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Complete Duplication of Bladder and Urethra: A Case Report with Review of the Literature
Vol. 109, March Printed in U.S.A.
THE JOURNAL OF UROLOGY
Copyright © 1973 by The Williams & Wilkins Co.
COMPLETE DUPLICATION OF BLADDER AND URETHRA: A CASE REPORT WITH REVIEW OF THE LITERATURE J. P. SINGH, S. MEHRA
AND
V. NAGABHUSHANAM
From the Willingdon Hospital, New Delhi, India
through a racquet-shaped inc1s10n made in the mid-dorsal line of the proximal half of the penis. The urethra extended from the mid-dorsal shaft proximally and then dipped below and behind the symphysis pubis to become continuous with the accessory bladder. To expose the retropubic part of the accessory urethra, the vertical incision was extended proximally to join the suprapubic one. It was necessary to divide the pubic symphysis. The accessory bladder was continuous with and densely adherent to the normal bladder but the 2 cavities did not communicate with each other. The accessory bladder was 1 ½ inches in diameter and lobular in shape. While the bladders were being separated, the musculature of the normal bladder was shaved off at one site, thereby exposing the intact mucosa with the 2 bladders again separated anatomically. The gap in the bladder muscle was closed with catgut. There was no ureteral opening in the accessory bladder (figs. 2 and 3). Postoperatively the child experienced sepsis of the suprapubic wound for which he was given appropriate antibiotics. Histopathological examination of the accessory bladder and urethra revealed the specimen to be structurally compatible with normal bladder and urethra and to be lined with typical urothelium.
Duplication of the bladder and urethra is a rare anomaly. In complete duplication each urethra originates from a separate bladder; they pass down parallel to each other through 1 penis and have 2 external meatuses. Usually the anomalous urethral canal runs dorsal to the normal urethra, is narrow and its external meatus is situated somewhere between its normal location and the base of the penis. One of the 2 normal ureters or an accessory ureter may or may not open into the duplicated bladder. This anomaly may be associated with a duplicated genital system, colon or anus. To date 14 cases of complete duplication of the bladder and urethra have been described. In each of these cases there were other anomalies. CASE REPORT
A 6-month-old boy was admitted to the pediatric surgical service of this hospital on December 13, 1971 with intermittent purulent discharge from the dorsum of the penis occurring 3 times since birth. There were no associated urinary symptoms. Careful examination showed that the discharge came from a very small opening situated just beyond the middle of the dorsum of the penile shaft. Abdominal palpation (confirmed under general anesthesia) revealed a rounded, firm but nontender swelling 2 inches in diameter in the suprapubic region. Preoperative pus swab taken from the local site revealed Staphylococcus pyogenes sensitive to erythromycin, chloramphenicol and rovamycin. A probe could easily be passed into the sinus for 1½ to 2 inches inside the pelvis and well beyond the parietes. Contrast medium was injected into the opening via a polythene tube. After 10 cc of dye had been injected the abdominal swelling became more prominent. X-ray revealed a radiopaque irregular shadow in the suprapubic region in continuity with the accessory urethra (fig. 1).
DISCUSSION
Review of the literature on double bladder and urethra is confusing because many cases are not true cases of complete duplication and many reports are unclear. 1 After studying the literature we find that complete duplication of the urethra can be classified as follows: 1) Complete duplication of the urethra only-with or without associated congenital anomalies. Both urethras originate from 1 bladder through separate openings and pass down through 1 penis. 2 There are 40 cases of this type described in the literature. 2-• 2) Complete duplication of the urethra and bladder-with or without associated anomalies of the genital system, colon or anus.
TREATMENT
The child was given erythromycin for 3 days and underwent operation on December 22. Preoperatively, methylene blue was injected into the accessory urethra and bladder via a polythene tube. This stained the entire duplicated urethra and bladder. Via a suprapubic transverse incision the normal bladder as well as the overlying accessory bladder was exposed. The accessory bladder was adherent to the anterior surface of the normal bladder. The accessory urethra was dissected
1 Boissonnat, P.: Two cases of complete double functional urethra with a single bladder. Brit. J. Urol., 33: 453, 1961. 2 Casselman, J. and Williams, D. I.: Duplication of the urethra. Acta Urol. Belg., 34: 535, 1966. 3 Karanjavala, D. K.: An unusual case of complete reduplication of the urethra. Aust. New Zeal. J. Surg., 39: 284, 1970. •Tripathi, V. N. P. and Dick, V. S.: Complete duplication of male urethra. J. Urol., 101: 866, 1969.
Accepted for publication June 2, 1972. 512
COMPLETE DUPLICATION OF BLADDER AND URETHRA
513
FIG. 1. A, x-ray taken in initial stage of injection of contrast medium into accessory urethra via polythene tube. Note that accessory urethra is situated on dorsal part of penis. B, x-ray shows accessory bladder and urethra-former is completely outlined.
LH'T
U!?.&;'T(:llt.. -
-
R.161-!T
u~~-- -
FIG. 2. Schematic representation of the double bladder and urethra as shown in figure 1
Each urethra originates from a separate bladder and passes down through 1 penis. We have found 14 cases of complete duplication of the urethra and bladder in the literature. 5 - 11
FIG. 3. Operative specimen. Bladder and urethra
5 Bates, R. C.: A case of double vagina, uterus and bladder. Rhode Island Med. J ., 14: 192, 1931. 'Lanman, T. H. and Mahoney, P. J.: Intravenous urography in children. Amer. J. Dis. Child., 42: 611, 1931. 7 Nesbit, R. M. and Bromme, W.: Double penis and double bladder. Amer. J. Roentgen., 30: 497, 1933. 'Ravitch, M. M. and Scott, W.W.: Duplication of the entire colon, bladder and urethra. Surgery, 34: 843, 1953. 9 Satter, E. J. and Mossman, H. W.: A case report of a double bladder and double urethra in the female child. J. Urol., 79: 274, 1958. 10 Swenson, 0. and Oeconomopoulos, C. T.: Double lower genitourinary systems in a child. J. Urol., 85: 540, 1961. 11 Burns, E., Cummins, H. and Hyman, J.: Incomplete duplication of bladder with congenital solitary kidney: report of a case. J. Urol., 57: 257, 1947.
514
SINGH, MEHRA AND NAGABHUSHANAM
Of the 14 cases, 6 were in female subjects and 8 were in male subjects. In all cases there were associated anomalies of the genital, upper urinary or lower gastrointestinal systems alone or in various combinations. Six cases of bladder and urethral duplication were associated with genital tract abnormalities, 2 cases were associated with anomalies of the colon and anus and 6 were associated with both genital and colonic abnormalities. According to Burns and associates duplication of the bladder may occur by the sagittal septum or the coronal septum (frontal septum). 11 There is no mention of a separate ureteral opening into the accessory bladder in 3 of the 14 reported cases. However, in the remaining cases such an opening is reported. Our case is unusual in that there was complete duplication of the bladder and urethra with the accessory urethra originating from the accessory
bladder. The accessory bladder did not communicate with the normal bladder or with any ureter. According to Burns and associates in every case of duplication of the bladder, a ureter opens into the accessory bladder. 11 However, there was no such opening in our case. No case of this type has been described previously (except for the 3 incompletely documented cases mentioned previously). In all cases reported in the literature there have been associated congenital anomalies. SUMMARY
An unusual case of duplication of the bladder and urethra in a male subject without associated congenital anomalies is reported and a descriptive classification is suggested. The importance of methylene blue staining before excision is emphasized. X-ray findings and methods of dissection are also presented.
THE JOURNAL OF UROLOGY
Copyright © 1973 by The Williams & Wilkins Co.
COMPLETE DUPLICATION OF BLADDER AND URETHRA: A CASE REPORT WITH REVIEW OF THE LITERATURE J. P. SINGH, S. MEHRA
AND
V. NAGABHUSHANAM
From the Willingdon Hospital, New Delhi, India
through a racquet-shaped inc1s10n made in the mid-dorsal line of the proximal half of the penis. The urethra extended from the mid-dorsal shaft proximally and then dipped below and behind the symphysis pubis to become continuous with the accessory bladder. To expose the retropubic part of the accessory urethra, the vertical incision was extended proximally to join the suprapubic one. It was necessary to divide the pubic symphysis. The accessory bladder was continuous with and densely adherent to the normal bladder but the 2 cavities did not communicate with each other. The accessory bladder was 1 ½ inches in diameter and lobular in shape. While the bladders were being separated, the musculature of the normal bladder was shaved off at one site, thereby exposing the intact mucosa with the 2 bladders again separated anatomically. The gap in the bladder muscle was closed with catgut. There was no ureteral opening in the accessory bladder (figs. 2 and 3). Postoperatively the child experienced sepsis of the suprapubic wound for which he was given appropriate antibiotics. Histopathological examination of the accessory bladder and urethra revealed the specimen to be structurally compatible with normal bladder and urethra and to be lined with typical urothelium.
Duplication of the bladder and urethra is a rare anomaly. In complete duplication each urethra originates from a separate bladder; they pass down parallel to each other through 1 penis and have 2 external meatuses. Usually the anomalous urethral canal runs dorsal to the normal urethra, is narrow and its external meatus is situated somewhere between its normal location and the base of the penis. One of the 2 normal ureters or an accessory ureter may or may not open into the duplicated bladder. This anomaly may be associated with a duplicated genital system, colon or anus. To date 14 cases of complete duplication of the bladder and urethra have been described. In each of these cases there were other anomalies. CASE REPORT
A 6-month-old boy was admitted to the pediatric surgical service of this hospital on December 13, 1971 with intermittent purulent discharge from the dorsum of the penis occurring 3 times since birth. There were no associated urinary symptoms. Careful examination showed that the discharge came from a very small opening situated just beyond the middle of the dorsum of the penile shaft. Abdominal palpation (confirmed under general anesthesia) revealed a rounded, firm but nontender swelling 2 inches in diameter in the suprapubic region. Preoperative pus swab taken from the local site revealed Staphylococcus pyogenes sensitive to erythromycin, chloramphenicol and rovamycin. A probe could easily be passed into the sinus for 1½ to 2 inches inside the pelvis and well beyond the parietes. Contrast medium was injected into the opening via a polythene tube. After 10 cc of dye had been injected the abdominal swelling became more prominent. X-ray revealed a radiopaque irregular shadow in the suprapubic region in continuity with the accessory urethra (fig. 1).
DISCUSSION
Review of the literature on double bladder and urethra is confusing because many cases are not true cases of complete duplication and many reports are unclear. 1 After studying the literature we find that complete duplication of the urethra can be classified as follows: 1) Complete duplication of the urethra only-with or without associated congenital anomalies. Both urethras originate from 1 bladder through separate openings and pass down through 1 penis. 2 There are 40 cases of this type described in the literature. 2-• 2) Complete duplication of the urethra and bladder-with or without associated anomalies of the genital system, colon or anus.
TREATMENT
The child was given erythromycin for 3 days and underwent operation on December 22. Preoperatively, methylene blue was injected into the accessory urethra and bladder via a polythene tube. This stained the entire duplicated urethra and bladder. Via a suprapubic transverse incision the normal bladder as well as the overlying accessory bladder was exposed. The accessory bladder was adherent to the anterior surface of the normal bladder. The accessory urethra was dissected
1 Boissonnat, P.: Two cases of complete double functional urethra with a single bladder. Brit. J. Urol., 33: 453, 1961. 2 Casselman, J. and Williams, D. I.: Duplication of the urethra. Acta Urol. Belg., 34: 535, 1966. 3 Karanjavala, D. K.: An unusual case of complete reduplication of the urethra. Aust. New Zeal. J. Surg., 39: 284, 1970. •Tripathi, V. N. P. and Dick, V. S.: Complete duplication of male urethra. J. Urol., 101: 866, 1969.
Accepted for publication June 2, 1972. 512
COMPLETE DUPLICATION OF BLADDER AND URETHRA
513
FIG. 1. A, x-ray taken in initial stage of injection of contrast medium into accessory urethra via polythene tube. Note that accessory urethra is situated on dorsal part of penis. B, x-ray shows accessory bladder and urethra-former is completely outlined.
LH'T
U!?.&;'T(:llt.. -
-
R.161-!T
u~~-- -
FIG. 2. Schematic representation of the double bladder and urethra as shown in figure 1
Each urethra originates from a separate bladder and passes down through 1 penis. We have found 14 cases of complete duplication of the urethra and bladder in the literature. 5 - 11
FIG. 3. Operative specimen. Bladder and urethra
5 Bates, R. C.: A case of double vagina, uterus and bladder. Rhode Island Med. J ., 14: 192, 1931. 'Lanman, T. H. and Mahoney, P. J.: Intravenous urography in children. Amer. J. Dis. Child., 42: 611, 1931. 7 Nesbit, R. M. and Bromme, W.: Double penis and double bladder. Amer. J. Roentgen., 30: 497, 1933. 'Ravitch, M. M. and Scott, W.W.: Duplication of the entire colon, bladder and urethra. Surgery, 34: 843, 1953. 9 Satter, E. J. and Mossman, H. W.: A case report of a double bladder and double urethra in the female child. J. Urol., 79: 274, 1958. 10 Swenson, 0. and Oeconomopoulos, C. T.: Double lower genitourinary systems in a child. J. Urol., 85: 540, 1961. 11 Burns, E., Cummins, H. and Hyman, J.: Incomplete duplication of bladder with congenital solitary kidney: report of a case. J. Urol., 57: 257, 1947.
514
SINGH, MEHRA AND NAGABHUSHANAM
Of the 14 cases, 6 were in female subjects and 8 were in male subjects. In all cases there were associated anomalies of the genital, upper urinary or lower gastrointestinal systems alone or in various combinations. Six cases of bladder and urethral duplication were associated with genital tract abnormalities, 2 cases were associated with anomalies of the colon and anus and 6 were associated with both genital and colonic abnormalities. According to Burns and associates duplication of the bladder may occur by the sagittal septum or the coronal septum (frontal septum). 11 There is no mention of a separate ureteral opening into the accessory bladder in 3 of the 14 reported cases. However, in the remaining cases such an opening is reported. Our case is unusual in that there was complete duplication of the bladder and urethra with the accessory urethra originating from the accessory
bladder. The accessory bladder did not communicate with the normal bladder or with any ureter. According to Burns and associates in every case of duplication of the bladder, a ureter opens into the accessory bladder. 11 However, there was no such opening in our case. No case of this type has been described previously (except for the 3 incompletely documented cases mentioned previously). In all cases reported in the literature there have been associated congenital anomalies. SUMMARY
An unusual case of duplication of the bladder and urethra in a male subject without associated congenital anomalies is reported and a descriptive classification is suggested. The importance of methylene blue staining before excision is emphasized. X-ray findings and methods of dissection are also presented.