黃裕賓¹、黃子豪^1,2、黃志賢^1,2

台北榮民總醫院 泌尿部¹；國立陽明大學醫學院 泌尿學科 書田泌尿科學研究中心²

Yu-Pin Huang¹, Tzu-Hao Huang ^1,2, William J. Huang^1,2

Department of Urology, Taipei Veterans General Hospital¹

Department of Urology, School of Medicine and Shu-Tien Urological Research Institute,

National Yang-Ming University, Taipei, Taiwan²

 

Adrenal ectopy is an extremely uncommon condition, which was caused by abnormal embryological development of the adrenal glands. Morgagni firstly described adrenocortical tissue noted in the spermatic cord of a child during cadaveric dissection in the eighteenth century¹. Due to Morgagni’s findings, accessory adrenal tissue may be found in at least 50 % of neonates and young children in some reports^1,2, but it regresses and disappears within time. Hence, the incidence of ectopic adrenal tissue is thought to be low in adult population. Senescende et al reported that twenty-one adult cases in his systematic review were all male, whose average age was 43.5 years old (range 19–78 years) at diagnosis³ Most lesions were noted on right side (71.4 %). The percentage of ectopic adrenocortical tissue found in hernia sacs and spermatic cord in his report was 47.6 and 33.3 %, respectively³. Here we report a female case of hernial sac with aberrant adrenocortical tissue.

 

A 70-year-old woman who denied systemic disease before presented with enlarged protruding mass over left inguinal area. She mentioned that the protruding mass had existed for over fifty years, which was accompanied with intermittent pain over left inguinal area after long walks. According to her statement, the mass became more prominent on standing or cough, while disappeared on lying down. Given her symptoms, she came to our urology out-patient clinics for help. Physical examination did not reveal incarcerated sign, and inguinal hernia was impressed. The laboratory findings were within normal range, except for mild anemia. Therefore, she underwent left hernioplasty four days later. During operation, a huge indirect hernia sac protruding from left internal inguinal ring was identified; in the sac we noticed a piece of yellowish soft tissue, which was suspected to be an ovary-like component. We consulted a gynecologist during operation, but he also could not sure if the tissue is ovary on appearance and palpation. The soft tissue was dissected meticulously from the hernia sac for pathologic examination. Histopathologic examination ultimately showed ectopic adrenal cortical tissue, which was positive for synaptophysin and negative for chromogranin A. Thus, the final pathologic report was hernial sac with aberrant adrenocortical tissue. She was discharged on post-operative day 1 and was stable on follow-up outpatient visit.

 

Few cases of hernial sac with aberrant adrenocortical tissue had been reported before, which was associated with abnormalities in embryogenesis. Currently, no investigations or treatments are indicated in resection, but pathological examination of any suspicious abnormalities of hernia sac is commended.

 

1. Schechter DC, et al. Ann Surg. 1968 Mar:167(3):421–426
2. Anderson JR, et al. Postgrad Med J. 1980 Nov;56(661):806-808
3. Senescende L, et al. Hernia. 2016 Dec;20(6):879-885