Vol. 93, Feb.
THE JOURNAL OF UROLOGY
Copyright © 1965 by The Williams & Wilkins Co.
Printed in U.S.A.
CONGENITAL RECTOURETHRAL FISTULA: REPORT OF CASE WITHOUT RECTAL ANOMALY ECTOR LEDUC While rectourethral fistula is uncommon, it is seen frequently with such anomalies as atresia of the anus or rectum. All of the congenital varieties reported in the literature are associated with anomalies of development of the rectum, and Browne states unequivocally that rectourethral fistula is never seen in association with a properly developed rectum. The literature as quoted by Stewart and associates indicates that about 1: 5000 births result in some type of anorectal deformity, 70 per cent with fistulas, and Campbell gives the figure of 1: 1560 with such deformities at autopsy. All modern textbooks report fairly large series of this disorder, all in connection with rectal anomalies. EMBRYOLOGY
It is generally agreed that the cause of rectourinary fistula is due to a failure of the urorectal septum completely to divide the cloaca into an anterior urethrovesical segment and a posterior rectal segment. The resultant fistula depends on which portion of the saddle-like partition is defective. SYMPTOMS
There is some variation in symptomatology, according to the location and size of the fistula. There is always urinary infection, most often with the history of passage of gas and fecal material in the urine. In infancy, meconium in the urine may be noted in the first few days of life. In the majority of cases not repaired when the rectal anomaly is dealt with, there is passage of urine into the rectum. The patient with the longest duration of symptoms (45 years) hitherto reported was described in the Journal of Urology by Atassi and McEver.
measures taken to determine whether a concomitant rectourinary fistula is present. This should include careful examination of the urine for evidence of meconium, cystogram and urethogram prior to operative treatment of the rectal deformity. In older persons, a history of passage of gas and/or feces per urethram requires definitive diagnostic study as does passage of urine per rectum. Sometimes the discovery of the urinary orifice is difficult; at other times, as in J\!Iagee's case, one can pass the cystoscope into the opening and out the anus. One should certainly have an excretory urogram performed, as many cases have anomalies of the upper urinary tract. Careful rectal palpation for evidence of a dimple or depression at the site of the orifice may be of diagnostic import. TREATMENT
Most authors have recommended perinea! section and closure of the separate orifices into the bowel and the urethra. Weyrauch's technique of perineal dissection and closure so as to separate the two openings is applicable to the acquired type and certainly would be useful in the congenital variety. Campbell and Laird both favor the Young-Stone operation, but Campbell warns of the danger of later anal stricture. Many use suprapubic cystostomy to divert the urine, and some also use colostomy to divert the fecal stream. The size and location of the openings might demand diversion of both urinary and fecal current to ensure the maximum chance for healing, but the smaller caliber fistulas may be dealt with by simple perinea! dissection and closure over urethral catheter, using appropriate antibiotics for control of infection. CASE REPOR'r
DIAGNOSIS
The diagnosis must be suspected until proved absent in every case of imperforate anus, and Accepted for publication .Tune 18, 1964. Read at annual meeting of Western Section, American Urological Association, Inc., Coronado, California, February 17-20, 1964. 272
A 59-year-old white man was seen on August 19, 1963. Since the age of 3 years he had had recurrent attacks of severe urinary tract infection. These consisted of severe pain and burning on urination associated with pyuria. About twenty cystoscopic examinations had been done by different urologists in various parts of the
CONGENITAL RECTOURETHRAL FISTULA
country. In 1945 he was examined at the Mayo Clinic and advised to have a transurethral prostatectomy. In May 1953 he began to pass gas through the urethra. This would occur once or twice weekly, and was more apt to happen when he was constipated. No feces were noted, although the urine continued to be grossly infected. No urine was passed by way of the rectum. Later in 1953 he consulted another urologist who did further studies, including cystoscopy and pyelography. No infection was found in the upper urinary tract, but there was an anterior ball-valve hypertrophy of the prostate, and a transurethral prostatectomy was done in the fall of 1953. From that time on, the acute attacks of cystitis ceased, but gross pyuria continued despite the use of all the available antibiotics and urinary antiseptics. He was treated intensively for a year or so, then advised to continue on longterm sulfonamide therapy. In January 1954 he had a proctoscopic examination and barium enema, which failed to show a fistula. In 1956 a laparotomy was performed, with the assistance of his urologist. The gallbladder was removed because of stones. A complete examination of the intestinal tract failed to reveal any connection with the bladder. Except for the intermittent passage of gas per urethram, he did fairly well. Since the first of the year he had had more severe pyuria and had been under intensive medical therapy for the previous 7 months.
273
While under medical care, a methylene blue enema was carried out, at which time the patient passed part of the enema through the urethra. He was then referred for urologic study. The patient had had pneumonia in 1945 and 1958. Appendectomy was performed at age 14. Tonsillectomy and submucous resection of the nasal septum had been done and all teeth had been removed for pyorrhea. He denied any operative procedure or abscess formation in or about the anus and rectum. Venereal disease was denied. The family history and marital history were non-contributory. Physical examination disclosed a slender white man who was afebrile. On abdominal examination there was a vaguely defined mass in the right mid-abdomen. The genitalia were normal. On rectal examination the prostate was palpable and appeared to be almost flat, with no induration. On withdrawing the examining finger from the rectum, a dimple or depression was felt in the anterior rectal wall at about the level of the proximal anal sphincter. He was therefore hospitalized for further study. On August 22, 1963 a cystogram was made in the anterior-posterior and right semilateral positions and showed no deformity or fistula. The bladder was drained and air injected for contrast and a urethrogram was made (fig. 1, A). No fistula was demonstrated. Cystourethroscopic examination showed no definite opening in the
Fm. 1. A, urethrogram in right semi-lateral position, with air in bladder. No fistula demonstrated. B, cystogram made in right semi-lateral position by injection of dye through 4F catheter passed through rectal opening of rectourethral fistula. Note catheter entering distal prostatic portion of urethra to left of midline.
274
LEDUC
urethra. The prostate had been well resected; several open prostatic duct orifices were seen and probed with 4F ureteral catheters without success. An upper tract study was made, showing a crossed renal ectopia with fusion on the right side. Study of the urine from each ureteral catheter showed no pyuria, no bacteria on gram stain and none on cultural study. Culture of urine from the bladder gave a growth of Escherichia coli and paracolon organisms. On the same day, Dr. Myron Saltz performed proctoscopic and anoscopic examinations, and confirmed the suspected presence of an opening at the site of the depression felt on rectal examination. He was able to pass a ureteral catheter, size 4F, through the opening into the prostatic portion of the urethra and on into the bladder. Cystograms were again made, showing the location of the urethral orifice to be in the distal prostatic urethra (fig. l,B). A diagnosis of rectourethral fistula was made from these studies. On August 26, a filiform attached to a flexible 8F follower was passed through the rectal opening and a perinea! dissection then made. The dissection was carried to the posterior surface of the prostate and the fistula was identified. It was easily dissected free from the surrounding tissues and found to be a tubular structure about 5 mm. in diameter, passing from the center of the anteri01· wall of the rectum, obliquely anterolaterally to enter the prostatic capsule at the left inferior portion, proximal to the sphincter. There was no induration about the tract and no adhesions to the surrounding structures. The filiform and follower were removed, and the tract was severed at the prostatic capsule and at the anterior wall of the rectum. Each opening was sutured with 4-0 chromic catgut sutures, reinforcing the rectal opening with a second layer of 2-0 chromic. The openings were so placed as to be separated from each other by a distance of about 1.5 cm. and were further separated by suturing the lcvator ani muscles between the rectal wall and the prostate. The remainder of the closure was routine, with a small Penrose drain left in place. The bladder was drained with an 16F catheter. The patient remained afebrile. There was no induration nor exudate from the perinea! incision. The drain was removed in 72 hours and the urethral catheter on the eighth postoperative day, the patient leaving the hospital on the
following day. He has been observed systematically in the office, and mild pyuria secondary to chronic prostatitis has responded to treatment. No further passage of gas per urethram has been noted, and there are no symptoms pertaining either to the rectum or the urinary tract. DISCUSSION
It is believed that this is the first case report of a man with a rectourethral fistula of congenital origin without associated rectal anomaly. Magee's case so diagnosed had such a large fistula that it was possible to pass a cystoscope through the urethra into the fistula and out through the anus. The duration of symptoms was reported to be 2 years in this German prisoner of war, and it seems impossible to believe that such a fistula would not have produced symptoms from infancy had it been of congenital origin. In the case under discussion, it is felt that the small size of the fistula and the location of the rectal orifice close to the internal anal sphincter are reasonable explanations of the failure of this patient to pass feces into the urinary tract, or urine into the bowel. No explanation is evident concerning the failure to pass gas at an earlier age, but it is highly reasonable to suppose that infected material must have passed through it in order to cause the repeated infections of the urinary tract from age 3, the source of which defied discovery on numerous urologic examinations. Only when the dimple in the anterior rectal wall was found on digital rectal examination was there a lead to the definitive diagnosis. SUMMARY
The literature has been surveyed and no report found of congenital rectourethral fistula without rectal anomaly. Report of a man, aged 59, with a rectourethral fistula of congenital origin without rectal anomaly, has been presented. It is recommended that careful digital rectal and anoscopic examinations be made when fistula is suspected but not found by other usual 1nethods of examination. Closure of small urethrorectal fistulas may safely be made by perineal dissection and repair of both openings without cystostomy or colostomy. 2330 First Avenue, San Diego, California REFERENCES ATAssr, S. A. AND McEvER, V. W., JR.: Urethro-
rectal fistula, 45 years in duration. J. U rol., 89: 60, 1963.
COXGENITAL RECTOURETHHAL FISTULA
BENSON, C. D., lVlT;sTAR.D, W. T., RAVITCH, lVI., SNnrnR, W. II., JR., AND WEu:n, K. J.: Pedi .. atric Surgery. Chicago: Yearbook Medical Publishers, Jnc., vol. 2, p. 824, Hl62. BuowNE, D.: Some congenital deformities of the rectum, anus, vagina nnd urcthrn. Ann. Roy. Col. Surg., 8: 173--192, 1951. CAMPBELL, M. F.: Urethro-rcctal fistula . .J. Urol., 76: 411, Hl5G. CAMPBELL, M. F.: Clinical Pediatric Urology. Philadelphia: W. B. Saunders Co., Hl51. CAMPBELL, M. F.: Urology. Philadelphia: W. B. Saunders Co., HJ54. CrmrsTOPER, F.: Textbook of Surgery. Philadel-phia: W. B. Saunders Co., 7th ed., 1960. DUFFY, J. J.: Imperforat.e anus and recto-urethral fistula in a. 27-year-old man . .J.A.M.A., 145: 545-548 , 1951. GILLASPY, C. C. AND YAN HEGTERGHEM, 0 . .J.: Unilateral renal agenesis associated with a recto-urethral fistula. Brit. J. Urol., 34: 270-273, 1962. GooowrN, W. E., T1;RNER, R. D. AND WIN'l'ER, C. C. · Hect.o-urinary fistula; principles of management and the technique of surgical closure. J. U rol., 80: 246-254, 1958. GRoss, IL E.: The Surgery of Infancy and Childhood. Philadelphia: W. B. Saunders Co., 1953. HAMPTON, ,l. M. AND BACUN, H . .E.: Diagnosis and surgical management, of recto-urethra.I fistula. Dis. Colon Rectum, 4: 177-180, 1961. LAIRD, D. R. · Procedures used in treatment of
complicated fistulas. Amer. ,J. :-iurg., 76: 701 l\J48. LEWIS, L. G.: Hepair of recto-urethral fistulns. .J. Urol., 57: 1173-1181, Hl47. MAGEE, R. K.: Recto-urethral fistula; report of ,., case. Lancet, 1: 140, HJ48. ;\JuGENT, ,J. .J .. A method used to cure a redourethral fistula. J. Urol., 57: 35G-:358, Hl-!7 POT'l'S, W., RIKER, W. L. AND DEBOER, A : lrn perforate anus with recto-vesical and urethral, vaginal and perinea] fistulas. Ann Surg., 140: 381-395, 1954. SMrrH, A.G. AND BooNE, A. W.: Eetopic urethral anus in recto-urethral fistula. A.M.iL Arcli. Surg., 73: 215-219, 1957. STEWART, R. L. AND Itoss, ,J. A.: Congenital rectourinary fistula. Brit. ,J. Urol., 53: ?HJ-722, 1945. SWENSON, 0.: Pediatric Surgery. ~ew York: Appleton-Century-Crofts, 2nd ed., 1962, pp. 449-462. \' osE, S. N.: A technique for repair of recl.o urethral fistula. J. Urol., 61: 791-794, 1949. WENZEL, J. F. AND JENKINS, E. A.: Recto-urethral fistula. J. Mich. Med. Soc., 49: 67-68, HJ,50 W~JYRAUCH, H. M., JR.: A critical study of surgical principles used in repair of recto-urethral fistula, Stanford Med. Bull., 9: 2-12, 1951. YouNG, H. H.: Imperforate anus· bowel into urethra; hyposadias. 1448--1451, 1936