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Congenital diverticulum of male urethra
CONGENITAL DIVERTICULUM OF MALE URETHRA NELSON
RODRIGUES
NETTO,
GUSTAV0
CASERTA LEMOS,
JOAQUIM
FRANCISCO
FLAVIO
DE
JR., M.D.
M.D.
ALMEIDA
LUIZ ORTIZ HERING,
CLARO,
M.D.
M.D.
From the Division of Urology, University of Campinas Medical School-UNICAMF’, and Urology Department of the Hospital Beneficencia Portuguesa of Sao Paulo, Brazil
ABSTRACT-Congenital diverticulum of the male urethra is an uncommon condition. Urinary tract infection, urethral obstruction, and disturbances in micturition are the most common symptoms. Six cases of congenital diverticulum of the male urethra were studied with regard to diagnosis and treatment. Four patients underwent endoscopic treatment of the diverticulum. The procedure is simple and rapid, with low incidence of complications and high rates of success.
Congenital diverticulum of the male urethra is a relatively rare condition, frequently difficult to differentiate from acquired diverticula.’ It often causes urinary tract infection, urethral obstruction, and disturbances in micturition. Diagnosis is made through urethrography and urethroscopic examination2 Surgical treatment is performed through diverticulectomy or by opening the neck of the diverticulum endoscopically. The endoscopic technique proved to be efficient due to its facility, low incidence of morbidity, and high rates of success. 3 In the present study, we report on 6 cases of congenital diverticulum of the male urethra, commenting on diagnosis and treatment. The symptoms occurring in these patients are listed in Table I. Case Reports Case 1 A twenty-three-year-old white man suffering from dysuria for two years was referred to our clinic. He showed no mictional disorders nor urethral secretion, nor was there a history of urethritis, urethral surgery, or genital injury.
UROLOGY
i SEPTEMBER1984
/ VOLUMEXXIV,NUMBER3
Findings on clinical examination were normal. Urinalysis and urine culture as well as complete blood count, and urea and creatinine levels were within normal limits. Voiding cystourethrography revealed a diverticulum in the bulbar urethra. Urethrocystoscopy confirmed this finding, showing the ostium of the diverticulum at the ventral portion of the bulbar urethra. The diverticulum was excised via a perineal approach, and a urethral tube was kept in place for seven days postoperatively. Convalescence was uneventful, with no complications during follow-up.
TABLEI.
Symptoms Dysuria Posturinary dribbling
Postmicturitional scrotal tumefaction Spermatorrhea Frequency Hematuria
Symptomatology No. of Patients 3
Per Cent
4
50 66
1 1 1 1
16 16 16 16
239
FIGURE 1. Case 2. Voiding cystourethrogram shows diverticulum (A)and diagrammatic representation (B) of findings at urethroscopy.
Case 3
Case 2
A twenty-four-year-old white patient was hospitalized with the chief complaints of posturinary dribbling and spermatorrhea. There was no history of previous urethritis or genital injury. No abnormality was detected on general physical and local examination. Urinalysis and culture as well as complete blood count were normal. Voiding cystourethrography demonstrated a paraurethral saccular formation in the bulbar region (Fig. 1). Urethrocystoscopy confirmed the diagnosis, revealing a small orifice of about 3 mm on the floor of the bulbar urethra. External digital expression of the bulbar region resulted in the outflow of a yellowish white liquid through the ostium of the diverticulum. The patient underwent endoscopic treatment, which consisted of transurethral unroofing using the Sachse knife. Postoperatively, a urethral catheter was used for three days. Six years after surgery he is free of symptoms.
240
A thirty-seven-year-old white patient was seen, with a fifteen-year history of dysuria and postmicturitional tumefaction of the penoscrotal region. Clinical examination revealed a painless tumorous formation of soft consistency in the penoscrotal junction, with no evidence of inflammation. The mass disappeared when resulting in outflow of urine compressed, through the urethral meatus. Urinalysis revealed leukocyturia, and culture showed more than 100,000 colonies of Escherichia coli. A retrograde urethrogram and voiding cystourethrogram revealed a diverticulum situated in the bulbar urethra and a reflux of the contrast material toward the seminal vesicles. Urethrocystoscopy confirmed the diagnosis of diverticulum of the bulbar urethra, which was unroofed transurethrally with the Sachse knife. Postoperatively, a urethral catheter was kept in place for four days. A follow-up eight months after surgery showed no mictional abnormalities nor swelling in the perineum, and urinalysis was normal. Case 4
A fifty-five-year-old white patient was hospitalized, with a complaint of dysuria and voiding frequency for ten years, which had worsened over the last month. There was no history of injury or urethritis. Clinical examination revealed no abnormalities. Urinalysis and urine culture showed urinary tract infection caused by Staphylococcus aureus. Urethrocystography showed a diverticulum of the bulbar urethra, and urethrocystoscopy revealed an ostium of the diverticulum at the ventral portion of the
UROLOGY
/ SEPTEMBER1984
i VOLUMEXXIV,
NUMBER3
\ 0\ / /
FIGURE 2. Diverticulum (A) sac&at type and (B) tubular type.
8
d ; bulbar urethra. An endoscopic opening of the neck of the diverticulum was performed, and a catheter was kept in place for four days postoperatively, Eighteen months later micturition and urinalysis are normal, with only moderate burning of the urethra. Case 5 A seventeen-year-old white patient was seen with a complaint of posturinary dribbling from infancy, which had worsened greatly over the last three months. There was no history of urethritis nor injury. Clinical examination including urinalysis, urine culture, and complete blood count was normal. Urethrocystography revealed a diverticulum of the penile urethra. The patient was advised to undergo surgical treatment with an endoscopic approach. However, he did not agree to this and did not return for further clinical follow-up. Case 6 An eighteen-year-old white man was seen with the chief complaints of posturinary dribbling, terminal hematuria, and fever (38” C). He had no previous urethritis or genital trauma. A voiding cystourethrogram and urethroscopy demonstrated a paraurethral saccular formation approximately 3 by 2 cm, communicating with the lumen of the bulbar urethra by a small orifice of about 4 mm. The patient underwent internal urethrotomy under direct vision with the Sachse knife. Convalescence was uneventful, and he is free of symptoms twenty months after surgery. Comment Diverticulum of the urethra is a saccular or tubular dilation of the urethral wall that communicates with the lumen by means of a neck. Urethral diverticulum is not a common condi-
UROLOGY
/
SEPTEMBER
1984
/ VOLUME
tion and may be congenital or acquired. Congenital urethral diverticulum occurs in approximately 1 to 10 when compared with acquired diverticulum, affecting the penile and bulbar urethra.2 Various theories attempt to explain the formation of congenital urethral diverticula. The first hypothesis is that it would be derived from the partial lack of spongy body, representing incomplete hypospadias4 Another hypothesis would be the association with valves of the anterior urethra, appearing secondarily to these, which are, in turn a consequence of the absence of linkage between urethral segments5 The third theory is that congenital urethral diverticula would arise from the spontaneous rupture of the paraurethral cysts toward the lumen of the urethra. These cysts are secondary to the obstruction of the abnormal paraurethral gland? or of ectopic epithelial cell nests.7 Congenital urethral diverticula may be either saccular or tubular (Fig. 2). The saccular type has a true neck and may cause urinary obstruction since its posterior segment compresses the urethra and interrupts the urinary flow when the saccular cavity fills at the beginning of micturition (Fig. 1).3 The tubular or diffuse type is located more proximally to the urethral bulb, where urinary stasis occurs along with its complications, such as urinary stasis and calculosis. This type, which can also be called megaurethra or urethral ectasia,$ is less frequent than the saccular type and may be associated with the prune-belly syndrome.g The symptoms depend largely on the size of the diverticulum. The smaller ones often show no symptoms, becoming evident only when other problems such as urethritis occur. The larger diverticula tend to retain urine, harboring infection and leading to difficulties in micturition and dysuria. When located in the posterior urethra, it may resemble prostatitis 2.3.10
XXIV. NUMBER 3
241
FIGURE 3. Case 2. Postoperative voiding tourethrogram shows ample communication tween diverticulum and urethral lumen.
cysbe-
Depending on the position of the diverticulum, it can be palpated when filled with urine (Case 3); postmictional dribbling is common (Case 5) and ejaculatory disturbances may be noted (Case 2). Hematuria has been described as a common symptom although it was not found in our cases.’ Diagnosis of a urethral diverticulum is often clinical. In the clinical examination, the mass is palpated in the bulbar region; it disappears when compressed and is followed by outflow of urine or infected material through the urethra. Confirmation of the diagnosis is made through retrograde and mictional urethrocystography and urethroscopy. Often it is useful to perform external manual palpation of the diverticulum to locate its orifice, which is sometimes very small. 3~10Treatment of a urethral diverticulum may be performed through open surgery, with a perineal approach, excising the diverticulum and suturing the urethra (Case 1). Another type of treatment is via an endoscopic approach using the Sachse knife. The ori-
242
fice of the diverticulum is incised, and ample communication is obtained between the diverticulum and urethral lumen (Cases 2, 3, and 4) (Fig. 3).3 Endoscopic treatment of the urethral diverticulum is advantageous when compared with diverticulectomy since it is a quicker procedure, involving less surgical injury and incidence of complications, and a shorter hospital stay.3 From the results obtained in our study, we believe the endoscopic technique to be the procedure of choice for therapy of congenital diverticulum of the male urethra, the open approach being reserved for those patients in whom the diverticula are very large or in whom endoscopic treatment has failed. UNICAMP Sio Paulo, Brazil (DR. NETTO) References 1. Sweetser TH Tr:Congenital urethral diverticula of the male patient. J Urol 97:-93 (1967). 2. Mohan V, et al: Urethral diverticulum in male subjects: report of 5 cases, ibid 123: 592 (1980). 3. Ortilip SA, Gonzalez R, and Williams RD: Diverticula of the male urethra. ibid 124: 350 (1980). 4. Voillemier LD‘ Trait& des‘ Maladies des Voies Urinaires, Paris, V. Mason and FIls, 1868. 5. Boissonnat P, and Duhamel B: Congenital diverticulum of the anterior urethra associated with aplasia of the abdominal muscles in the male infant, Br J Ural 34: 59 (1962). 6. Johnson FP: The latter development of the urethra in the male, 1 Urol 4: 447 (1920). 7. Siiter F: Ein Beitrag Zur Histologie und genese der congenitalen Divertikel der mannlichen Harnrohre. Arch Klin Chir 87: 225 (1908). 8. Dorairajan T: Defects of spongy tissue and congenital diverticula of the senile urethra. Aust NZ 1 Sure 32: 209 (1963). 9. Kirkis DR, and Grossman H: CbngeGital saccular anterior urethral diverticulum, Radiology 140: 367 (1981). 10. Nickel WR, and Plumb RT: Other infections and inflammation of the external genitalia, in Harrison JH, et a2 (Eds): Campbell’s Urology, Philadelphia, W. B. Saunders, 1978, pp 678680.
UROLOGY
/
SEPTEMBER
1984
/ VOLUME XXIV, NUMBER 3
RODRIGUES
NETTO,
GUSTAV0
CASERTA LEMOS,
JOAQUIM
FRANCISCO
FLAVIO
DE
JR., M.D.
M.D.
ALMEIDA
LUIZ ORTIZ HERING,
CLARO,
M.D.
M.D.
From the Division of Urology, University of Campinas Medical School-UNICAMF’, and Urology Department of the Hospital Beneficencia Portuguesa of Sao Paulo, Brazil
ABSTRACT-Congenital diverticulum of the male urethra is an uncommon condition. Urinary tract infection, urethral obstruction, and disturbances in micturition are the most common symptoms. Six cases of congenital diverticulum of the male urethra were studied with regard to diagnosis and treatment. Four patients underwent endoscopic treatment of the diverticulum. The procedure is simple and rapid, with low incidence of complications and high rates of success.
Congenital diverticulum of the male urethra is a relatively rare condition, frequently difficult to differentiate from acquired diverticula.’ It often causes urinary tract infection, urethral obstruction, and disturbances in micturition. Diagnosis is made through urethrography and urethroscopic examination2 Surgical treatment is performed through diverticulectomy or by opening the neck of the diverticulum endoscopically. The endoscopic technique proved to be efficient due to its facility, low incidence of morbidity, and high rates of success. 3 In the present study, we report on 6 cases of congenital diverticulum of the male urethra, commenting on diagnosis and treatment. The symptoms occurring in these patients are listed in Table I. Case Reports Case 1 A twenty-three-year-old white man suffering from dysuria for two years was referred to our clinic. He showed no mictional disorders nor urethral secretion, nor was there a history of urethritis, urethral surgery, or genital injury.
UROLOGY
i SEPTEMBER1984
/ VOLUMEXXIV,NUMBER3
Findings on clinical examination were normal. Urinalysis and urine culture as well as complete blood count, and urea and creatinine levels were within normal limits. Voiding cystourethrography revealed a diverticulum in the bulbar urethra. Urethrocystoscopy confirmed this finding, showing the ostium of the diverticulum at the ventral portion of the bulbar urethra. The diverticulum was excised via a perineal approach, and a urethral tube was kept in place for seven days postoperatively. Convalescence was uneventful, with no complications during follow-up.
TABLEI.
Symptoms Dysuria Posturinary dribbling
Postmicturitional scrotal tumefaction Spermatorrhea Frequency Hematuria
Symptomatology No. of Patients 3
Per Cent
4
50 66
1 1 1 1
16 16 16 16
239
FIGURE 1. Case 2. Voiding cystourethrogram shows diverticulum (A)and diagrammatic representation (B) of findings at urethroscopy.
Case 3
Case 2
A twenty-four-year-old white patient was hospitalized with the chief complaints of posturinary dribbling and spermatorrhea. There was no history of previous urethritis or genital injury. No abnormality was detected on general physical and local examination. Urinalysis and culture as well as complete blood count were normal. Voiding cystourethrography demonstrated a paraurethral saccular formation in the bulbar region (Fig. 1). Urethrocystoscopy confirmed the diagnosis, revealing a small orifice of about 3 mm on the floor of the bulbar urethra. External digital expression of the bulbar region resulted in the outflow of a yellowish white liquid through the ostium of the diverticulum. The patient underwent endoscopic treatment, which consisted of transurethral unroofing using the Sachse knife. Postoperatively, a urethral catheter was used for three days. Six years after surgery he is free of symptoms.
240
A thirty-seven-year-old white patient was seen, with a fifteen-year history of dysuria and postmicturitional tumefaction of the penoscrotal region. Clinical examination revealed a painless tumorous formation of soft consistency in the penoscrotal junction, with no evidence of inflammation. The mass disappeared when resulting in outflow of urine compressed, through the urethral meatus. Urinalysis revealed leukocyturia, and culture showed more than 100,000 colonies of Escherichia coli. A retrograde urethrogram and voiding cystourethrogram revealed a diverticulum situated in the bulbar urethra and a reflux of the contrast material toward the seminal vesicles. Urethrocystoscopy confirmed the diagnosis of diverticulum of the bulbar urethra, which was unroofed transurethrally with the Sachse knife. Postoperatively, a urethral catheter was kept in place for four days. A follow-up eight months after surgery showed no mictional abnormalities nor swelling in the perineum, and urinalysis was normal. Case 4
A fifty-five-year-old white patient was hospitalized, with a complaint of dysuria and voiding frequency for ten years, which had worsened over the last month. There was no history of injury or urethritis. Clinical examination revealed no abnormalities. Urinalysis and urine culture showed urinary tract infection caused by Staphylococcus aureus. Urethrocystography showed a diverticulum of the bulbar urethra, and urethrocystoscopy revealed an ostium of the diverticulum at the ventral portion of the
UROLOGY
/ SEPTEMBER1984
i VOLUMEXXIV,
NUMBER3
\ 0\ / /
FIGURE 2. Diverticulum (A) sac&at type and (B) tubular type.
8
d ; bulbar urethra. An endoscopic opening of the neck of the diverticulum was performed, and a catheter was kept in place for four days postoperatively, Eighteen months later micturition and urinalysis are normal, with only moderate burning of the urethra. Case 5 A seventeen-year-old white patient was seen with a complaint of posturinary dribbling from infancy, which had worsened greatly over the last three months. There was no history of urethritis nor injury. Clinical examination including urinalysis, urine culture, and complete blood count was normal. Urethrocystography revealed a diverticulum of the penile urethra. The patient was advised to undergo surgical treatment with an endoscopic approach. However, he did not agree to this and did not return for further clinical follow-up. Case 6 An eighteen-year-old white man was seen with the chief complaints of posturinary dribbling, terminal hematuria, and fever (38” C). He had no previous urethritis or genital trauma. A voiding cystourethrogram and urethroscopy demonstrated a paraurethral saccular formation approximately 3 by 2 cm, communicating with the lumen of the bulbar urethra by a small orifice of about 4 mm. The patient underwent internal urethrotomy under direct vision with the Sachse knife. Convalescence was uneventful, and he is free of symptoms twenty months after surgery. Comment Diverticulum of the urethra is a saccular or tubular dilation of the urethral wall that communicates with the lumen by means of a neck. Urethral diverticulum is not a common condi-
UROLOGY
/
SEPTEMBER
1984
/ VOLUME
tion and may be congenital or acquired. Congenital urethral diverticulum occurs in approximately 1 to 10 when compared with acquired diverticulum, affecting the penile and bulbar urethra.2 Various theories attempt to explain the formation of congenital urethral diverticula. The first hypothesis is that it would be derived from the partial lack of spongy body, representing incomplete hypospadias4 Another hypothesis would be the association with valves of the anterior urethra, appearing secondarily to these, which are, in turn a consequence of the absence of linkage between urethral segments5 The third theory is that congenital urethral diverticula would arise from the spontaneous rupture of the paraurethral cysts toward the lumen of the urethra. These cysts are secondary to the obstruction of the abnormal paraurethral gland? or of ectopic epithelial cell nests.7 Congenital urethral diverticula may be either saccular or tubular (Fig. 2). The saccular type has a true neck and may cause urinary obstruction since its posterior segment compresses the urethra and interrupts the urinary flow when the saccular cavity fills at the beginning of micturition (Fig. 1).3 The tubular or diffuse type is located more proximally to the urethral bulb, where urinary stasis occurs along with its complications, such as urinary stasis and calculosis. This type, which can also be called megaurethra or urethral ectasia,$ is less frequent than the saccular type and may be associated with the prune-belly syndrome.g The symptoms depend largely on the size of the diverticulum. The smaller ones often show no symptoms, becoming evident only when other problems such as urethritis occur. The larger diverticula tend to retain urine, harboring infection and leading to difficulties in micturition and dysuria. When located in the posterior urethra, it may resemble prostatitis 2.3.10
XXIV. NUMBER 3
241
FIGURE 3. Case 2. Postoperative voiding tourethrogram shows ample communication tween diverticulum and urethral lumen.
cysbe-
Depending on the position of the diverticulum, it can be palpated when filled with urine (Case 3); postmictional dribbling is common (Case 5) and ejaculatory disturbances may be noted (Case 2). Hematuria has been described as a common symptom although it was not found in our cases.’ Diagnosis of a urethral diverticulum is often clinical. In the clinical examination, the mass is palpated in the bulbar region; it disappears when compressed and is followed by outflow of urine or infected material through the urethra. Confirmation of the diagnosis is made through retrograde and mictional urethrocystography and urethroscopy. Often it is useful to perform external manual palpation of the diverticulum to locate its orifice, which is sometimes very small. 3~10Treatment of a urethral diverticulum may be performed through open surgery, with a perineal approach, excising the diverticulum and suturing the urethra (Case 1). Another type of treatment is via an endoscopic approach using the Sachse knife. The ori-
242
fice of the diverticulum is incised, and ample communication is obtained between the diverticulum and urethral lumen (Cases 2, 3, and 4) (Fig. 3).3 Endoscopic treatment of the urethral diverticulum is advantageous when compared with diverticulectomy since it is a quicker procedure, involving less surgical injury and incidence of complications, and a shorter hospital stay.3 From the results obtained in our study, we believe the endoscopic technique to be the procedure of choice for therapy of congenital diverticulum of the male urethra, the open approach being reserved for those patients in whom the diverticula are very large or in whom endoscopic treatment has failed. UNICAMP Sio Paulo, Brazil (DR. NETTO) References 1. Sweetser TH Tr:Congenital urethral diverticula of the male patient. J Urol 97:-93 (1967). 2. Mohan V, et al: Urethral diverticulum in male subjects: report of 5 cases, ibid 123: 592 (1980). 3. Ortilip SA, Gonzalez R, and Williams RD: Diverticula of the male urethra. ibid 124: 350 (1980). 4. Voillemier LD‘ Trait& des‘ Maladies des Voies Urinaires, Paris, V. Mason and FIls, 1868. 5. Boissonnat P, and Duhamel B: Congenital diverticulum of the anterior urethra associated with aplasia of the abdominal muscles in the male infant, Br J Ural 34: 59 (1962). 6. Johnson FP: The latter development of the urethra in the male, 1 Urol 4: 447 (1920). 7. Siiter F: Ein Beitrag Zur Histologie und genese der congenitalen Divertikel der mannlichen Harnrohre. Arch Klin Chir 87: 225 (1908). 8. Dorairajan T: Defects of spongy tissue and congenital diverticula of the senile urethra. Aust NZ 1 Sure 32: 209 (1963). 9. Kirkis DR, and Grossman H: CbngeGital saccular anterior urethral diverticulum, Radiology 140: 367 (1981). 10. Nickel WR, and Plumb RT: Other infections and inflammation of the external genitalia, in Harrison JH, et a2 (Eds): Campbell’s Urology, Philadelphia, W. B. Saunders, 1978, pp 678680.
UROLOGY
/
SEPTEMBER
1984
/ VOLUME XXIV, NUMBER 3