Endoscopy 9(1977) 110-112
0 Georg Thieme Verlag, Stuttgart
Endoscopic Management of Rare Congenital Malformations of the Male Urethra C. C. Schulman, F. D'Udekem Department of Urology, Brugman University Hospital, Univ. of Brussels
Partial duplication of the urethra and dilatation of Cowper's duct are very rare congenital anomalies of the lower urinary tract which can be diagnosed and located by endoscopy. Treatment consists in transurethral resection of the septum between the urethra and the accessory channel. Key-Words : Urethral malformation, urethral
duplication, Cowper's glands, operative endo-
Transurethrale Behandlung von angeborenen MiBbildungen der Urethra
Die partielle Duplikation der Urethra und die Dilatation der Cowperschen Drüse sind angeborene Anomalien, die sehr selten vorkommen und die durch Endoskopie lokalisiert und diagnostiziert werden können. Die Behandlung besteht in der transurethralen Resektion des Septums zwischen der Urethra und dem Nebenkanal.
scopy, TUR, pediatric urology.
Among the urological malformations, those of the urethra are not frequent and sometimes difficult to diagnose, especially when they cause none or few symptoms. The
two reported cases are examples of the eventuality since only a systematic urologic work-up with a complete radiological and endoscopical investigation has enabled to establish the diagnosis of partial duplication
of the urethra in the first case and of congenital dilation of Cowper's duct in the second case. Endoscopy was the clue to diagnosis and allowed at the same time the trans-
urethral treatment of theses rare abnormalities. Case Reports Case I: This child was seen at the age of 10 for
enuresis. His mother had undergone a renal homotransplantation one year before and worried about the integrity of her son's urinary tract. The intravenous urogram showed a very discrete alteration of the left pyelocaliceal system. The micturating urethrocystogram revealed a partial
urethral duplication of the bulbomembranous urethra (Fig. 1). The endoscopy precised thq radiological
findings:
the
urethral
channel
splitted into two portions in the bulbous area and reunited in the urethral segment under the veru montanum. The ventral channel seemed a little narrower. The bladder neck was normal and the bladder showed moderate trabeculations. A transurethral endoscopic resection of the fibrous
septum was performed so as to reunite the two channels in one larg-e lumen at that level. Shortly after this endoscopic operation enuresis disappeared.
Case 2: A 35-years-old male was admitted with
pulmonary atelectasis and renal insufficiency (urea 80 meo, creatinine 2.6 me/o/m1). The excretory urogram showed hydronephrosis of the right kidney with complete parenchymal atrophy requiring a right nephrectomy. The left kidney showed some focal pyelonephritic scarrings. The retrograde urethrogram demonstrated two diverticulum-like channels in the ventral wall of the proximal bulbous urethra which enlarged posteriorly coursing backward below and parallel to the urethra and ending blindly in the area of the
urogenital diaphragm (Fig. 2). Endoscopic examination of the urethra confirmed the presence of two orifices 2-3 cm in front of the veru montanum. The two channels seen on the urethrogram opened in the urethra through those orifices. The
fibrous septa between the channels were also sectioned in order to open those ducts completely
and bring them in free communication with the urethral lumen.
Downloaded by: University of British Columbia. Copyrighted material.
Summary
111
Downloaded by: University of British Columbia. Copyrighted material.
Endoscopic Management of Rare Congenital Malformations
Fig. 1
Micturating urethrocystogram: Partial duplication of the bulbomembranous urethra.
Fig. 2
Retrograde urethrogram: Demonstrates two diverticulumlike channels in the ventral wall of the proximal bulbous urethra ending blindly and corresponding to congenitally dilated Cowper's duct.
Conzments
thra not to be confused with a urethral
According to a recent review by Williams (1975), the urethral malformations observed in the first case may be classified as a sagit-
diverticulum which would have disrupted at
tal spindle-type duplication. This case is comparable with the one reported by Melchior (1975) as well as with the first case
presented in Fellows and Johnston's paper (1974). As there were two channels in the bulbous area reuniting at their proximal and distal end, our first case represented undoubtedly a limited duplication of the ure-
its distal end at the time of endoscopy or with a retention cyst of Cowper's gland ruptured in the urethra (Moscowitz 1976). This congenital malformation is actually considered as very uncommon (Williams 1975). Endoscopic resection of the septum
between the two channels provided the restituo ad integrum of the lower urinary tract and led to the disappearance of enuresis.
C. C. Schulman, F. D' dekcm: Endoscopic Management of Rare Congenital Malformations
Different operations have been proposed to
correct partial urethral duplication: complete excision of the accessory urethral channel, marsupalisation of both channels followed by urethroplasty, injection of sclerosant agents, transurethral endoscopic resection of the septum separating the bulbar
urethra and the accessory urethral canal (Fellow and Johnston 1975). We advocate this last method of treatment for its simplicity and the absence of complications.
The second rare urethral anomaly was a congenitally dilated Cowper's duct. The urethrogram showed a reflux in two dilated ducts ending blindly at the level of the urogenital diaphram where Cowper's glands are located (Currarino and Fuqua 1972). Endoscopy confirmed this anatomic location of the orifices of Cowper's ducts on the ventral wall of the bulbous urethra opening through
a paramedian orifice (Moscowitz
1976).
These ducts were most probably congenitally
dilated since no proximal urethral obstruction was observed. This rare anomaly has been reported as an incidental finding during routine cystourethrography (Currarino
and Fuqua 1972). Attention has recently been drawn to the existence of retention
cysts of Cowper's duct (Edling 1953, Moscowitz 1976, Ansell 1976, Cook and Shaw 1961).
Our urethrographic findings were characteristic for this type of lesion (Moscowitz 1976). Our case could thus have been the result of a spontaneous rupture of a retention cyst of Cowper's duct which communi-
cated with the bulbous urethra. However, the bilaterality of the anomaly was against an acquired lesion and in favour of a congenital malformation. Endoscopic diagnosis of very rare anom-
alies of the urethra and their transurethral treatment during the same examination is relevant even in children. References Ansell, J. S.: Cysts of the ducts of Cowpers glands. J. Urol. 115 (1976) 390
Cook, F. E. Jr., J. L. Show: Cystic anomalies of the ducts of Cowper's gland. J. Urol. 85 (1961) 659
Currarino, G., F. Fuqua: Cowper's glands in the urethrogram. Amer. J. Roentgenol. 116 (1972) 838
&Ring, N. P.: Radiologic appearances of diverticula of male cavernous urethra. Acta radio]. 40 (1953) 1
Fellows, G. J., J. H. Johnston: Incomplete urethral duplication and urinary retention. Brit. J. Urol. 46 (1974) 449 :Melchior, H.: Duplication of the urethra - Transurethral treatment. Europ. Urol. 1(1975) 243 Moscowitz, P. S., N. A. Newton, R. L. Lebowitz: Retention cysts of Cowper's duct. Radiology 120 (1976) 377
Williams, D. 1., M. M. Kenazei: Urethral duplications in the Male. Europ. Urol. 1(1975) 209
Dr. C. C. Schulman, Dept. of Urology, Brugman Univ.-Hospital, Briissel, Belgien
Downloaded by: University of British Columbia. Copyrighted material.
112
0 Georg Thieme Verlag, Stuttgart
Endoscopic Management of Rare Congenital Malformations of the Male Urethra C. C. Schulman, F. D'Udekem Department of Urology, Brugman University Hospital, Univ. of Brussels
Partial duplication of the urethra and dilatation of Cowper's duct are very rare congenital anomalies of the lower urinary tract which can be diagnosed and located by endoscopy. Treatment consists in transurethral resection of the septum between the urethra and the accessory channel. Key-Words : Urethral malformation, urethral
duplication, Cowper's glands, operative endo-
Transurethrale Behandlung von angeborenen MiBbildungen der Urethra
Die partielle Duplikation der Urethra und die Dilatation der Cowperschen Drüse sind angeborene Anomalien, die sehr selten vorkommen und die durch Endoskopie lokalisiert und diagnostiziert werden können. Die Behandlung besteht in der transurethralen Resektion des Septums zwischen der Urethra und dem Nebenkanal.
scopy, TUR, pediatric urology.
Among the urological malformations, those of the urethra are not frequent and sometimes difficult to diagnose, especially when they cause none or few symptoms. The
two reported cases are examples of the eventuality since only a systematic urologic work-up with a complete radiological and endoscopical investigation has enabled to establish the diagnosis of partial duplication
of the urethra in the first case and of congenital dilation of Cowper's duct in the second case. Endoscopy was the clue to diagnosis and allowed at the same time the trans-
urethral treatment of theses rare abnormalities. Case Reports Case I: This child was seen at the age of 10 for
enuresis. His mother had undergone a renal homotransplantation one year before and worried about the integrity of her son's urinary tract. The intravenous urogram showed a very discrete alteration of the left pyelocaliceal system. The micturating urethrocystogram revealed a partial
urethral duplication of the bulbomembranous urethra (Fig. 1). The endoscopy precised thq radiological
findings:
the
urethral
channel
splitted into two portions in the bulbous area and reunited in the urethral segment under the veru montanum. The ventral channel seemed a little narrower. The bladder neck was normal and the bladder showed moderate trabeculations. A transurethral endoscopic resection of the fibrous
septum was performed so as to reunite the two channels in one larg-e lumen at that level. Shortly after this endoscopic operation enuresis disappeared.
Case 2: A 35-years-old male was admitted with
pulmonary atelectasis and renal insufficiency (urea 80 meo, creatinine 2.6 me/o/m1). The excretory urogram showed hydronephrosis of the right kidney with complete parenchymal atrophy requiring a right nephrectomy. The left kidney showed some focal pyelonephritic scarrings. The retrograde urethrogram demonstrated two diverticulum-like channels in the ventral wall of the proximal bulbous urethra which enlarged posteriorly coursing backward below and parallel to the urethra and ending blindly in the area of the
urogenital diaphragm (Fig. 2). Endoscopic examination of the urethra confirmed the presence of two orifices 2-3 cm in front of the veru montanum. The two channels seen on the urethrogram opened in the urethra through those orifices. The
fibrous septa between the channels were also sectioned in order to open those ducts completely
and bring them in free communication with the urethral lumen.
Downloaded by: University of British Columbia. Copyrighted material.
Summary
111
Downloaded by: University of British Columbia. Copyrighted material.
Endoscopic Management of Rare Congenital Malformations
Fig. 1
Micturating urethrocystogram: Partial duplication of the bulbomembranous urethra.
Fig. 2
Retrograde urethrogram: Demonstrates two diverticulumlike channels in the ventral wall of the proximal bulbous urethra ending blindly and corresponding to congenitally dilated Cowper's duct.
Conzments
thra not to be confused with a urethral
According to a recent review by Williams (1975), the urethral malformations observed in the first case may be classified as a sagit-
diverticulum which would have disrupted at
tal spindle-type duplication. This case is comparable with the one reported by Melchior (1975) as well as with the first case
presented in Fellows and Johnston's paper (1974). As there were two channels in the bulbous area reuniting at their proximal and distal end, our first case represented undoubtedly a limited duplication of the ure-
its distal end at the time of endoscopy or with a retention cyst of Cowper's gland ruptured in the urethra (Moscowitz 1976). This congenital malformation is actually considered as very uncommon (Williams 1975). Endoscopic resection of the septum
between the two channels provided the restituo ad integrum of the lower urinary tract and led to the disappearance of enuresis.
C. C. Schulman, F. D' dekcm: Endoscopic Management of Rare Congenital Malformations
Different operations have been proposed to
correct partial urethral duplication: complete excision of the accessory urethral channel, marsupalisation of both channels followed by urethroplasty, injection of sclerosant agents, transurethral endoscopic resection of the septum separating the bulbar
urethra and the accessory urethral canal (Fellow and Johnston 1975). We advocate this last method of treatment for its simplicity and the absence of complications.
The second rare urethral anomaly was a congenitally dilated Cowper's duct. The urethrogram showed a reflux in two dilated ducts ending blindly at the level of the urogenital diaphram where Cowper's glands are located (Currarino and Fuqua 1972). Endoscopy confirmed this anatomic location of the orifices of Cowper's ducts on the ventral wall of the bulbous urethra opening through
a paramedian orifice (Moscowitz
1976).
These ducts were most probably congenitally
dilated since no proximal urethral obstruction was observed. This rare anomaly has been reported as an incidental finding during routine cystourethrography (Currarino
and Fuqua 1972). Attention has recently been drawn to the existence of retention
cysts of Cowper's duct (Edling 1953, Moscowitz 1976, Ansell 1976, Cook and Shaw 1961).
Our urethrographic findings were characteristic for this type of lesion (Moscowitz 1976). Our case could thus have been the result of a spontaneous rupture of a retention cyst of Cowper's duct which communi-
cated with the bulbous urethra. However, the bilaterality of the anomaly was against an acquired lesion and in favour of a congenital malformation. Endoscopic diagnosis of very rare anom-
alies of the urethra and their transurethral treatment during the same examination is relevant even in children. References Ansell, J. S.: Cysts of the ducts of Cowpers glands. J. Urol. 115 (1976) 390
Cook, F. E. Jr., J. L. Show: Cystic anomalies of the ducts of Cowper's gland. J. Urol. 85 (1961) 659
Currarino, G., F. Fuqua: Cowper's glands in the urethrogram. Amer. J. Roentgenol. 116 (1972) 838
&Ring, N. P.: Radiologic appearances of diverticula of male cavernous urethra. Acta radio]. 40 (1953) 1
Fellows, G. J., J. H. Johnston: Incomplete urethral duplication and urinary retention. Brit. J. Urol. 46 (1974) 449 :Melchior, H.: Duplication of the urethra - Transurethral treatment. Europ. Urol. 1(1975) 243 Moscowitz, P. S., N. A. Newton, R. L. Lebowitz: Retention cysts of Cowper's duct. Radiology 120 (1976) 377
Williams, D. 1., M. M. Kenazei: Urethral duplications in the Male. Europ. Urol. 1(1975) 209
Dr. C. C. Schulman, Dept. of Urology, Brugman Univ.-Hospital, Briissel, Belgien
Downloaded by: University of British Columbia. Copyrighted material.
112
