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Two Cases of Subsymphyseal Epispadias in the Female Patient with Surgical Correction
Vol. 97, Mar.
THE JOURNAL OF UROLOGY
Copyright © 1967 by The Williams & Wilkins Co.
Printed in U.S.A.
TWO CASES OF SUBSYMPHYSEAL EPISPADIAS IN THE FEMALE PATIENT WITH SURGICAL CORRECTION HENRY K. FREEDMAN, H. LEONARD SCHLESINGER
Subsymphyseal epispadias in a female patient is a congenital deformity which is a variation of exstrophy of the bladder. The defect is rare and it is unusual to come across 2 cases within a year. Specific mention of this anomaly with a bibliography of previous cases is contained in Marshall and Muecke's article on exstrophy of the bladder. 1 In our patients the defect consisted of a widened and depressed symphysis due to separation of the pubic bones, non-union of the labia at the
AND
GEORGE E. RANDALL
Force Base Hospital in August 1963. She gave a history of stress incontinence since birth and occasional enuresis. When the girl was 5 years old she had had a transurethral operation but no improvement was noticed. At that time, her parents had been told that her bladder muscle had formed incompletely. Our examination showed non-union of the labia at the anterior commissure and a 2-inch depression over the pubic symphysis. The remainder of her physical examination was normal.
FIG.1
anterior commissure and an intact though foreshortened urethra. In each case the presenting symptom was incontinence and surgical cure was obtained by a Marshall-Marchetti vesicourethral suspension. We know of no other cases in which a cure was obtained by any method of treatment. CASE REPORTS
Case 1. A 15-year-old Caucasian high-school student was first seen at the Plattsburgh Air
Accepted for publication May 27, 1966. 1 Marshall, V. F. and Muecke, E. C.: Variations in exstrophy of the bladder. J. Urol., 88: 766-796
1962.
'
490
A survey film of the abdomen demonstrated a 2-inch separation of the symphysis (fig. 1, A). An excretory urogram was normal. A cystogram showed an irregular lower border of the bladder extending below the superior pubic rami (fig. 1, B). On cystoscopy the bladder and vesical neck appeared normal. The urethra was shorter than usual. In September 1963, a Marshall-Marchetti vesicourethral suspension was performed. The sutures lateral to the urethra were anchored to a fibrous rubbery band of tissue which connected the pubic bones. At the operation the peritoneum was opened and normal pelvic organs were seen.
SURGICAL CORRECTION OF SUBSYMPHYSEAL EPISPADIAS
Fm. 2 Postoperatively, and when seen 6 months later, the patient was completely asymptomatic. Case 2. A 10-year-old white schoolgirl was first seen by us in October 1963. In 1957 the patient had been operated upon because of total incontinence. A diagnosis of female epispadias had been made. The operative procedure consisted of cutting a wedge from the urethra and re-approximating the urethra over a number 18 sound in 2 layers. The patient's urinary incontinence, though initially improved, soon reverted to total incontinence. When we first saw the patient, urine dribbled constantly. She rarely voided with an actual stream but noticed that coughing, sneezing or running increased her incontinence. On physical examination the scar from the patient's previous operation was seen running from the umbilicus downward (fig. 2). The
491
symphysis had a rubbery feeling on pressure, and over it a depression was noted which blended below with the unfused labia. The urethra was seen just below the split labia minora. The vagina appeared as a tube with a marked depression of the tissues around the introitus. Urine leaked freely from the urethra. An x-ray film prior to an excretory urogram showed a 2}1-inch separation of the pubic bones. The kidneys were normal and the bladder quite obviously contained urine and acted as an imperfect reservoir. A cystogram was normal. On cystoscopic examination, the urethra was foreshortened. The bladder and vesical neck appeared normal. Angulation of the vesical neck by upward prrnsure within the vagina caused continence. A Marshall-Marchetti vesicourethral suspension was done with a considerable amount of difficulty because of the previous scarring. Once again the lateral urethral sutures were tied to the rubbery fibrous tissue connecting the pubic bones. Postoperatively, and when seen 8 months later the patient was continent of urine. However, sh~ had noticed that if she had a full bladder and did not empty it, she would lose small amounts of urine on coughing and sneezing. SUMMARY
Two cases of subsymphyseal epispadias are presented. Surgical cure was obtained by doing a Marshall-Marchetti vesicourethral suspension. To our knowledge this represents the first surgical correction of an unusual anomaly.
46 Cornelia St., Plattsburgh, New York 12901
THE JOURNAL OF UROLOGY
Copyright © 1967 by The Williams & Wilkins Co.
Printed in U.S.A.
TWO CASES OF SUBSYMPHYSEAL EPISPADIAS IN THE FEMALE PATIENT WITH SURGICAL CORRECTION HENRY K. FREEDMAN, H. LEONARD SCHLESINGER
Subsymphyseal epispadias in a female patient is a congenital deformity which is a variation of exstrophy of the bladder. The defect is rare and it is unusual to come across 2 cases within a year. Specific mention of this anomaly with a bibliography of previous cases is contained in Marshall and Muecke's article on exstrophy of the bladder. 1 In our patients the defect consisted of a widened and depressed symphysis due to separation of the pubic bones, non-union of the labia at the
AND
GEORGE E. RANDALL
Force Base Hospital in August 1963. She gave a history of stress incontinence since birth and occasional enuresis. When the girl was 5 years old she had had a transurethral operation but no improvement was noticed. At that time, her parents had been told that her bladder muscle had formed incompletely. Our examination showed non-union of the labia at the anterior commissure and a 2-inch depression over the pubic symphysis. The remainder of her physical examination was normal.
FIG.1
anterior commissure and an intact though foreshortened urethra. In each case the presenting symptom was incontinence and surgical cure was obtained by a Marshall-Marchetti vesicourethral suspension. We know of no other cases in which a cure was obtained by any method of treatment. CASE REPORTS
Case 1. A 15-year-old Caucasian high-school student was first seen at the Plattsburgh Air
Accepted for publication May 27, 1966. 1 Marshall, V. F. and Muecke, E. C.: Variations in exstrophy of the bladder. J. Urol., 88: 766-796
1962.
'
490
A survey film of the abdomen demonstrated a 2-inch separation of the symphysis (fig. 1, A). An excretory urogram was normal. A cystogram showed an irregular lower border of the bladder extending below the superior pubic rami (fig. 1, B). On cystoscopy the bladder and vesical neck appeared normal. The urethra was shorter than usual. In September 1963, a Marshall-Marchetti vesicourethral suspension was performed. The sutures lateral to the urethra were anchored to a fibrous rubbery band of tissue which connected the pubic bones. At the operation the peritoneum was opened and normal pelvic organs were seen.
SURGICAL CORRECTION OF SUBSYMPHYSEAL EPISPADIAS
Fm. 2 Postoperatively, and when seen 6 months later, the patient was completely asymptomatic. Case 2. A 10-year-old white schoolgirl was first seen by us in October 1963. In 1957 the patient had been operated upon because of total incontinence. A diagnosis of female epispadias had been made. The operative procedure consisted of cutting a wedge from the urethra and re-approximating the urethra over a number 18 sound in 2 layers. The patient's urinary incontinence, though initially improved, soon reverted to total incontinence. When we first saw the patient, urine dribbled constantly. She rarely voided with an actual stream but noticed that coughing, sneezing or running increased her incontinence. On physical examination the scar from the patient's previous operation was seen running from the umbilicus downward (fig. 2). The
491
symphysis had a rubbery feeling on pressure, and over it a depression was noted which blended below with the unfused labia. The urethra was seen just below the split labia minora. The vagina appeared as a tube with a marked depression of the tissues around the introitus. Urine leaked freely from the urethra. An x-ray film prior to an excretory urogram showed a 2}1-inch separation of the pubic bones. The kidneys were normal and the bladder quite obviously contained urine and acted as an imperfect reservoir. A cystogram was normal. On cystoscopic examination, the urethra was foreshortened. The bladder and vesical neck appeared normal. Angulation of the vesical neck by upward prrnsure within the vagina caused continence. A Marshall-Marchetti vesicourethral suspension was done with a considerable amount of difficulty because of the previous scarring. Once again the lateral urethral sutures were tied to the rubbery fibrous tissue connecting the pubic bones. Postoperatively, and when seen 8 months later the patient was continent of urine. However, sh~ had noticed that if she had a full bladder and did not empty it, she would lose small amounts of urine on coughing and sneezing. SUMMARY
Two cases of subsymphyseal epispadias are presented. Surgical cure was obtained by doing a Marshall-Marchetti vesicourethral suspension. To our knowledge this represents the first surgical correction of an unusual anomaly.
46 Cornelia St., Plattsburgh, New York 12901