Primary Carcinoma in a Diverticulum of the Female Urethra: Review of the Literature and Report of a Case

Primary Carcinoma in a Diverticulum of the Female Urethra: Review of the Literature and Report of a Case


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Vol. 82, No. 3, September 1959 Printed in U.S.A.

PRIMARY CARCINOMA IN A DIVERTICULUM OF THE FEMALE URETHRA: REVIEW OF THE LITERATURE AND REPORT OF A CASE JAMES W. FAULKNER Numerous publications appearing in the gynecological and urological literature attest to the increasing awareness of the presence of urethral diverticula in women with chronic urethral symptoms. Striking evidence of this fact is presented by contrasting the recent report of Edwin W. Brown, who detected 23 cases in 6:J,2 years in the private practice of urology,1 with the 22 cases diagnosed in a 19 year period at the Johns Hopkins Hospitals from 1931 to 1949. 2 Most urologists have had a varying number of personal cases and it is probable that the true incidence of clinical diagnosis is much higher than the representation in the literature would indicate. That some delay exists before the diagnosis is ordinarily established is suggested by a report in 1956 in which 24 of 66 patients found to have an urethral diverticulum had manifested symptoms for a period exceeding 5 years. 3 Such a time interval no doubt includes the formative periods of many diverticula, but is indicative that lengthy periods of discomfort could be curtailed by more prompt discovery of this condition. The etiology of these lesions has been accepted to be on an acquired basis in the majority of instances. Periurethral submucosal inflammation is the initial process and leads to abscess formation with subsequent rupture into the urethral lumen and consequent diverticulum formation. The presenting features of dysuria, dyspareunia, frequency, and dribbling have received thorough recognition, as have the physical findings of a suburethral mass and the occurrence of purulent matter and/or urine at the meatus as elicited with vaginal counterpressure. In a review of the literature, the presence of stones in these diverticula is reported to occur in 10 per cent of cases studied by Wharton and Kearns. The apparent ideal conditions of stasis and infection leading to stone formation have been mentioned by Kirby and Reynolds, who gathered reports of 44 stones Accepted for publication January 29, 1959. Brown, E. W.: Diverticulum of the female urethra. South. M. J., 49: 982-988, 1956. 2 Wharton, L. R. Jr. and TeLinde, R. W.: Urethral diverticulum. Obst. & Gynec., 7: 503-509, 1


3 Wharton, L. R. and Kearns, W.: Diverticula of the female urethra. J. Urol., 63: 1063-1076, 1950.

which had appeared in urethral diverticula. 4 One might conjecture that these same factors of chronic infection and irritation could act as inciting agents for neoplastic changes and result in the development of tumors in these pouches. However, to date, only three primary malignancies arising in an urethral diverticulum have been discovered in a survey of the literature.* Hamilton and Leach reported a primary adenocarcinoma developing in a 53-year-old woman who had noted vaginal bleeding for one year. 6 Wishard and Nourse found a transitional cell epithelioma in the diverticulum of a 39-year-old woman who had had chronic bladder irritability for many years. 6 Brown reported the second adenocarcinoma in a 40-year-old woman with recurrent cystitis for 6 years. The purpose of this paper is to report the fifth case of a primary tumor arising in a diverticulum of the female urethra, and to include it in evaluation of the few similar cases previously reported. CASE REPORT

Mrs. A. T. (Hosp. No. 5575), a 57-year-old widow, was first seen in the emergency room on January 16, 1955, with the presenting complaint of painful, bloody urination. She stated that she had been in good health until the preceding evening when she had experienced suprapubic aching and discomfort, associated with the passage of large amounts of blood in the urine. Dysuria and some impairment of the force of the stream were also noted, but there was no frequency or urgency. There had been no flank discomfort nor previous similar symptomatology. 4 Kirby, E. W. Jr. and Reynolds, C. J.: Diverticula of the female urethra. J. Urol., 62: 498-502,


* Personal communication with Edwin W. Brown reveals that Dr. Horace Atkinson found an extensive adenocarcinoma in 1954. The patient died of widespread involvement in 6 months despite urethral excision and suprapubic cystostomy. Details of this case are included through their courtesy. 6 Hamilton, J. D. and Leach, W. B.: Adenocarcinoma arising in a diverticulum of the female urethra. Arch. Path., 51: 90-97, 1951. 6 Wishard, W. N. Jr. and Nourse, M. H.: Carcinoma in diverticulum of female urethra. J. Urol., 68: 320-323, 1952.




Frn. 1. Diverticulogram reveals cerebriform pattern suggesting tumefaction within diverticulum. Bladder outlined by dotted line. In 1931 she had passed a small stone from the bladder, but had had no subsequent urological difficulties. There had been no exposure to tuberculosis and the family history was negative. She had always been in good health and had had two normal deliveries, the latter one some twentyeight years prior to her admission. Physical examination disclosed that positive findings were limited to the pelvic area, where blood was present at the external urethral meatus. One centimeter proximal and beneath the meatus was an exceedingly firm 3 cm. mass. The cervix and remainder of the vagina were normal. Laboratory studies disclosed a normal hemogram. A catheterized specimen of urine was yellow, slightly cloudy, with a pH of 7.2. The specific gravity was 1.005. There was a trace of albumin but no sugar or acetone. Microscopic examination disclosed an occasional white blood cell, a rare epithelial cell, and 5-6 red blood cells per high power field. A culture of this specimen was sterile. On January 17, 1955, cystoscopy and retrograde pyelography were accomplished. The bladder and ureteral orifices were normal and pyelograms showed no abnormality of the kidneys or ureters. Examination of the urethra, however, revealed two small apertures, each 2-3 mm. in diameter, about 12 mm. within the external urethral meatus. On visualizing these orifices with a direct vision cystoscope and using vaginal counterpressure on the suburethral mass, bloody material could be seen exuding into the urethra and within the diverticulum could be seen con-

tents that resembled brain-like tissue filling the enclosing sac. A small uret.eral catheter was passed into the diverticulum and a diverticulogram made (fig. 1). The radiological report was as follows: "There is evidence of an urethral diverticulum just about the level of the symphysis pubis. The cerebriform pattern of the diverticulogram may represent tumefaction within the sac." A presumptive diagnosis of a papillary tumor within an urethral diverticulum was made, and on January 19, 1955 the area was surgically exposed. Under general anesthesia and with a size 22F Foley catheter in the bladder, a longitudinal incision was made in the anterior vaginal wall from just beneath the external urethral meatus to the cervix. The vaginal flaps were elevated and the diverticulum freed from the pubocervical fascia by sharp dissection. It became apparent that the sac was firmly adherent to the undersurface of the urethra extending back to the region of the trigone. The diverticulum was dissected posteriorly and removed intact, taking

Frn. 2. Microscopic section of transitional cell epithelioma of urethral diverticulum reveals fine connective tissue stalks covered with transitional type of epithelium having hyperchromatic and pleomorphic nuclei.



1. Analysis of reported cases of primary carcinomas in diverticula of the female urethra

Authors Clinical Findings Pathology Treatment Type Results - - - - - - - - - - 1 - - - - - - - - - - - - - - - - - - - - - - - - 1 ------------1-------------------- - - - - - - - - - - - -

Hamilton and Leach 1951

Wishard and Nourse 1952 Brown, E.W. 1956

Age 53 Vaginal bleeding for 1 year. 3 cm. urethral mass


Age 39 Frequency and dysuria for 2 yr. 2 cm. urethral mass Age 46 Recurrent cystitis for 6 yr. "Pulpaceous urethral

Transitional cell ca.

100 roentgen pre-op. Surgical excision. 9

mo. post-op. excision of recurrence. 13 mo. 880 mg./hr. radium Surgical excision

Died 2 yr. after surgery Alive and well 7 yr.


Surgical excision. 8 mo. later implant five radon seeds

Alive and well 5 yr.


Total removal urethra. Permanent suprapubic cystostomy Surgical excision. 33 mo. post-op. small recurrence. 5000 roentgen to area

Died 6 mo. after surgery Alive and well 4 yr.


Atkinson, H. (unpublished) Faulkner, J. 1959

Age 53 Gross hematuria and dysuria for 1 yr. Age 57 Painful hematuria for 1 day 3 cm. urethral mass

Transitional cell ca.

with it a segment of the ventral aspect of the urethra about 2 cm. in length, extending to, and including, the vesical neck. The urethral and vesical neck mucosa was sutured over the exposed Foley catheter with interrupted mattress sutures of 000 atraumatic chromic catgut. All possible caution was exercised to restore the integrity of the bladder neck by bringing urethral musculature together with a second layer of similar sutures. Interrupted catgut sutures were then used to close the pubocervical fascia and the vaginal mucosa, this last mentioned layer being placed to the right side of the urethra by excising redundant tissue on that side, in order to avoid superimposition of all suture lines. The postoperative course was uneventful. The indwelling catheter was left in place for an extended period of 10 days in order to minimize the possibility of incontinence or fistula formation, after removal of almost the entire urethral floor. Upon removal of the catheter, the patient voided with good control and left the hospital 14 days after surgery. The report on the pathology specimen read: "The specimen was round, lobular and measured 3.5 cm. in diameter. The outer fibrous capsule appeared to be intact and was filled with a papillary soft, gray material which apparently arose from the mucosa of this diverticulum. On micro-

scopic examination, the material from the diverticulum appeared to be a papillary type of tumor which was made up of fine connective tissue stalks covered with a transitional type of epithelium having hyperchromatic and somewhat pleomorphic nuclei. A few mitotic figures were seen in the tumor, especially in the more cellular portions of the tumor." Diagnosis: Papillary transitional cell epithelioma of an urethral diverticulum (fig. 2). Signed James D. Barger. The patient was seen at intervals of 3 months after dismissal from the hospital. She remained well until October 1957 when a small nodule was noted in the :previous operative site. This histologically was a recurrence and was treated with approximately 5000 roentgen over a 4 week period. She has been without difficulty since that time and the urethra and vagina appeared to be normal in November 195S. COMMENT

Maximow and Bloom state that the female urethra is lined with a stratified squamous epithelium. Brack found that most primary carcinomas of the female urethra, in his collected series of 338 cases, were of the squamous cell type.7 All of his 10 personal cases were epider7 Brack, C. B. and Farber, G. J.: Carcinoma of the female urethra. J. Urol., 64: 710--715, 1950.



moid carcinomas. In contrast, of the five primary carcinomas in a diverticulum, which includes the present case, three have been adenocarcinomas, presumably from periurethral glands; while the case herein reported and that of Wishard and Nourse have been of the transitional cell variety. Regarding clinical features in the 5 cases, symptoms varied from mild vesical irritability to acute retention, but every patient had a suburethral mass and three of the five had gross hematuria. Symptom duration was only 1 day in our case, while the others had noted suggestive difficulty for from one to six years. Followup studies in the 5 cases have shown good results in three, with these patients currently surviving four to seven years apparently free from tumor. Twp patients died of their malignancy, one in 6 months, the other in 2 years (table 1). This record shows some improvement over carcinoma of the urethra itself where a 35 per cent five year survival has been attained. Possibly the "extraurethral" site of tumors in diverticula lends itself to more expeditious surgical removal and accounts for better results in regard to survival and function. Ancillary radiation was used preoperatively in one case and for the treatment of suspected recurrences in two others. From reports surveyed it would appear that the diagnosis of an urethral diverticulum should be made without undue difficulty by the prac-

ticing urologist. The high incidence of a suburethral mass and purulent matter at the urethral orifice (58 and 47 cases respectively of the 66 reviewed by Wharton and TeLinde) should serve to firmly establish this diagnosis. Yet TeLinde recently indicated that many of his cases had been examined repeatedly by urolog~ts and the diverticulum went undiscovered. 8 Such an oversight of a diverticulum containing a tumor is particularly undesirable for, though the number of cases is too small for analysis, it would appear that reasonably prompt removal of such tumors is associated with satisfactory long term results in a significant percentage of the cases seen. SUMMARY

A case of primary transitional cell carcinoma arising in a diverticulum of the female urethra is presented. Four previous reports are also reviewed as to their pathological features, clinical findings, and prognosis. In view of the reasonably good results of therapy in these cases, the desirability of prompt diagnosis and removal of urethral diverticula is mentioned.

636 Church St., Evanston, Ill. 8 TeLinde, R. W.: Discussion of paper of diverticulum of the female urethra by Knight, J. R. and Hill, N. N. Jr.: Am. J. Obst. & Gynec., 70:

1214-1218, 1955.