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Ovarian serous cystadenoma with ectopic adrenal tissue in a 65-year-old patient: A case report

Highlights • A very rare case of ectopic adrenal tissue (EAT) with ovarian serous cystadenoma is presented.• EATs usually disappear by age, however it can remain until old stages of life.• Surgically removal of all suspicious lesions is strongly recommended.

Although ectopic adrenal tissue can also occur in female genital structures, ovaries are rarely involved. There are only a few cases of ovarian ectopic adrenal tissue reported in Medline and to the best of our knowledge this case is the fourth one [6,7,16]. In this case report we present a very rare localization of ectopic adrenal tissue in the ovary of a 65-year-old patient and also draw attention to the risk of neoplastic transformation [14].
This work has been reported in line with SCARE Criteria [17].

Presentation of case
A 65-year-old patient was admitted to our clinic with complaints of postmenopausal vaginal bleeding and progressive pelvic pain for almost 6 months. Hypertension and asthma were recorded in the history and physical examination was unremarkable with the exception of bilateral lower quadrant tenderness. revealed a uterus with homogenous myometrium and increased endometrial thickness of 7 mm. The adnexa included 5 cm ovarian cysts with a benign appearance. Initial endometrial sampling was performed to examine possible underlying pathologies and the result indicated benign endometrial histology. Subsequently hysterectomy and bilateral salpingo-oophorectomy were performed via laparotomy. Frozen sections and the final pathological diagnosis were benign, indicating serous ovarian cystadenoma of both ovaries. The pathologist also reported that there was ectopic adrenal tissue on the wall of the left ovarian cystadenoma. Ectopic tissue and the left ovary were stained with hematoxylin-eosin (Fig. 1). Peritoneal cytology was also benign and the patient was discharged from hospital 3 days after surgery in a good condition.

Discussion
The adrenal glands are an important component of the endocrine system that have two embryological origins. While the adrenal cortex arises from the mesoderm, the adrenal medulla develops from the ectoderm. During embryological development medullary tissue invades the cortex along the central vein [1,2]. Defects in this penetration process may lead to small fragments, which subsequently separate and migrate with descent of the gonads [2,3]. This theory explains why the most common site for ectopic adrenal tissue is the spermatic cord region. Other reported localizations are in the retroperitoneal area, kidney, liver, pancreas, placenta, broad ligament, ovary and testis [2][3][4][5][6][7][8][9][10][11][12][13][14][15].
Ectopic adrenal tissue has been widely reported in different mammalian species, while in humans it has been reported that approximately 50% of newborns present with hormonally inactive or asymptomatic ectopic adrenal tissue [12,13]. However, these ectopic adrenal tissues become atrophic with age due to normally functioning adrenal glands and very rarely occur in adulthood. In some cases ectopic tissue may be hormonally active and cause corticotropin-independent Cushing's Syndrome [4]. Neoplastic transformation of ectopic adrenal tissue has also been reported [14]. As a consequence, it has been suggested that ectopic adrenal tissues should be excised if incidentally encountered during surgery [15].
Ectopic adrenal tissue is less frequent in female genital organs than in males, especially at older ages. The reported cases of ovarian ectopic adrenal tissue are limited to the papers published by Usta et al., Kim et al. and Yasar et al. [6,7,16]. To the best of our knowledge the case presented herein will be the fourth report in the English literature. Another important feature of our case is related to the age of the patient, who was 65 years old. As mentioned above, ectopic tissue generally disappears due to normally functioning adrenal glands. In our case, despite the absence of endocrine symptoms, ectopic adrenal tissue presented at a late stage of life.

Conclusion
In conclusion, ectopic adrenal tissue is a very rare entity in elderly females. It may cause endocrine symptoms such as fasciotruncal obesity, hypertension and easy bruising, but it is generally asymptomatic and revealed incidentally during surgery. Although ectopic adrenal tissues are mostly nonfunctional, some cases have demonstrated the risk of neoplastic transformation. Therefore, surgeons must be aware of this rare entity, which bears the risk of malignancy, and surgically remove all suspicious lesions.

Conflict of interest
The authors report no conflict of interest.

Funding
No special funding.

Ethical approval
None.

Consent
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Author contributions
Cagdas Sahin, Enes Taylan; study concept, data analysis and interpretation, writing the paper.
Ali Akdemir, Osman Zekioglu, Pervana Seyidova; data collection, data analysis and interpretation. Ahmet M. Ergenoglu; study concept and design, data analysis, writing the paper.

Guarantor
Ahmet M. Ergenoglu MD is the guarantor of this study.