An unusual case of quadruple polyorchidism in a human cadaver mimicking bilateral lipoma

Abstract

Polyorchidism is a rare congenital disorder defined as the presence of more than two testicles for which the underlying etiology is unknown. This report examines a novel case, correlates findings to the literature, and discusses potential mechanisms of dysgenesis. This study was conducted on a 96-year-old male cadaver. Meticulous skilled dissection was done with careful attention to the pelvis, the inguinal region, the scrotum and testes. Measurements of the scrotal testes (STs), supernumerary testes (SNTs), scrotal epididymides (SEs), supernumerary epididymides (SNEs), ductus deferens (DD) and supernumerary ductus deferens (SDD) were taken, and testicular volume was calculated. Sagittal sectioning of STs, SNTs, SEs and SNEs was performed and, using standard histologic protocol, tissues were processed, stained with H&E, and examined. Histological findings supported the anatomical suspicion of high, bilateral inguinal polyorchdism, or tetraorchdism, in this patient. STs and SNTs have normal, gross morphological features. The left SNT was supplied by the testicular artery, and the right SNT by the inferior epigastric artery. Both SNTs and SNEs were infiltrated with adipose tissue, whereas the scrotal homologues were histologically normal. Testicular volumes were 26.5 cm3 and 23.7 cm3, and 12.3 cm3 and 0.55 cm3, for the left and right STs and SNTs, respectively. The left and right SEs measured 7.2 cm and 7.0 cm; the left and right SNEs measured 4.7 cm and 1.7 cm, respectively. The left DD measured 40.2 cm from the scrotum. The SDD from the left SNT measured 5.9 cm and joined the main ductus 16 cm proximal to its origin. The right main DD measured 39.2 cm, and the SDD from the right SNT was 3.2 cm and joined the main DD 17.4 cm proximal to its origin. The current work suggests a novel classification for polyorchidism of Type 3, Subgroup B, or SNT attached to the draining epididymis and vas deferens without reproductive potential and SNT located outside the scrotal sac. Clinicians must correlate anatomical and histological findings in suspected polyorchids to avoid misdiagnosis of SNTs as benign lipomas.

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“…There are fewer than 200 human cases reported in medical literature. In addition, seven documented cases exist in the animal kingdom: two horses, two dogs, two cats, and a hummingbird (Roca-Ferrer et al, 2015;Tamminen et al, 2012;Witt and Bautista, 2011;Aziz et al, 2016;Talarico et al, 2022). In human polyorchids, diagnosis in patients ranges from 4 weeks to 75 years in age with a median age of 17 years (Mathur et al, 2002;Bergholz, 2009;Mittal et al, 2018;Talarico et al, 2022).…”

“…(Arslanoglu, 2013). Occasionally, there are some additional abnormalities seen in polyorchids that include testicular maldescent (40%), inguinal hernia (30%), testicular torsion (13%), hydrocele (9%), and malignancy (6%) (Mathur et al, 2002;Tonape et al, 2012;Talarico et al, 2022). Furthermore, polyorchidism is associated with an increased risk of testicular cancer (Talarico et al, 2018;Bergholz et al, 2009).…”

“…In the majority of polyorchid cases, the supernumerary testis (SNT) or testes (SNTs) are located on the left side with a 3:1 ratio (left > right) (Sheah et al, 2004). SNTs are typically smaller than the two normal testes (Artul and Habib, 2014;Cohen et al, 2017;Talarico et al, 2022). Prior research has shown that inguinal SNTs without reproductive potential tend to be infiltrated with adipose tissue with increased patient age (Talarico et al, 2022).…”

“…SNTs are typically smaller than the two normal testes (Artul and Habib, 2014;Cohen et al, 2017;Talarico et al, 2022). Prior research has shown that inguinal SNTs without reproductive potential tend to be infiltrated with adipose tissue with increased patient age (Talarico et al, 2022). This has led to the suggestion of a novel anatomical-functional classification in polyorchidism, Type 3, Subgroups A and B (Talarico et al, 2022).…”

“…Prior research has shown that inguinal SNTs without reproductive potential tend to be infiltrated with adipose tissue with increased patient age (Talarico et al, 2022). This has led to the suggestion of a novel anatomical-functional classification in polyorchidism, Type 3, Subgroups A and B (Talarico et al, 2022). The mechanism(s) of polyorchidism is unknown, but it is hypothesized to result from a malfunction or duplication of the gonadal ridge during embryological development, possibly involving molecular or genetic factors that predispose a patient to developing SNTs.…”