Abstract
BackgroundSteroid cell tumors of ovary account for less than 0.1% of all ovarian tumors and these tumours may present at any age in association with interesting presentations related to hormonal activities. The subtype, not otherwise specified (NOS), is associated with androgenic changes in 56-77% and Cushing syndrome in 6-10%. Due to the rarity of available data regarding these tumors, little is known about their malignant potential and metastatic behaviour. We hereby report an unusual metastasis of steroid cell ovarian neoplasm presented with both Cushing syndrome and hyperandrogenemia.Case presentationA 31-year-old woman, who had a past medical history of ovarian tumor resection (left ovarian thecoma was initially diagnosed at that time), presented with hirsutism, hypertension and menstrual disorder. Also, laboratory work-up revealed hypercortisolism and androgen excess. Computerized tomography (CT) of the abdomen showed abdominal paraaortic masses, multiple intrahepatic nodules and retroperitoneal lymph nodes enlargement. Positron emission tomography/computed tomography (PET/CT) scan demonstrated metastatic lesions. Her ovarian tumor sections were re-examined and pathology result was corrected to steroid cell tumor (NOS) associated with active cell growth and necrosis. Subsequent excision of metastatic lesions yielded clinical improvement promptly and metastasis of steroid cell tumor was confirmed by postoperative pathological studies. However, one year after the surgical management of metastasis, recurrence happened while radiotherapy was ineffective. The patient finally died of tumor metastatic recurrence.ConclusionThis case reports a rare coexistence of Cushing syndrome and hyperandrogenemia which occurs based on metastasis of steroid cell ovarian neoplasm. It presents a real diagnostic challenge to both clinicians and pathologists. Therefore, it is very important to establish a final diagnosis by pathological studies along with clinical manifestations and imaging findings. Besides, it is necessary to improve follow-up of patients with this kind of tumors.
Highlights
This case reports a rare coexistence of Cushing syndrome and hyperandrogenemia which occurs based on metastasis of steroid cell ovarian neoplasm
It is very important to establish a final diagnosis by pathological studies along with clinical manifestations and imaging findings
Further observation into the nature, biology and behaviour of the tumor may improve the understanding of therapeutic value of radiation or chemotherapy over time. This case reports a rare coexistence of Cushing syndrome and hyperandrogenemia which can occur based on ovarian pathology in females
Summary
This case reports a rare coexistence of Cushing syndrome and hyperandrogenemia which can occur based on ovarian pathology in females. It is very important to establish a final diagnosis by pathological studies along with clinical manifestations and imaging findings. This patient developed steroid-secreting metastasis three years after the excision of primary tumor, and one year after surgical management of metastasis, recurrence happened while radiotherapy was ineffective. These features of this case illustrate the necessity for this kind of tumor follow-up. All authors read and approved the final manuscript
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