Double left sided cryptorchidism in a 3-year-old male: A case report of polyorchidism

Abstract

Polyorchidism is a rare congenital urogenital abnormality with a limited number of reports in the literature. The principal consideration in the management of polyorchidism is the prevention of future risk of malignancy. We present a case of a 3-year-old male presenting with left-sided cryptorchidism found to have a left-sided supernumerary testicle. He was treated with orchiectomy of both testes. While rare, the possibility of supernumerary testicle should be considered during surgery for cryptorchidism. Non-scrotal supernumerary testes should generally be excised to reduce the risk of malignancy.

1 Introduction

Polyorchidism is a rare congenital testicular anomaly with approximately 200 cases reported in the literature.

The most common variant is triorchidism with the presence of 2 testes on one side and a normal contralateral testis. Tetraorchidism has also been described, most commonly with 2 testes bilaterally, while a single case of 3 testicles unilaterally with a normal contralateral side has been reported once in the literature. The supernumerary testicle is most often left sided, found in the scrotum, and most commonly associated with other diagnoses such as inguinal hernia, cryptorchidism, or torsion being the most common. ^, In the available literature, malignancy is associated with a supernumerary testicle in approximately 5 % of examined cases. ^,

We present a case of incidentally discovered polyorchidism at the time of elective surgery for orchidopexy for cryptorchidism along with a discussion of the available literature. This manuscript was prepared following the CARE guidelines ( https://www.core-statement.org ).

2 Case presentation

The patient is a healthy 3-year-old male with an undescended left testicle noted at birth. He was referred for surgery for potential orchidopexy. On exam he had a normal right testis but a non palpable left testis. A preoperative ultrasound identified an undescended left testis within the left inguinal canal, but did not identify the left intrabdominal testicle. On the right side, there was a normal testicle with a 4mm extra-testicular mass abutting the testicle.

The patient was taken for a groin exploration, possible diagnostic laparoscopy and possible orchidopexy versus orchidectomy. Intraoperatively a standard groin dissection was carried out. Within the inguinal canal, structures consistent with a hernia sac, vas deferens, and vessels were identified. The hernia sac was isolated from the other cord structures and divided. Once divided, the hernia sac was followed proximally and further dissected to perform a high ligation of the sac. During the proximal dissection and with traction on the proximal hernia sac, a testicle emerged through the internal ring, from within the abdominal cavity. This structure appeared to be a normal but small testicle. It measured 1.4×1.3×0.7 cm. This structure had an independent blood supply and but no macroscopically identifiable vas deferens entering this testicle.

At this point, the structure initially identified as the spermatic cord was followed both proximally and distally. Distally, the vas and vessels passed through the external ring down toward the scrotum. With traction distally, another structure was exteriorized into the wound. This was a diminutive structure, measuring 0.7×0.6×0.4 cm. It had the appearance of an atrophied testicle. This structure had a macroscopically identified blood supply as well as a spermatic cord. Distally, it was attached to what appeared to be gubernacular tissue. We then followed the initial set of cord structures proximally and they could be followed through the internal ring. These structures traveled in a separate direction from the first testis and its blood supply ( Fig. 1 ). At this point, we were left with a previously intra-abdominal testis with a blood supply but no macroscopically identifiable vas deferens along with a separate set of cord structures leading towards an inguinal structure that appeared to be an atrophied testicle likely from a previous torsion. We consulted 2 other paediatric surgeons (urology and general) who agreed with the findings and helped to formulate the subsequent operative plan.