ABSTRACT Objectives Megalophallus is a rare complication of priapism and sickle cell disease. More commonly, priapism in those patients is recurrent, difficult to prevent and ends up in erectile dysfunction as a result of cavernous tissue fibrosis or surgical intervention. A 33 year old man presented to us with a history of neglected priapism for 12 days 2 years previously. He developed a permanent unusual enlargement of the penis and erectile dysfunction. The patient determined that his new penile size is nonfunctional and unacceptable. We evaluated the patient and set out to surgically fashion the penis into a usable size, with adequate rigidity and stable condition. Material and Methods For 9 years the patient had frequent episodes of priapism which resolved spontaneously. The last episode was unsuccessfully treated conservatively and eventually the patient received a Winter shunt. The penis became enlarged and erections soft. Upon examination with intracavernous injection of 60 mg papaverine, the penis became mildly engorged and it measured 21 cm in circumference and 18 cm in length from the pubic pad of fat to the tip of the glans. MRI showed that the enlargement is mainly due to massive enlargement of corpora cavernosa. We based our goals on normative data for penile length and circumference. Under general anesthesia, we degloved the penis using a circumcision incision. The tunica albuginea was exposed laterally. A 3 cm wide strip was marked on the lateral side of each corpus cavernosum and excised along the long axis of the penis from 1 cm proximal to the coronal sulcus to the pubic bone. The cavernous tissue underneath was fibrotic and was excised to fit in the cylinders of an AMS 700 Ultrex penile prothesis. The tunica albuginea was run with an absorbable suture and the pump and reservoir were placed in the scrotum and retropubic space. Inflation of the prosthesis resulted in rupture of the suture line for 5 cm on the right side just proximal to the glans. Repeated removal of underlying cavernous tissue was not enough to accommodate the cylinder and a free tunica albuginea patch graft was sewn in. Results At the end of surgery, the penile length was left intact, while the circumference was reduced to 15 cm. Conclusion Reduction phalloplasty is feasible to manage the unusual cases of megalophallus. Extensive dissection and cavernous tissue removal necessitate penile prosthesis implantation. This is particularly justified in cases associated with erectile dysfunction complicating priapism. Disclosure Was this work supported by industry? No.