To the editor: Ectopic adrenal tissue is common in male surgery, especially near the gonadal area; however, it is extremely rare in adult females.1,2 In this study, we present a 46-year-old woman with a mass of ectopic adrenal tissue in the retroperitoneal region.
A 46-year-old Chinese woman presented to the outpatient clinic with complaints of irregular menstruation for over two years. During the gynecological body examination, a 6 cm×5 cm×4 cm mass was touched in the left lower quadrant near the ovary. Transvaginal sonography (TVS) revealed a 5.9 cm×4.5 cm×5.0 cm cystic mass on the left posterior of the uterorectal pouch, which was presumed to be ovarian cyst. During the laparoscopy examination, a 6 cm×5 cm×4 cm cystic mass was found outside the left peritoneum in the pelvic cavity, close to side of the left adnexa. The dull red mass with abundant blood supply was completely resected after electric coagulation hemostasis. After the wall of the smooth cyst was cut open postoperation, chocolate viscous liquid and a little dull red tissue was exposed. The entire cystic tissue was delivered and sent for biopsypathological examination (Figure 1A).
The submitted cystic tissue was packaged by a mutilayer fiber outer membrane that had a loose connection with the mass. Hematoxylin and eosin (HE) staining showed trabecular pattern cells with eosinophilic cytoplasmic staining without any nuclear atypia, either atypical mitosis, with some small cell nests of scanty basophilic cytoplasm in the central portion, as seen in the zona fasciculata and reticularis (Figure 1B and 1C). The immunohistochemistry examination (IHC) showed a diffused and strong cytoplasmic expression of pan-cytokeratin (P-CK), within the zona glomerulosa and zona fasciculata on the side close to the capsule, indicating its epithelial origin (Figure 1D). On the other side away from the envelope, diffused cytoplasmic chromogranin A (CgA) expression was visible at the zona reticularis (Figure 1E). The stains for neuron special enolase (NSE) were detected in reticularis cells at widespread cytoplasm and some nucleus, but were negative in vascular cells (Figure 1F). The IHC stains showed obvious cytoplasmic positive reaction for synaptophysin (SYN) at an inboard zone of the zona fasciculata and zona reticularis, especially at the zona fasciculata (Figure 1G). Positive stains of S-100 were detected in the zona fasciculata (Figure 1H). These findings confirmed the neuroendocrine function of this mass. The stains for epithelial membrane antigen (EMA), HMB45, DES, Melan-A, and SMA were negative, and Ki-67 was less than 1%. The Ki-67 cell index size and uniformity of the cells contributed to exclude pheochromocytoma and teratoma. Based on cell morphology, neuroendocrine function, and epithelium-derived cytokeratin, the pathologic diagnosis of the cyst was ectopic adrenal tissue. Recalling history, although indications of serum hormone levels, blood pressure, and other symptoms were absent, ectopic adrenal tissue does not cause clinically significant abnormalities consistent with the reported literature. One year after operation, no side effects were noted and no abnormal clinical manifestations were observed.
Ectopic adrenocortical tissue is a relatively common benign lesion and can be found anywhere along the gonadal descent during pediatric surgery, with a rather higher detection ratio in children and men than in adults and women.1,3 Among previously published studies on ectopic adrenal tissue in women, only one case was located at the retroperitoneal region, which was identified as adrenocortical carcinoma histologically.4
1. Dobanovacki DS, Maric DM, Maric DL, Vuckovic N, Jokic RR, Slavkovic AR, et al. Ectopic adrenocortical tissue: an incidental finding during inguinal surgery in children. Fetal Pediatr Pathol 2013; 31: 32-37.
2. Mares AJ, Shkolnik A, Sacks M, Feuchtwanger MM. Aberrant (ectopic) adrenocortical tissue along the spermatic cord. J Pediatr Surg 1980; 15: 289-292.
3. Mendez R, Tellado MG, Somoza I, Liras J, Sanchez-Abuin A, Pais E, et al. Ectopic adrenal tissue in the spermatic cord in pediatric patients: surgical implications. Int Braz J Urol 2006; 32: 202-207.
4. Yokoyama H, Adachi T, Tsubouchi K, Tanaka M, Sasano H. Non-functioning adrenocortical carcinoma arising in an adrenal rest: immunohistochemical study of an adult patient. Tohoku J Exp Med 2013; 229: 267-270.