A CASE OF DUPLICATION OF PENILE URETHRA By J. P. REIDY, F.R.C.S. Stoke Mandeville THIS congenital deformity is of rare occurrence. Gross and Moore (195o) summarised the findings of eighty-three cases. The accessory urethra can present :-- i. As a complete and separate passage from bladder to glans on the dorsum of the penis. 2. As an epispadias, i.e., with dorsal bladder opening and dorsal gutter. 3- As an incomplete reduplication extending from bladder outwards and ending blindly on the dorsum of the penis. 4. As an incomplete reduplication on the dorsum of the penis, extending: upwards and ending blindly. 5. As a complete reduplication ventral to the normal urethra. 6. As a" Y "urethra with union of true and accessory urethra: distal to external sphincter. 7- As a "Y " urethra opening at the penoscrotal junction, and joining the usual urethra proximal to this. Gross and Moore cite nineteen cases in which the accessory dorsal urethra has been complete, and show also the many variations that may occur in the formation of blind accessory urethra:. Arnold and Kaylor (1953) and Wrenn and Michie (1957) add two more cases of complete accessory dorsal urethra. Slotkin and Mercer (1953) also describe such a case. Clinieally.--Certain cases of double urethra must pass unrecorded, either because there is no apparent abnormality, or because no particular symptom has arisen requiring treatment. The commonest symptom is that of infection of the urethra either non-specific or gonorrheal. Once acquired, infection is more resistant to treatment in the accessory urethra. Patients with blind-ending accessory urethra: are likely to seek treatment only because of infection. Gross and Moore report that where two complete urethra: emerge from the bladder there is much less disturbance of urinary control than might be expected. Double streams have been reported without incontinence. Incontinence has been reported. In the present Case Report (J. S., 1941), little or no urine was voluntarily passed through the accessory urethra, probably due to the greater calibre of the normal urethra. There is no evidence of incontinence in this case, and therefore the sphincter mechanism was presumably intact. Slotkin and Mercer report a similar case of epispadias with double urethra. Wrenn and Michie report a case of complete double urethra with stress incontinence from the dorsal urethra. I99
200 BRITISH JOURNAL OF PLASTIC SURGERY Treatment.--Where infection is present this requires suitable treatment. Gross and Moore recommend excision of the blind urethra pouch or tunnel, and also state that it is unwise to attempt union between normal and accessory urethra. Treatment may also be advisable for cleft prepuce, for dorsal groove or other deformity, such as chordee. Excision of the complete accessory urethra is also recommended. Slotkin and Mercer excised the epispadias groove and urethra in their case, as also did Wrenn and Michie. Arnold and Kaylor were concerned to treat the gross dorsal penile curvature in their case, without risk of damage to the sphincter mechanism. They adopted 1952 FIG. I Double urethra. Catheters in dorsal epispadias and in ventral (normal) urethra. the procedure of lifting the whole dorsal urethra, relieving the dorsal curvature and replacing the dorsal urethra. Some recurrence of the dorsal curvature resulted. Meeter (I955) described treatment of one case of a blind accessory urethra by using fulguration. Case Report.--J. S., born I94I. Double Urethra: Normal ventral urethra. Complete dorsal accessory. On Examination (January I952).--Normal penile urethra with normal glandular meatus (Fig. i). Dorsal curvature of penis with dorsal groove leading proximally to an epispadias opening into the bladder--with continence. Pubic arch normal. Passes water from both tracts--9/io via normal urethra, I/IO via epispadias. Investigation.--Cystoscopy through normal urethra--ureters normal, trigone normal. Bougie passed through epispadias opening can be seen in bladder through the cystoscope --but there is a transverse bar of tissue between the two urethral openings. Discussion.--The late Professor J. Whillis favoured preservation of the dorsal urethra by reconstruction, because of risk to sphincter mechanism which might follow excision. Professor T. P. Kilner advised excision of the dorsal track. Decision taken to preserve the dorsal urethra.
A CASE OF DUPLICATION OF PENILE URETHRA 201 Operation I.--Dorsal curvature corrected and proximal portion of epispadias gutter converted into a dorsal urethra by down-turned flap for lining and lateral penile skin flaps for cover. Operation 2.--Distal part of dorsal urethra reconstructed using Denis Browne procedure (Fig. 2). Small fistula in dorsal urethra followedm2 cm. from tip of penis (Fig. 3). FIG. 2 Fro. 3 Fig. 2.--1952. Reconstruction of dorsal epispadiac urethra. Catheter in each urethra into bladder. Fig. 3.- 1952. After repair. Micturition from ventral urethra. Fig. 4.--I962. Penis showing normal glandular meatus, dorsal urethral meatus, dorsal urethral fistula. FIG. 4 Progress.--In January 1962 patient reports that he passes most urine from normal ventral urethra, few drops passed at times from the dorsal urethra. But since I956 there has been a painful purulent discharge from normal urethra, and purulent non-painful discharge from dorsal urethra. Infection is non-specific, and cleared up with treatment but tended to recur at intervals. Since 1958 there has been no discharge from the normal urethra, but discharge has persisted in the dorsal urethra. Erections and ejaculation are normal.
202 BRITISH JOURNAL OF PLASTIC SURGERY FIG. 5 FiG. 6 Fig. 5.--I962. Penis--catheter in each urethra into bladder. Fig. 6.--I962. X-ray of pelvis--showing catheter in each urethra entering bladder. FIG. 7 FIG. 8 Fig. 7.--1962. At operation. Dissection and excision of whole dorsal urethra up Fig. 8--1962. After operation. Dorsal suture line. to bladder. glandular meatus. Single normal urethra and
A CASE OF DUPLICATION OF PENILE URETHRA 203 On Examination (January i962).--normal ventral urethra with normal meatus on -the glans (Fig. 4). There is also a dorsal urethral opening at the coronal level under the prepuce. There is a small fistula in the dorsal urethra 2 cm. proximal to the tip of the penis. Testes are normal. Normal erection--with side twist. Investigation.--Smear from urethral discharge--diphtheroids, micrococci, and lactobacilli. Catheters were placed into both urethrae, and seen to enter the bladder separately (Figs. 5 and 6). Treatment (24th January I963).--At operation the dorsal penile skin was reflected to each side, and the whole dorsal urethra excised in toto. Dissection was carried up under the intact pubic arch to the urogenital diaphragm, ligated under traction and divided (Fig. 7). Dorsal penile skin was replaced. Indwelling catheter in the normal urethra for three days. 29th January I962. Urine culture negative. Satisfactory healing (Fig. 8). Histology of Excised Accessory Urethra.--" The small sinus-like tract is lined by a transitional type of squamous epithelium with no keratin formation. There is some.chronic inflammatory change in the wall, but no indication of malignancy." Embryology.--Slotkin and Mercer think that the anomaly is due to a continuation of the splitting process of the urorectal septum with a consequent bifurcation of the urethral anlage into a dorsal and ventral portion, partial or complete. SUMMARY A further case of complete dorsal accessory urethra with continence is described, and some points from the literature are reviewed. Features of this case are :-- I. The reconstruction of the epispadias urethra (at age I I years). 2. The very little passage of urine from the dorsal urethra. 3. The subsequent non-specific infection of both urethrae. 4. And finally excision (at age 2I years) of the whole dorsal urethra. REFERENCES ' ARBLASTER, J. (I959). Radiography, 25, II8. ARNOLD, M. W., and KAYLOR, W. M. (I953). J. Urol., 70, 746. GRoss, R. E., and MOORE, T. C. (1950). Arch. Surg., 6o, 749. MEETER, U. (I955). U.S. armed Forces reed. J., 6, 43 o. SLOTKIN, E. A., and MERCER, A. (I953). J. Urol., 7 o, 743- WRENN, E. L., and MICHIE, A. J. (I957). Ann. Surg., T45, IX9.