We describe the case of a 48-year-old quadriplegic black man with history o
f C4-C5 cervical spine and cord injury secondary to a fall, who presented t
o the University of Cincinnati Medical Center Urology Service with obstruct
ive symptoms at urination. A bulbous urethral stricture was diagnosed and s
ubsequently resected with primary urethral reanastomosis. On pathologic exa
mination, the surgical specimen contained an epithelioid leiomyoma at the s
ite of the urethral stricture. Although leiomyomas of the female urethra ar
e relatively common, we identified only 2 previously reported cases of leio
myomas of the male urethra in the English-language medical literature. To t
he best of our knowledge, we describe the third case of leiomyoma of the ma
le urethra, the first of the epithelioid type.