Urethral amyloidosis is a rare condition in which eosinophilic amyloid proteins are deposited in the urethra. Only a small number of reports on urethral amyloidosis have been published. Increased interest has been associated with this disease due to its clinical similarities with urothelial carcinoma. A biopsy of the lesion and a histological examination are essential for the correct diagnosis. Conservative management has been suggested by various urologists as the optimal treatment approach for urethral amyloidosis; however, recurrence and urethral stricture are common, and typically further treatment is required. Urethroplasty has been used in a limited number of urethral amyloidosis cases, with beneficial short-term outcomes; however, long-term follow-up data are lacking. The present case report describes the cases of 2 patients with urethral amyloidosis who underwent urethroplasty without recurrence or progression for >2 years. These findings indicate that urethroplasty is beneficial for the long-term management of urethral amyloidosis.