Virilization of a postmenopausal woman by a mucinous cystoadenoma

Abstract

ObjectiveTo describe the case of the most hyperandrogenaemic ovarian mucinous cystadenoma reported to date.MethodsWe present the clinical, laboratory and radiologic findings in a patient with an unusual diagnosis according to age and the clinical behaviour of the tumour, as well as a review of relevant literature.ResultsA 77-year-old women came to our consult because of fronto-parietal alopecia and an augmentation of the abdominal perimeter since 1 year ago. Clitoromegaly was observed during the physical examination. Hormonal analysis showed elevated testosterone and dehydroepiandrostenerone-sulphate levels (659 ng/dL and 1950 ng/ml, respectively), and imaging examination described an andexal cystic mass dependent on the right ovary. Pathological diagnosis was “mucinous cystoadenoma”. After surgery, clinical and analytical alterations were normalized.ConclusionAlthough ovarian mucinous cystadenomas are classically classified as “nonfunctional” tumours, they exceptionally can act as functional, and produce testosterone levels as high as directly secreting hormones or germ cell tumours.

Postmenopausal virilization secondary to a large ovarian cystadenoma with stromal hyperthecosis

“…Accessory sources of androgen production should also be considered, most notably the adrenal glands 9 which, in this case, were negative for abnormalities on CT and MRI. Our patient, as a postmenopausal individual, therefore represents a truly rare and unique presentation with only 4 previously existing reported cases to the best of our knowledge 5‐7,10 . We have highlighted the salient features of these cases in Table 2.…”

“…Ovarian cystadenomas are traditionally considered “nonfunctional” 7 and therefore associated symptoms are generally secondary to compressive effects on adjacent organs such as the urinary bladder, colon, small bowel, ureters, and abdominal wall. In rare circumstances, such as seen here, these tumors may be a source of androgen secretion, a phenomenon more commonly occurring in adolescent and pregnant women 8 .…”

“…Our patient, as a postmenopausal individual, therefore represents a truly rare and unique presentation with only 4 previously existing reported cases to the best of our knowledge. 5 , 6 , 7 , 10 We have highlighted the salient features of these cases in Table 2 . These previously reported cases have ranged from 68–80 years of age and where clinical information is available, alopecia is a common feature.…”

“…Similarly, stromal cell stimulation by the increased blood flow demand of these giant tumors or secretion of certain factors or antibodies from these tumors that may mimic the effects of LH could possibly explain these unique findings. 5 However, in the absence of direct evidence and uniformity (one case had decreased LH levels 7 ), these theories remain questionable. Another theory highlighted by Okolo et al 6 postulates that the pressure effect of these masses may induce luteinization and functional activity within certain areas.…”