Ectopic aldosteronoma associated to another adrenocortical adenoma in the adrenal gland of the same side

Mazza, Enrico; Papotti, Mauro; Durando, R; Robecchi, A; Camanni, F.

doi:10.1007/bf03349816

Cited by 11 publications

(7 citation statements)

References 20 publications

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“…Adrenal cortical adenomas have been described in the retrocaval region, spinal canal and suprarenal. [8][9][10] To our knowledge this is the first case of an ectopic virilising tumour that was …”

Section: Discussionmentioning

confidence: 84%

Case report: Virilisation of initially obscure origin in a postmenopausal woman

Renckens¹,

Zant²,

Hasper³

et al. 2005

BJOG

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In 2003, a 63-year-old female patient was presented to the gynaecological clinic. Her main complaint was of severe hirsutism, for which she had undergone several intense pulsed light hair removal therapy sessions since 1999. In 2002, she had consulted an ENT specialist because of a deepening of the voice, which was particularly annoying to her because she sang in a choir. She had undergone an abdominal hysterectomy and left oophorectomy in 1983 because of a benign non-hormone secreting cyst. Physical examination revealed clitoromegaly and severe hirsutism, albeit partly masked by the light therapy. The right ovary was not enlarged on pelvic examination nor visible on transvaginal ultrasound examination. Laboratory tests showed an elevated plasma testosterone of 14 nmol/l (normal value 0.3-3 nmol/l) and normal levels of thyroxine, thyroid stimulating hormone, cortisol, adreno-cortico-trophic hormone, prolactin, human chorionic gonadotrophin, progesterone, oestradiol and dihydroepiandrostendione sulphate. The luteinising and follicle stimulating hormone levels were in the postmenopausal range. A differential diagnosis of an androgen-producing tumour in the ovary or the adrenal cortex was made.A computerised tomography scan could not confirm an adrenal adenoma, ovarian tumour or any other tumour so it was decided to first remove the right ovary laparoscopically. To our disappointment, an atrophic ovary with only a small, simple cyst was found. Histology was normal and the plasma testosterone-level remained elevated.In our continued search for of a testosterone-producing tumour, a PET scan and a 131 I-norcholesterol scan under dexamethasone suppression were subsequently performed. The PET scan showed a normal tracer uptake. 131 I-norcholesterol scan revealed a lesion in the right lower abdomen (Fig. 1). The integrated SPECT (low dose) CT scan localised the tumour anatomically: in the retroperitoneal region near the caecum and the ovarian vessels (Fig. 2). Revision of the first CT scan, with this knowledge in mind, demonstrated the initially overlooked small tumour.The patient was scheduled for surgery with the aid of a gamma probe. During operation, a black tumour of 2-2.5 cm, localised next to the caecum, was detected and extirpated. The use of the gamma probe was not very helpful because of physiological tracer uptake in the bowel.Histology showed a benign adrenal-type adenoma and post-operatively the plasma testosterone levels immediately returned to normal values. Although the hirsutism decreased, the clitoromegaly persisted, as expected. DiscussionHyperandrogynism is one of the most common endocrine disorders in women, affecting approximately 7% of reproductive-aged women. [1][2][3] The polycystic ovary syndrome is accounting for 80% of the cases of hyperandrogenism. 4 Virilisation, requiring very high levels of androgens, is very rare and usually the result of a tumour arising in the ovaries (arrhenoblastoma, adrenal and hilus cell tumours) or in the adrenal cortex. Ectopic virilising tumours are extremely rar...

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Section: Discussionmentioning

confidence: 84%

Case report: Virilisation of initially obscure origin in a postmenopausal woman

Renckens¹,

Zant²,

Hasper³

et al. 2005

BJOG

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“…Nevertheless, tumors arising from adrenal rests are on record, often benign and non-functioning. In a few cases, ectopic tissue may be directly responsible for Conn's syndrome [59] or ACTH-independent Cushing 's syndrome, due to adenomas [27,28,39,60,61] and exceptionally to carcinomas [32,62,63]. Indeed, adenoma is not easily differentiated from normal ectopic tissue, except for an aberrant hormonal production or a large size.…”

Section: Discussionmentioning

confidence: 99%

Adrenal Rests in the Uro-genital Tract of an Adult Population

et al. 2021

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Ectopic adrenal rests are a rare condition which can be found in various sites, generally in the retroperitoneum or pelvis along the path of gonadal descent. Their real prevalence is unknown. Males are more commonly affected, at least in the pediatric age. Adrenal rests are usually clinically silent and incidentally found in surgical samples, mostly in the pediatric population, and rarely in adults. With the aim of increasing knowledge and estimating the prevalence of ectopic adrenocortical tissue in the adult population, 44 adrenal rests in the urogenital tract of 40 adults are described. These represent approximately 0.07% of the total number of urogenital and gynecological surgeries performed in the 22 considered years. Adrenal rests were identified in the spermatic cord (10 males) and in paraovarian, parasalpingeal, or infundibulopelvic ligament locations (30 females). All but one was incidental findings. One case regarded an adrenocortical carcinoma arisen in adrenal rests. A literature review of adrenal ectopia in the urogenital tract of adults identified 57 reported cases from 53 patients, with similar clinicopathological features as those of our series, with the exception of a lower incidence of parasalpingeal locations. Despite their limited clinical implications, awareness of ectopic adrenal rests is essential also in adults for at least two reasons: (a) to correctly identify sources of adrenocortical hormone production in case of adrenal insufficiency or hormonal imbalance and (b) to avoid misinterpretations in the diagnostic workup of renal cell carcinoma, adrenocortical tumors, and rare gonadal neoplasms, including Sertoli/Leydig cell tumors.

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“…Few cases of hormonally active ectopic adrenal tumor have been described. For instance, Mazza et al found an ectopic aldosteronoma that was located in the retrocava location that were successfully treated by a conventional open approach 5 …”

Section: Discussionmentioning

confidence: 99%

A rare case of virilizing adult ectopic adrenal tumor

Dotto

Marx

Bastos

et al. 2019

Urology Case Reports

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Ectopic adrenal gland is an atypical medical condition with a prevalence of less than 1%. It occurs due to an abnormal gland development during embryonic life and can be found in different parts of the body. Rarely, these ectopic glands can be hormonally active and present as bulky masses. Herein we report a case of a patient who underwent laparoscopic surgery for a large myelolipoma associated with an androgen producing adenoma in an ectopic adrenal gland.

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Ectopic aldosteronoma associated to another adrenocortical adenoma in the adrenal gland of the same side

Cited by 11 publications

References 20 publications

Case report: Virilisation of initially obscure origin in a postmenopausal woman

Case report: Virilisation of initially obscure origin in a postmenopausal woman

Adrenal Rests in the Uro-genital Tract of an Adult Population

A rare case of virilizing adult ectopic adrenal tumor

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