Primary Amenorrhea Accompanied by Adrenal Adenoma: Start of Menarche Soon after Tumor Resection.

Takahashi, Yoshiyuki O.; Ninomiya, Jun; Horiguchi, Jun; Shimizu, Hiroyuki; Sato, Minoru; Koibuchi, Yukio; Yoshida, Takashi; Yoshida, Miho; Takata, Daisuke; Odawara, Hiroki; Yokoe, Takao; Iino, Yuichi; Morishita, Yasuo; Mori, Masatomo

doi:10.2169/internalmedicine.41.972

Cited by 5 publications

(6 citation statements)

References 17 publications

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“…In human medicine, adrenal and ovarian venous sampling has been described to differentiate adrenal from ovarian production of progesterone. 13 In women, hyperprogesteronemia usually causes amenorrhea [13][14][15][16][17] ; the dog in Case 1 also had delayed oestrus (13 months, when its owners reported that it previously had regular oestrus every 6 months). The effect of progesterone on the mood and behaviour is not clear.…”

Section: Discussionmentioning

confidence: 99%

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Hyperprogesteronism associated with adrenocortical tumours in two dogs

Farges,

Crawford,

da Silva

et al. 2024

Vet Record Case Reports

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A 9‐year‐old, entire, female, soft‐coated Wheaten terrier and a 7‐year‐old, male, neutered greyhound were presented for clinical signs suggestive of hyperadrenocorticism (polyuria, polydipsia, and in the first case, polyphagia and weight gain). Adrenocorticotrophic hormone stimulation test and low‐dose dexamethasone suppression test ruled out excessive production of cortisol. Imaging (abdominal ultrasound and computed tomography) revealed an adrenal mass in both cases. Further hormonal testing revealed hyperprogesteronism. In the first case, surgical treatment was not considered due to vascular invasion of the mass, the patient was treated successfully with trilostane and was still stable 18 months later. In the second case, adrenalectomy was performed, and the clinical signs resolved, the dog was stable 10 months later. We report two clinical cases of progesterone‐secreting adrenocortical tumours and their management.

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Section: Discussionmentioning

confidence: 99%

“…There are only few reports of progesterone-secreting adrenocortical tumours in humans, with other intermediate steroids usually being also increased in these cases. [13][14][15][16][17] Clinical signs usually include menstrual disorders with amenorrhea or oligomenorrhea in women. There is no report of this tumour in a human male patient.…”

Section: Discussionmentioning

confidence: 99%

Hyperprogesteronism associated with adrenocortical tumours in two dogs

Farges,

Crawford,

da Silva

et al. 2024

Vet Record Case Reports

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“…Progesterone-producing adrenal tumours are extremely rare in humans, with four cases reported in women with secondary amenorrhoea or oligomenorrhoea. 34 – 37 …”

Section: Discussionmentioning

confidence: 99%

“…Progesterone-producing adrenal tumours are extremely rare in humans, with four cases reported in women with secondary amenorrhoea or oligomenorrhoea. [34][35][36][37] Mammary enlargement in male cats is rare and can be caused by neoplasia or fibroadenomatous hyperplasia (MFH). 38 MFH is a benign proliferation of ductal epithelium and mammary stroma, usually seen in young female cats.…”

Section: Discussionmentioning

confidence: 99%

Gynaecomastia in a male neutered cat with an adrenal tumour and associated hyperprogesteronism, hypercortisolism and hyperaldosteronism

Lenord²,

Lau

et al. 2021

Journal of Feline Medicine and Surgery Open Reports

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Case summary A 7-year-old male neutered domestic longhair cat was presented with chronic progressive gynaecomastia, polydipsia, polyphagia, weight loss and poor fur regrowth. Sexualised behavioural changes were not reported and virilisation was not present on physical examination. Pertinent haematology, biochemistry and urinalysis findings at the time of referral included mild hypokalaemia. Left adrenomegaly and mild prostatomegaly were identified on a CT scan. Evaluation of adrenal hormones with a low-dose dexamethasone suppression test, serum progesterone, testosterone, oestradiol, plasma aldosterone, renin, plasma metanephrine and normetanephrine measurement supported a diagnosis of hyperprogesteronism, hyperaldosteronism and hypercortisolism. Adrenalectomy was performed and histopathology was consistent with an adrenocortical tumour. Clinical signs and hormone elevations resolved postoperatively. Relevance and novel information To our knowledge, this is the second report of gynaecomastia secondary to an adrenal tumour in a male neutered cat and the first associated with hyperprogesteronism.

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“…Overall, however, the total number of the report of adrenal tumor producing mineralocorticoid intermediates was not so large even now. Progesterone producing adrenal tumors are even rarer [5,6]. Only two cases with progesterone producing adrenal tumors have been reported, and both presented with amenorrhea.…”

Section: Introductionmentioning

confidence: 99%

Close examination of steroidogenesis disorders in a DOC- and progesterone-producing adrenocortical carcinoma

Sone

Shibata

Homma

et al. 2008

Endocr

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We report a case of hypertension, hypokalemia, and amenorrhea accompanying an adrenocortical carcinoma. A 27-year-old woman was admitted to our hospital because of a left adrenal incidentaloma. She presented with hypertension, hypokalemia, and amenorrhea; her plasma renin activity was low, but her plasma aldosterone concentration was normal, as were cortisol and androgens. By contrast, her serum concentrations of deoxycorticosterone (DOC), 18-hydroxydeoxycorticosterone, and progesterone were high, and her urinary steroid profile showed elevated secretion of 17-deoxysteroids and 11-deoxysteroids (progesterone, DOC, 11-dehydrocorticosterone, and 11-deoxycortisol), and 3beta-hydroxy 5-en steroids (pregnenolone, 17-hydroxypregnenolone, and DHEA). Decreased ratios of metabolites of (1) 17-OHpregnenolone to pregnenolone and 17-OHprogesterone to progesterone, (2) corticosterone to DOC and cortisol to 11-deoxycortisol, and (3) progesterone to pregnenolone, 17-OHprogesterone to 17-OHpregnenolone and androstenedione to DHEA suggested the impairment of 17alpha-hydroxylase, 11beta-hydroxylase, and 3beta-HSD activities, respectively. After the tumor was removed, levels of all adrenal steroids were normalized. Based on the Weiss criteria, the tumor was diagnosed as an adrenocortical carcinoma, and immunohistochemical analysis of steroidogenic enzymes revealed disorganized steroidogenesis in the tumor tissue. With adrenocortical carcinomas, heterogeneity of individual steroid producing enzymes within tumor cells can lead to hypersecretion of various steroid intermediates, even when steroid end products are within the normal range.

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Primary Amenorrhea Accompanied by Adrenal Adenoma: Start of Menarche Soon after Tumor Resection.

Cited by 5 publications

References 17 publications

Hyperprogesteronism associated with adrenocortical tumours in two dogs

Hyperprogesteronism associated with adrenocortical tumours in two dogs

Gynaecomastia in a male neutered cat with an adrenal tumour and associated hyperprogesteronism, hypercortisolism and hyperaldosteronism

Close examination of steroidogenesis disorders in a DOC- and progesterone-producing adrenocortical carcinoma

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