Abstract
Cryptorchidism is associated with a higher risk of malignancy, infertility, and torsion. Torsion of an intra-abdominal testis is a rare cause of acute abdominal pain in the post-pubertal male but must be considered in men presenting with abdominal pain and a history of cryptorchidism. We present an unusual case of a patient with acute abdominal pain found to have torsion of a left intra-abdominal testis and his management.
Keywords: Intra-abdominal testis, Cryptorchidism, Testicular torsion, Undescended testis, Laparoscopic orchidectomy
1. Introduction
Cryptorchidism, or the undescended testis, is associated with an increased risk of malignancy and infertility. Patients with a cryptorchid testis are also at higher risk of testicular torsion, given the lack of normal fixation to the scrotum. Although uncommon, particularly in the post-pubertal male, torsion of a cryptorchid testis located within the abdomen can occur, presenting with non-specific abdominal pain. We present the unusual case and management of a patient presenting with acute abdominal pain found to have torsion of an intra-abdominal testis.
2. Case presentation
A gentleman in his forties presented to the emergency department with acute left lower quadrant abdominal pain. His pain was colicky in nature and associated with nausea. He had no urinary or bowel symptoms. He reported a medical history of a prior sleeve gastrectomy but was otherwise fit and well. On examination, he was acutely tender on palpation in the left iliac fossa.
Blood tests were performed which revealed normal inflammatory markers, serum lipase and liver function. He then underwent computed tomography (CT) imaging of his abdomen and pelvis which demonstrated a 2.8cm ovoid soft tissue structure in the left iliac fossa with fat stranding and twisting of the associated vessel (Fig. 1). On further examination, he was noted to have an empty left hemi-scrotum and reported having never had a left testis within the scrotum but could not remember having a previous surgery on his left testicle.
An ultrasound confirmed a left abdominal testis with tenderness on probe pressure and no vascularity on Doppler imaging (Fig. 2) and a normal right testis within the scrotum. Testicular tumour markers (β-human chorionic gonadotropin [β-hCG], lactate dehydrogenase [LDH] and alpha fetoprotein [AFP]) were performed which were all normal.
The patient underwent a diagnostic laparoscopy which identified a necrotic-appearing testicle above the opening of the deep inguinal ring with a twisted spermatic cord (Fig. 3a). The spermatic cord was ligated using ligating clips and transected (Fig. 3b), with the defect in the deep ring closed with a Vicryl® suture.
The patient had an uncomplicated recovery and was discharged home the day after his operation. Histopathology revealed changes consistent with an infarcted/torted cryptorchid testis with no evidence of dysplasia or malignancy.
3. Discussion
Cryptorchidism occurs in approximately 1–4% of full-term newborn males and 30% of premature male infants.1 Approximately 50% of males with cryptorchidism will have spontaneous descent of the testis within the first three months; if no descent occurs, preferred treatment is to attempt orchidopexy during late infancy or early childhood.1,2 Therefore, it is fairly uncommon to see males in the post-pubertal age group with a cryptorchid testis that has not had previous orchidopexy. If not within the scrotum, the testis is either ‘intra-abdominal’ (located proximal to the internal inguinal ring, near the iliac vessels or kidney), ‘intra-canalicular’ or ‘inguinal’ (located between the internal and external inguinal rings), suprascrotal (located between the mid-scrotal position and external inguinal ring) or ectopic (outside of the normal anatomic path of testicular descent); if the testis is not palpable, the majority of cryptorchid testis are intra-abdominal.1,3,4
There is an increased risk of malignancy and infertility associated with cryptorchidism, thought possibly due to the abnormal transformation of germ cells caused by heat stress.1,5 In males who have an intra-abdominal testis left untreated past the age of 11, the risk of malignancy is reported to be up to 30 to 35 times higher, with spermatogenesis virtually absent.1,5 Given these risks, the recommended management of a cryptorchid testis (with or without torsion) in a post-pubertal male is orchidectomy. Children with a cryptorchid testis (with or without torsion) are recommended to have orchidopexy, however in those with torsion, salvage rates are low (between 30 and 40%), thought to be due to non-specific symptoms leading to misdiagnosis and delayed treatment.6, 7, 8 The approach to patients (either pre- or post-pubertal) depends on the location of the testis.3,7 In those with a palpable testis (within the inguinal or suprascrotal region), an inguinal approach is preferred.3,7 In those with a non-palpable testis, an examination under anaesthetic may reveal a subsequent palpable testis and an inguinal approach can be used, however if still not palpable, laparoscopy is the gold-standard approach.2,3,7 Laparoscopic orchidectomy is associated with reduced pain, minimal blood loss and quicker recovery.2 There is also an increased risk of malignancy in the contralateral testis in men with a cryptorchid testis, and therefore patients should be counselled on regular self-examination of the remaining testis.1,9
Testicular torsion most commonly occurs in males under the age of 25.1 Torsion of an intra-abdominal testis is rare, particularly in a post-pubertal male, but there are approximately 52 cases reported in the literature.5,9,10 Only nine of these reports revealed benign histopathology; the other reports discovered testicular malignancy, with seminoma the most common pathology, but sarcoma, teratoma, mixed germ cell tumour, embryonal carcinoma, choriocarcinoma and carcinoma in situ also reported.11 Often patients present with non-specific abdominal pain, nausea, vomiting and anorexia, and may be incorrectly diagnosed as appendicitis, diverticulitis, or renal colic.5,9,10 A careful history and examination are required to elucidate a history of cryptorchidism given the non-specific symptoms patients present with. Given that malignancy appears to be commonly associated with intra-abdominal testicular torsion, it is important to ensure appropriate investigation for testicular malignancy is performed when managing intra-abdominal testis torsion so that suitable management is undertaken. Most patients undergo laparoscopic orchidectomy, however for large intra-abdominal testicular malignancies laparotomy has been performed and a recent case report was published where a patient underwent a robotic-assisted surgical exploration and orchidectomy for a torsion of a benign intra-abdominal testicle.9,10
4. Conclusion
While torsion of an intra-abdominal testis is rare, it needs to be considered as a differential diagnosis in any male presenting with generalised abdominal pain and a history of cryptorchidism. Given the increased risk of malignancy in patients with cryptorchidism, clinicians should consider an associated testicular malignancy when deciding on management and ensure appropriate investigations are performed prior to surgery. A minimally invasive approach to removing the testis is preferred if feasible.
Funding
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
CRediT authorship contribution statement
Katherine Ong: Conceptualization, Writing – original draft, Writing – review & editing. Kai Hellberg: Conceptualization, Supervision, Writing – review & editing. Steve P. McCombie: Supervision, Writing – review & editing.
Declarations of competing interest
None.
Appendix.
Fig. 1.
Axial CT scan showing an ovoid structure within the left iliac fossa with twisting of the associated vessel
Fig. 2.
Doppler Ultrasound demonstrating the left testicle with no vascular flow detected
Fig. 3.
a (top image): Intra-operative image of torted intra-abdominal testis
b (bottom image): Intra-operative image of ligated left spermatic cord
References
- 1.Sepúlveda M., Barceló F., Egaña P. The undescended testis in children and adolescents. Part 1: pathophysiology, classification, and fertility- and cancer-related controversies. Pediatr Surg Int. 2022;38:781–787. doi: 10.1007/s00383-022-05110-5. [DOI] [PubMed] [Google Scholar]
- 2.Kim J., Chua M., Ming J., et al. A critical review of recent clinical practice guidelines on management of cryptorchidism. J Pediatr Surg. 2018;53:2041–2047. doi: 10.1016/j.jpedsurg.2017.11.050. [DOI] [PubMed] [Google Scholar]
- 3.Chedrawe E., Keefe D., Romao R. Diagnosis, classification, and contemporary management of undescended testicles. Urol Clin. 2023;50:477–490. doi: 10.1016/j.ucl.2023.04.011. [DOI] [PubMed] [Google Scholar]
- 4.Hack W., Meijer R., Bos S., Haasnoot K. A new clinical classification for undescended testis. Scand J Urol Nephrol. 2003;37(1):43–47. doi: 10.1080/00365590310008686. [DOI] [PubMed] [Google Scholar]
- 5.Chung J., Lee S. Individualized treatment guidelines for postpubertal cryptorchidism. World J Mens Health. 2015;33(3):161–166. doi: 10.5534/wjmh.2015.33.3.161. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6.Naouar S., Braiek S., El Kamel R. Testicular torsion in undescended testis: a persistent challenge. Asian J Urol. 2017;4(2):111–115. doi: 10.1016/j.ajur.2016.05.007. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 7.Zhong H., Tang L., Bi Y. Cryptoorchid testicular torsion in children: characteristics and treatment outcomes. Asian J Urol. 2021;23:468–471. doi: 10.4103/aja.aja_10_21. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 8.Dupond-Athénor A., Peycelon M., Abbo O., et al. A multicenter review of undescended testis torsion: a plea for early management. J Pediatr Urol. 2021;17:191e1–191e6. doi: 10.1016/j.jpurol.2020.12.004. [DOI] [PubMed] [Google Scholar]
- 9.Nickalls O., Tan C., Thian Y. A torted ruptured intra-abdominal testicular seminoma presenting as an acute abdomen. J Radiol Case Rep. 2015;9(12):21–28. doi: 10.3941/jrcr.v9i12.2589. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 10.Taskovska M., Hawlina S. Intra-abdominal testicular torsion as a cause of acute abdominal pain in patient with silver-russell syndrome: first case of robot-assisted laparoscopic surgical exploration with orchidectomy. J Endourol Case Rep. 2020;6(4):332–335. doi: 10.1089/cren.2020.0065. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11.Hutcheson J.C., Khorasani R., Capelouto C.C., Moore F.D., Jr., Silverman S.G., Loughlin K.R. Torsion of intraabdominal testicular tumors. A case report. Cancer. 1996 Jan 15;77(2):339–343. doi: 10.1002/(SICI)1097-0142(19960115)77:2<339::AID-CNCR17>3.0.CO;2-2. [DOI] [PubMed] [Google Scholar]