Therapeutic Strategy

Case Review

CASE REVIEW Abdominal Pain Associated With an Intra-Abdominal Gonad in an Adult Christian Twiss, MD, Michael Grasso III, MD New York University School of Medicine, New York Controversy still surrounds management of cryptorchidism in adults. Options include orchiectomy, orchiopexy, and close observation. What about fertility if orchiectomy is chosen? How is the risk of cancer affected when observation is the choice? Follow the reasoning for the management strategy for this adult presenting with abdominal pain. [Rev Urol. 2000;2(3):178-181] Key words: Cryptorchidism • Fertility • Orchiectomy A 44-year-old man presented with right lower quadrant pain that had progressed in severity over a 3-month period. The pain radiated to his groin and was not alleviated by over-the-counter pain medications. His past medical history was significant for untreated right-sided cryptorchidism first noted in childhood. He had no previous medical problems and had fathered 5 children. The past surgical history and review of systems were noncontributory. On physical examination, the patient, an obese man, had mild right lower quadrant tenderness. The genitourinary examination revealed an empty right hemiscrotum with no palpable canalicular or ectopic gonad. The left testicle was descended and normal to palpation. Pertinent laboratory data indicated normal -fetoprotein levels and -human chorionic gonadotropin levels. Complete blood count, liver function tests, and urinalysis results were also normal. A CT scan of the abdomen and pelvis revealed a 3  4-cm intra-abdominal testicle inferior to the lateral border of the right rectus muscle and superior to the external iliac vessels at the level of the internal inguinal ring (Figure 1). In light of the patient’s symptoms, laparoscopy was performed to visualize the intra-abdominal testicular mass and potentially perform a laparoscopic orchiectomy. The endoscopic findings confirmed that an intra-abdominal testicle was located at the internal inguinal ring (Figure 2). When the testicle was retracted medially, a looping vas deferens and hernial sac were noted. The spermatic vessels, vas deferens, and gubernaculum were clipped and divided. The defect in the peritoneum was repaired with an endostitch device, and the testicle was removed from the abdomen using an endosac. The patient went home the morning after the procedure and returned to full activity on postoperative day 7. Microscopic examination of the testicle revealed atrophy with an almost complete lack of germ cells (Figure 3). There was no evidence of neoplasm or carcinoma in situ. 178 REVIEWS IN UROLOGY SUMMER 2000 Cryptorchidism Discussion The management of cryptorchidism in postpubertal patients remains controversial. The 3 basic management choices are orchiectomy, orchiopexy, and close observation. The choice of therapy should take into consideration the risks associated with an undescended testicle, which include cancer and testicular torsion. If orchiectomy is considered, issues involved in loss of the testicle must also be addressed, including fertility and cosmetic changes. The presence of 1 normally descended testis makes hormonal function of minimal concern in unilateral cryptorchidism. Ultimately, the therapeutic strategy must be decided on an individual basis. One of the more important issues in the management of cryptorchidism in adults is the increased risk of testicular cancer. The possibility of cancer was considered in this case, because patients with carcinoma of an intraabdominal testicle commonly present with abdominal pain.1,2 The undescended testicle is 35 to 48 times more likely to become cancerous than a normally descended testicle3; 11% of all testicular tumors arise in undescended testes.4 Although tumors occur more frequently in these testes, they tend to occur between the third and fourth decades of life. The histologic distribution of these testicular tumors is comparable to testicular tumors occurring in intrascrotal testes.5 This increased risk of cancer has been used as a justification for orchiectomy in unilateral disease.5-7 With unilateral cryptorchidism, there is also an increased risk of cancer in the contralateral, normally descended testicle,3 which has prompted some authors to recommend increased cancer surveillance in all men with a history of cryptorchidism.8,9 While this seems reasonable, to our knowledge there are currently no studies demonstrating an effective follow-up strategy for these patients in terms of both cancer detection and cost-effective- Figure 1. An abdominal CT scan demonstrating a right-sided, intra-abdominal testicle. ness. In addition, the low absolute risk of cancer in patients who have undergone surgery for cryptorchidism has prompted some authors to challenge the requirement for special surveillance in these patients.10 Thus, it remains unclear whether there is a need for special surveillance in men who have had surgery for cryptorchidism, and, if there is a need, exactly what methods of surveillance should be implemented. It has been estimated that torsion, a rare but significant complication of undescended testes, occurs in 64% of intra-abdominal testicles that are cancerous.11 The increased size of the malignant testis can promote twisting of the testicle on its mesentery. The typical presenting symptoms of torsion of an undescended testicle are abdominal and inguinal pain that may be intermittent, prolonged, or of sudden onset.12 The possibility of carcinoma with torsion was initially considered in this case. Torsion can progress to infarction and rupture of the testicle, which can lead to hemoperitoneum and generalized peritonitis with a potentially lethal outcome.13 Thus, suspicion of torsion of an intra-abdominal testicle should be high in men presenting with abdominal pain and an empty hemiscrotum. The surgical and pathologic findings in this patient excluded the above scenarios. The nature of this patient’s pain remains unclear. It may have been due to the unfavorable location of the testis. Regardless of the cause, unresolving pain is a clear indication for surgical intervention in patients with cryptorchidism. While there are distinct risks associated with retaining an undescended testicle, there are also several concerns associated with removal. Fertility is an obvious concern, but it is probably not a significant reason for retaining an abdominal testicle in an adult with unilateral cryptorchidism. Sperm count and motility remain poor in unilateral undescended testicles, even after orchiopexy, in the postpubertal period.14 Moreover, because most undescended testicles produce no or negligible amounts of mature sperm after puberty,7 the intra-abdominal testicle contributes little with regard to fertili- SUMMER 2000 REVIEWS IN UROLOGY 179 Cryptorchidism continued Figure 2. Laparoscopic view of the intra-abdominal testicle showing its position at the internal inguinal ring. Figure 3. Microscopic view of the pathologic specimen demonstrating Leydig cell predominance and an almost complete lack of germ cells within the seminiferous tubules. ty. A recent study found that among men attempting to father children, failure to achieve paternity was twice as common in men with a history of unilateral cryptorchidism as compared 180 REVIEWS IN UROLOGY SUMMER 2000 with normal controls.15 It is notable, however, that this patient fathered 5 children, and that many oligospermic men are able to father children.16 This should be emphasized when dis- cussing fertility with men who elect orchiectomy for undescended testes. Ultimately, the current evidence suggests that the risk of cancer and torsion should take precedence over concerns about loss of fertility in these cases. Orchiectomy of an intra-abdominal testicle carries its own inherent surgical risk to the patient. Farrer and colleagues9 initially demonstrated that after 32 years of age, the risk associated with anesthesia is greater than the risk of death from germ-cell testicular tumors in men with a history of cryptorchidism. In their original manuscript, these authors recommended observation in patients older than 32 and orchiectomy in postpubertal patients with impalpable testes who were younger than 32 or unable to participate in surveillance of the testicle. More recently, the same authors now recommend either orchiectomy or orchiopexy for all patients with impalpable testes, regardless of age, and for patients with palpable undescended testes who are younger than 32 years of age.3 Close observation is reserved for men with palpable undescended testes who are older than 32.3 Indeed, these and other investigators agree that while orchiopexy is not therapeutic with regard to fertility, orchiopexy accompanied by surveillance of the testicle is an appropriate treatment in patients who for psychological or cosmetic reasons do not wish to undergo orchiectomy.8,9 In the present case, the significant pain, increased risk of cancer and testicular torsion, and the fact that the right testicle was impalpable justified an orchiectomy despite the fact that the patient was older than 32. We recommended a laparoscopic orchiectomy, a procedure that has been recognized as appropriate in the literature since the first reported procedure in 1992.17 While there are currently no controlled, clinical trials comparing laparoscopic and open orchiectomy, the cases reported in the literature Cryptorchidism Main Points • The risk of cancer is significantly higher in an undescended testicle than in a descended one. • Risk of torsion is also high in malignant intra-abdominal testicles. • Most undescended testes in adult men produce little or no mature sperm. • Pain, impalpability of the testicle, and increased risk of cancer and torsion are indications for orchiectomy. • Following unilateral orchiectomy, it is unclear whether there is need for close observation of the remaining testicle. suggest that blood loss, hospital stay, time to return to normal activity, and, most significantly, postoperative pain are minimized with this approach.17-19 The procedure is safe, and our experience with laparoscopic orchiectomy echoes the findings of these authors. In summary, we performed a laparoscopic orchiectomy on an adult presenting with abdominal pain associated with a unilateral intra-abdominal testicle. The indications for the procedure were intractable pain, the impalpability of the testicle, the risk of cancer, and the risk of torsion. In light of recent evidence, loss of fertility is not a major concern in adults with unilateral cryptorchidism, because the undescended testicles produce virtually no mature sperm. Whether these patients require increased surveillance of the remaining gonad after surgery and which methods should be used for cancer detection remain areas that require further investigation. ■ Acknowlegment We thank Jerry Waisman, MD, for preparing and photographing the pathologic specimen. References 1. Hogan P, Smedley H, Sikora K. Abdominal pain as a presenting symptom of male germ cell tumour. Br J Urol. 1985;57:197-199. 2. Raina V, Shukla NK, Gupta NP, et al. Germ cell tumours in uncorrected cryptorchid testis at Institute Rotary Cancer Hospital, New Delhi. Br J Cancer. 1995;71:380-382. 3. Rajfer J. Congenital anomalies of the testis and scrotum. In: Walsh PC, Retik AB, Vaughn ED Jr, Wein AJ, eds. Campbell’s Urology. Philadelphia: WB Saunders; 1998:2172-2191. 4. Abratt RP, Reddi VB, Sarembock LA. Testicular cancer and cryptorchidism. Br J Urol. 1992;70: 656-659. 5. Batata MA, Whitmore WF Jr, Chu FC, et al. Cryptorchidism and testicular cancer. J Urol. 1980; 124:382-387. 6. Ford TF, Parkinson MC, Pryor JP. The undescended testis in adult life. Br J Urol. 1985;57:181-184. 7. Rogers E, Teahan S, Gallagher H, et al. The role of orchiectomy in the management of postpubertal cryptorchidism. J Urol. 1998;159:851-854. 8. Taha SA, Abdulkader A, Kamal BA, Anikwe RA. Management of an unusually high postpubertal presentation of cryptorchidism. Int Surg. 1990; 75:105-108. 9. Farrer J, Walker A, Rajfer J. Management of the postpubertal cryptorchid testis: a statistical review. J Urol. 1985;134:1071-1076. 10. Pinczowski D, McLaughlin JK, Lackgren G, et al. Occurrence of testicular cancer in patients operated on for cryptorchidism and inguinal hernia. J Urol. 1991;146:1291-1294. 11. Riegler HC. Torsion of intra-abdominal testis: an unusual problem in diagnosis of the acute surgical abdomen. Surg Clin North Am. 1972;52:371374. 12. Williamson RC. Torsion of the testis and allied conditions. Br J Surg. 1976;63:465-476. 13. Radford PJ, Greatorex RA. Torsion of a malignant undescended testis mimicking appendicitis. Br J Clin Pract. 1992;46:209. 14. Grasso M, Buonaguidi A, Lania C, et al. Postpubertal cryptorchidism: review and evaluation of the fertility. Eur Urol. 1991;20:126-128. 15. Lee P, O’Leary L, Songer N, et al. Paternity after bilateral cryptorchidism. Arch Ped Adol Med. 1997;151:260-263. 16. Coughlin M. Fertility and subfertility among formerly cryptorchid men. In: Lee P, Ehrlich R, eds. The Undescended Testis: An Update. Dial Ped Urol 1999;22:1. 17. Beck RO, Nicholl P, Hickey NC, Black J. Laparoscopic excision of an intra-abdominal testis. Br J Urol. 1992;70:105-106. 18. Hennigan TW, Young RA. Laparoscopic orchidectomy in cryptorchidism. Br J Surg. 1992;79:1166. 19. Thomas MD, Mercer LC, Saltzstein EC. Laparoscopic orchiectomy for unilateral intra-abdominal testis. J Urol. 1992;148:1251-1253. LOOK FOR ON THE INTERNET AT MEDREVIEWS.COM FOR DETAILS, SEE PAGE 157 SUMMER 2000 REVIEWS IN UROLOGY 181