Diphallia or penile duplication is a rare congenital malformation of the penis. Reported incidence is 1 in every 5 to 6 million births. Diphallia are about 100 cases have been reported worldwide. Pseudodiphallia is a type of diphallia wherein a small accessory penis-like appendages attached to the main penile structure. Diphallia may be associated with urogenital, musculoskeletal, lumbosacral and gastrointestinal anomalies.

Herein presenting a 12-year-old male was brought to the urology outpatient clinic for external genital malformation. The patient previously underwent anoplasty for an imperforate anus. Physical examination noted an extra accessory glans penis inferior and ventral to the right side of the main glans penis and distal penile hypospadias was observed. Intraoperatively, penile degloving was performed followed by artificial erection. Chordee correction was performed by dorsal plication and creation of glans wings. Followed by urethroplasty and barrier flap using tunica vaginalis and glansplasty was done.

There are only about 7 reported cases of pseudodiphallia available. The proposed etiology is the lack or failure of fusion or branching of mesodermal anlagen of the genital tubercle by 4 or 5th week of gestation.

Pseudodiphallia is a rare congenital urologic condition and should be evaluated in detail to exclude any systemic anomalies. Treatment should be individualized based on careful considerations of functionality, ethical concerns, and cosmesis.