PMC10352618_01
A coronal abdominal CT post-contrast reveals a uterus (Ut) and vagina (Va) dorsal to the urinary bladder (UB) and ventral to the colon. A contrast-filled urethra is also visualized to the right of the vagina. The uterine horns are visualized extending caudally (asterisks).
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F1
A complex, lobulated cystic structure arising from the right dorsal margin of the prostate was present (Figure 1). Since recovering from surgery, the patient's clinical symptoms have resolved (Supplementary Figure 1).
(A) Intraoperatively, the uterus (Ut) was identified as dorsal to the urinary bladder (UB) with two fluid-filled horns (asterisks) that extended through the inguinal rings into the scrotum (Sc). A rudimentary cervix delineated the uterus from the vagina (Va). (B) The uterine horns were found to envelop the spermatic cords following excision. (C) An intraoperative fluoroscopic contrast cystourethrogram identified the prostatic utricle (arrow) as a communication between the prostatic urethra (Ur) and the vagina (Va) (Note: to the left side of the image dorsal to the urinary bladder (UB), a radiopaque surgical sponge is observed). (D) The vagina (Va) was dissected to identify and occlude communication with the prostatic urethra. With a digital expression of the urinary bladder, communication was evident between the vagina and prostatic urethra via a urine stream (arrow).
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F2
The uterus was identified with two moderately distended fluid-filled horns extending toward the inguinal rings (Figure 2A). The uterine body had a thin membranous band (suspected rudimentary cervix), demarcating a division between the uterine body and the vagina before continuing down to the level of the prostate (Figure 2A). The testes and spermatic cord were enveloped within the descending uterine horns tracing through the inguinal canal (Figure 2B). Under fluoroscopic guidance, a contrast cystourethrogram evidenced two small ostia at the level of the colliculus seminalis that communicated with the uterine/vaginal stump (Figure 2C, Supplementary Video 2). The dorsal aspect of the stump was sharply incised distally, and the bladder was infused with sterile saline and expressed, allowing gross visualization of the Ostia (Figure 2D).
The reproductive tract of the patient. (A) Cross-section of the rudimentary uterine horn consisting of an epithelial-lined (arrowhead) lumen (Lu) containing necrotic cellular debris, a subepithelial stroma (Ss), smooth muscle layers (Sm), a serosa (asterisk), and an intramuscular rudimentary ductus deferens (arrow). Hematoxylin and eosin (HE). (B) Stratified squamous epithelium (Se) and loose collagenous subepithelial stroma (Ss) of the rudimentary vaginal tissue HE. (C) Rudimentary ductus deferens with a lumen (Lu) lined by a pseudostratified columnar epithelium (Ce) HE. (D) Testis with multifocal atrophied seminiferous tubules (At) lined by a single layer of Sertoli cells (arrowhead) and the seminiferous tubules with germinal epithelial degeneration characterized by a disorganized arrangement of germ cells (arrow) HE.
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F3
On histologic examination, the rudimentary uterine body and uterine horns (uterus masculinus) consisted of tubular structures (Figure 3A) lined by round to polygonal to columnar epithelial cells, multinucleated giant cells, and multifocal ulceration. The rudimentary vagina was lined by a one to five-cell thick, stratified, polygonal to squamous epithelium (Figure 3B). Deep to the epithelium, the rudimentary uterine body, uterine horns, and vagina were composed of a loose collagenous subepithelial stroma, an inner circumferential layer of smooth muscle, an outer longitudinal layer of smooth muscle, and serosa (Figure 3A). Cross-sections of rudimentary ductus deferens were randomly interspersed throughout the smooth muscle layers of the rudimentary uterus (Figures 3B, C) and the vagina and within the connective tissue adjacent to the right testis and epididymis. Within the left and right testes, <10% of the seminiferous tubules had evidence of complete spermiogenesis, and <10% of the seminiferous tubules were atrophied and lined by a single layer of Sertoli cell (Figure 3D). The remaining seminiferous tubules had evidence of germinal epithelial degeneration with a disorganized arrangement of germ cells (Figure 3D) and rare, intraluminal, multinucleated spermatids.