An Unusual Cause of Inguinal Hernia in a Male Patient: Endometriosis

Abstract

Endometriosis is an ectopic endometrium, commonplaced in the female. But its occurence in the man is mysterious. In the literature two cases of endometriosis in men have occured following surgery.1 The present case is the first reported arising in the inguinal region, near the ductus deference. Forty-nine years old man was presented to hospital with left inguinal hernia. He was operated from that area for 3 times, before. Furthermore he has unexplained infertility. His physical examination revealed normal external genitelia. He underwent surgical operation for hernia. In the operation, a mass was discovered beside the spermatic cord and sent to our laboratory. Macroscopically the mass was 8×7×6 cm in diameters (Fig. 1). In dissection cystic cavity was seen and beside it, ductus deferens was identified. Cyst wall was containing small lumens that was thought as small vessels. In microscopic examination, cyst was lined with columnar epithelial cells and simple tubular invaginations showing the same type of cell lining, and cellular stroma, with typical features of endometrial mucosa. In addition, there was smooth muscle proliferation and some endometrial glands and stroma in these areas (Fig. 2). The pathological diagnosis was endometriosis. Chromosom analysis was normal (46, XY). The patient had an uneventful postoperative course and was discharged on the seven postoperative day. The patient decided on regularly follow-up in the out-patient department. Fig. 1 Uterus-like mass beside the deferent ductus, holded area in the figure. Fig. 2 High power field of endometriotic area (H&E stain, ×200). Three theories of the pathogenesis of endometriosis have been proposed in the female: transplantation, coelomic metaplasia, and embryologic cell rests.2,3 Transplantation theory is obviously impossible in the male because of the absence of a source of menstruel material.1 The coelomic metaplasia could occur secondary to inflammatory and hormonal influences. This theory could explain endometriosis in women with Mullerian agenesis, who have an absent uterus or the occasional presence of endometriosis in men.4 Third theory, also known as induction theory, is based on induction of the embryologic cell rests. Between the utricle and the appendices testes, cell rests may persist. These mullerian cell rests would be expected to lie along the course of the ejaculatory and deferent ducts.1 In this case, endometriosis was diagnosed along the ductus deferens; so this theory is very likely for this case. This patient has used a drug named of Fertilin, for sperm maturation and activation. The drug consists of clomiphen citrate that shows antiestrogenic affect by binding estrogen receptors in hypothalamus and hypophysis.5 It is a contrary event for the cases in the literature that were associated with prolonged estrogen therapy which appearing to be necessary for the development of endometriosis in the male. But also it is surprising not to have seen this phenomenon more frequently in men with prostatic carcinoma treated with estrogens. So, in our case additional factor must have been for the development of the lesion. In conclusion, endometriosis within an inguinal hernia is an exceptionally rare event; most surgeons are unlikely to see one. All previously reported elderly cases underwent estrogen therapy for prostatic carcinoma, contrary, our patient had antiestrogenic therapy for infertility.