporting

confidence: 63%

Case of ovarian steroid cell tumor diagnosed after presenting acute heart failure

Matsuda

Yamaguchi

Kaseki

et al. 2020

J of Obstet and Gynaecol

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We report a rare case of an ovarian steroid cell tumor with a diagnosis prompted by heart failure symptoms. A 28-year-old Japanese nulligravida/nullipara with a chief complaint of respiratory discomfort during physical exertion and exhibiting heart failure symptoms was referred to our hospital. She also had signs of virilization, including secondary menorrhea since the age of 20, hirsutism and balding. Cushing's syndrome was suspected, and further examinations showed hypertestosteronemia and right ovarian tumor. Symptomatic treatment for heart failure with diuretics and antihypertensives was followed by abdominal right adnexectomy performed due to the androgen-producing ovarian tumor. The tumor was solid and larger than a fist, and confirmed as a steroid cell tumor through postoperative histopathology. Serum total testosterone levels normalized at day 3 postoperatively, and menstruation resumed 2 months later. Our case was diagnosed due to heart failure symptoms, and its treatment resulted in improvement in virilization signs.

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Section: Discussionsupporting

confidence: 63%

Case of ovarian steroid cell tumor diagnosed after presenting acute heart failure

Matsuda

Yamaguchi

Kaseki

et al. 2020

J of Obstet and Gynaecol

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“…Similarly, in transgender population studies, testosterone increased blood pressure [ 37 ]. An association of male sex steroids with the reduction of pressure natriuresis has been proposed previously as a contributing cause of hypertension [ 15 ]. In our review of childhood PASACT cases, only one patient had arterial hypertension, which normalized 2 months after surgery [ 15 ].…”

Section: Discussionmentioning

confidence: 99%

“…An association of male sex steroids with the reduction of pressure natriuresis has been proposed previously as a contributing cause of hypertension [ 15 ]. In our review of childhood PASACT cases, only one patient had arterial hypertension, which normalized 2 months after surgery [ 15 ]. In this case, the levels of 11-deoxycorticosterone, a potent mineralocorticoid, were normal.…”

Section: Discussionmentioning

confidence: 99%

Peripheral precocious puberty in Li–Fraumeni syndrome: a case report and literature review of pure androgen-secreting adrenocortical tumors

Ryckx,

De Schepper,

Giron

et al. 2023

J Med Case Reports

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Introduction Pure androgen-secreting adrenocortical tumors are a rare but important cause of peripheral precocious puberty. Case presentation Here, we report a pure androgen-secreting adrenocortical tumor in a 2.5-year-old boy presenting with penile enlargement, pubic hair, frequent erections, and rapid linear growth. We confirmed the diagnosis through laboratory tests, medical imaging, and histology. Furthermore, genetic testing detected a pathogenic germline variant in the TP53 gene, molecularly confirming underlying Li–Fraumeni syndrome. Discussion Only 15 well-documented cases of pure androgen-secreting adrenocortical tumors have been reported so far. No clinical or imaging signs were identified to differentiate adenomas from carcinomas, and no other cases of Li–Fraumeni syndrome were diagnosed in the four patients that underwent genetic testing. However, diagnosing Li–Fraumeni syndrome is important as it implies a need for intensive tumor surveillance and avoidance of ionizing radiation. Conclusion In this article, we emphasize the need to screen for TP53 gene variants in children with androgen-producing adrenal adenomas and report an association with arterial hypertension.

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“…The increased DHEAS was used as a marker of increased adrenal activity, so the circulating DHEAS level has been used to screen androgen from ovary or adrenal gland [2]. Functional adrenal tumors mainly include those secreting aldosterone or cortisol, and a few secreting both cortisol and androgen, but pure androgen-secreting adrenal tumors (PASATs) are rare, most of which are reported as case studies, and this kind of PASATs that only secrete testosterone with normal adrenal androgens (DHEA, DHEAS and androstenedione) are more rare [3]. Herein, we report a case of benign androgen-secreting adrenal tumor in a postmenopausal elderly woman, with markedly elevated testosterone levels while DHEAS and androstenedione were within normal range.…”

Section: Introductionmentioning

confidence: 99%

A rare case of pure testosterone-secreting adrenal adenoma in a postmenopausal elderly woman

Zhou¹,

Chen²,

Li³

2019

BMC Endocr Disord

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BackgroundHyperandrogenemia is more common in puberty and reproductive age, but relatively rare in postmenopausal women. Postmenopausal virilization may result from androgen-producing tumors. Androgen-secreting adrenal tumors are rare in clinical practice and are diagnosed as adrenocortical carcinoma, most of which can co-secrete androgen and cortisol. Highly elevated serum testosterone level with normal adrenal androgens such as dehydroepiandrosterone (DHEA), dehydroepiandrosterone sulfate (DHEAS) and androstenedione is usually regarded as ovary origin. Here we describe an unusual case of a postmenopausal woman with markedly elevated serum testosterone level, while DHEAS, androstenedione, 17-hydroxyprogesterone and cortisol were within the normal range.Case presentationA 67-year-old postmenopausal woman with hirsutism in the upper lip and armpit, accompanied by clitoromegaly for 5 months. Hormonal evaluation showed markedly elevated serum testosterone level (714.8 ng/ml), whereas DHEAS, androstenedione, 17-hydroxyprogesterone, and cortisol were within the normal range. Imaging examination showed a mass of 1.5 cm in diameter in the left adrenal gland and normal appearance of both ovaries. PET-CT indicated that it was a case of benign adrenal adenoma and excluded ovarian abnormalities and other ectopic tumors. Thus, a pure testosterone-secreting adrenal tumor was suspected and then adrenalectomy was performed. Histology and immunohistochemistry furtherly confirmed the benign adrenocortical adenoma with immunohistochemistry positive for inhibin α, melan A, β-captenin, SYN (focal), Ki-67(< 3%), and negative for chromogranin (CgA), cytokeratin (CK), S-100, P53. After surgery, the level of testosterone returned to normal range and the clinical symptoms also subsided.ConclusionsPure testosterone-secreting adrenal adenomas are extremely rare, but it can induce severe hyperandrogenism and virilization. The source identification of hyperandrogenemia only based on the levels of testosterone, DHEAS and androstenedione is limited. It is important to evaluate not only ovaries but also adrenals in all women with virilization particularly during menopause, even their androstenedione, DHEA and DHEAS level are normal.

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Pure Androgen-Secreting Adrenal Adenoma Associated with Resistant Hypertension

Cited by 9 publications

References 18 publications

Case of ovarian steroid cell tumor diagnosed after presenting acute heart failure

Case of ovarian steroid cell tumor diagnosed after presenting acute heart failure

Peripheral precocious puberty in Li–Fraumeni syndrome: a case report and literature review of pure androgen-secreting adrenocortical tumors

A rare case of pure testosterone-secreting adrenal adenoma in a postmenopausal elderly woman

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