Adenomatoid Tumor of the Adrenal Gland: Report of Two Cases and Review of the Literature

Adenomatoid tumor (AT) is an uncommon benign neoplasm of mesothelial origin, usually occurring in the female and male genital tracts. Extragenital localization such as the adrenal gland is extremely rare. Until now, only 39 cases of adrenal AT have been reported in the English literature. Here we report two novel cases of adrenal AT that occurred in male patients aged 30 and 31 years. The tumors were discovered incidentally by computed tomography (CT). Macroscopically, the tumors were unilateral and solid, and the greatest dimension of the tumors was 3.5 and 8.0 cm, respectively. Histologically, the tumors consisted of angiomatoid, cystic, and solid patterns and infiltrated the adrenal cortical or medullary tissue. The tumor cells had low nuclear/cytoplasmic ratio, with no pathological mitosis or nuclear pleomorphism. Thread-like bridging strands and signet-ring-like cells could be seen. Immunohistochemically, the tumor cells were positive for epithelial markers (AE1/AE3, CK7) and mesothelial markers (D2-40, calretinin, and WT-1). The Ki-67 index was approximately 1 and 2%, respectively. The differential diagnosis of adrenal AT includes a variety of benign and malignant tumors. The patients had neither local recurrence nor distant metastasis at 21 and 8 months after removal of the tumor. In the literature review, we comprehensively summarized the clinical, morphological, immunohistochemical, and prognostic features of adrenal AT. Adrenal ATs are morphologically and immunophenotypically identical to those that occur in the genital tracts. Combining the histology with immunohistochemical profiles is very supportive in reaching the diagnosis of this benign tumor, helping to avoid misdiagnosis and overtreatment.

Peritoneal well-differentiated papillary mesothelioma associated with infertility in a 37-year-old woman

Well-differentiated papillary mesothelioma (WDPM) is an uncommon mesothelial tumor. The lesions may be single or multiple and usually behave in a benign or indolent fashion, sometimes persisting for many years. In the present case, a 37-year-old woman had experienced primary infertility for 12 years, and a diagnostic laparoscopy was performed. Approximately 200 mL of dark red, free fluid in the pelvis and more than 10 yellow-white nodules on the surface of the right round ligament, sacrum ligament, right fallopian tube, and both sides of the uterus were found. A lesionectomy was performed and immunohistochemical markers indicated WDPM with adenomatoid tumor. The patient was monitored by computed tomography and serum CA125 (cancer antigen 125) levels for 49 months with no recurrence. WDPM and adenomatoid tumor are both benign tumors of mesothelial origin. Because of the lack of effective radical treatment, regular follow-up is sufficient. However, the effects of estrogen and progesterone on WDPM and adenomatoid tumors during ovulation or pregnancy remains unclear. Although WDPM is not life threatening, a strategy to fulfill the fertility requirements of women with this condition is a new challenge for infertility doctors.