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Calculus-containing Urethral Diverticulum in a Woman: Report of a Case
CALCULUS-CONTAINING URETHRAL DIVERTICULUM IN A WOMAN: REPORT OF A CASE CHARLES C. HIGGINS
AND
PHILIP R. ROEN
From the Cleveland Clinic, Cleveland
Diseases of the urethra are frequently overlooked as a cause of urinary symptoms in women. Folsom was one of the first to emphasize this fact and to focus the attention of the urologist on this small but very important anatomical structure. He pointed out the significance of the presence of glandular elements in the posterior urethra, which are productive of urinary symptoms after infection occurs. It is the purpose of this report to cite an unusual cause of such urinary symptoms in a woman, in which the lesion may have arisen from such a gland of the posterior urethra, and to call attention to the female urethra as the possible source of urinary complaints. Urethral diverticulum in women is rather uncommon. Although exact statistics as to the frequency of this lesion are not available, 19 such cases were found in a IO-year period at the Mayo Clinic (cited by Parmenter). At the Cleveland Clinic only 7 cases including the one herein reported have been observed in the 22 years since 1921. Calculus formation in a diverticulum of the urethra is extremely rare. In a review of the literature prior to 1939, Gaston and Ferrucci were able to collect 33 cases and added one of their own. We have been able to find published reports of 4 additional cases which makes a total of 39 cases in the literature including the one presented here. Standard textbooks leave much to be desired in describing this condition. In the basic work of Young, diverticulum of the female urethra is not mentioned. More modern textbooks devote but a few lines (Hinman) or fail to describe the lesion (Eisendrath and Rolnick); however, the latest comprehensive text on urology (Lowsley and Kirwin) describes the condition in detail. Nevertheless, it is evident that the literature gives scant consideration to this entity. ORIGIN
Urethral diverticulum may be defined as a pouch formed by dilatation of a portion of the urethrovaginal septum, and communicating with the urethral canal. The pouch may communicate widely with the urethra, or it may have a narrow or tubular opening into it. The etiology of urethral diverticulum itself is unsettled, both the congenital and acquired theories having been quoted as explanation for its origin. Probably a urethral diverticulum occasionally does arise as a result of anomalous development, but it is more frequently acquired. It is quite likely that the true congenital diverticulum of the urethra more often presents features that simulate cystocele than do those sacs which arise secondarily. There are two principal causes of the acquired type: (1) Trauma may produce a weakened point in the urethral wall, and later, 715
716
CHARLES C. HIGGINS AND PHILIP R. ROEN
herniation may occur through this area. Trauma may be in the form of passage of a calculus, instrumentation, or childbirth. Undoubtedly, the last is the most frequent causative factor. Instrumentation is not so often the cause of this intraurethral injury in the female as it is in the longer pendulous urethra of the male. (2) Infection: An inflammatory process in the periurethral tissues may serve to produce a diverticulum. Inflammatory occlusion of a urethral duct may produce a retention cyst of its associated gland, and subsequent suppuration and rupture into the urethral lumen serves by repetition of these events to produce an increasingly larger sac. If the mouth of the diverticulum is wide, it is doubtful whether calculus formation can occur, for stone formation in such a diverticulum is probably due to local urinary stasis with infection which allows for the precipitation of urinary salts. Thus, theoretically the presence of a calculus in a diverticulum would presuppose a narrow opening between the urethra and the diverticulum. Such was the finding in our case. The rarity of the finding of such calculi is probably result of the periodic emptying of the diverticulum with coitus, changes of posture, and so forth. No doubt in an occasional case it is entirely conceivable that a stone descending from the upper urinary tract may become lodged in a preformed urethral diverticulum, where it increases in size by accretion. Bibus has indicated that in such cases multiple stones are to be found, whereas, a single stone is usually present when calculus formation occurs primarily within the diverticulum. Symptoms of urethral diverticulum, with or without calculus, are those of lower urinary tract infection including frequency and dysuria, nocturia, pyuria, and rarely hematuria, and in instances in which the calculus is large, urethral obstruction may occur. Local examination reveals a smooth, spherical, visible, and palpable swelling of the anterior vaginal well, just posterior to the external urethral meatus. This must not be confused with cystocele. In the presence of a calculus, this swelling is hard. Urethroscopic inspection and x-ray, especially a cystogram with urethral catheter in place, confirm the diagnosis. The treatment of urethral diverticulum with or without stone is entirely surgical. The sac of the diverticulum is readily approached through incision in the anterior vaginal wall. Resection of the sac with resuture of the urethral and vaginal walls has given universally satisfactory results. Diversion of the urinary stream for approximately a week is a necessary feature of the treatment. In most cases this may be accomplished by an inlying catheter; rarely, suprapubic cystotomy may be required. In all reported cases healing has occurred promptly without the formation of urethrovaginal fistula.
a
REPORT OF A CASE
A 66 year old woman registered at the Cleveland Clinic on September 24, 1942 complaining of dysuria, frequency, difficulty in voiding, and slight terminal hematuria. The past history was irrelevant except for the fact that the patient had experienced a severe attack of "cystitis" which had subsided in a month's time some 3 to 4 years ago. The present urinary symptoms had appeared about
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FIG. 1
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Fm. 1. X-ray film of the pelvis showing the large calculus Fw. 2. The calculus and its relation to the bladder; cystogram obtained by intravenous dye.
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CHARLES C. HIGGINS AND PHILIP R. ROEN
8 months prior to her visit to the Clinic. The symptoms had been prcgressive, especially the difficulty in voiding. Physical examination revealed a somewhat obese, elderly woman who showed no abnormal findings except for the presence of a hard, bulging mass in the anterior vaginal wall. This was clinically diagnosed as a calculus ·within a diverticulum of the urethra.
DIVERTICULU M Fm. 3. Schematic drawing illustrating a large diverticulum of the urethra
The red blood study showed a very mild hypochromic anemia; the white blood count was normal. The blood urea and sugar were normal. The complement fixation test was negative. X-ray study disclosed a large calculus below the level of the bladder (figs. 1 and 2). The urogram showed a normal upper urinary tract. Urinalysis revealed a number of white blood cells and red blood cells. The urine culture showed B. coli. At operation the anterior vaginal wall was incised longitudinally, the diverticulum sac was opened, and the calculus extracted. The sac (fig. 3) was then resected, and the orifice into the urethra closed by purse-string suture. This orifice was quite narrow with a diameter of approximately 5 mm. The vaginal
CALCULUS-CONTAINING URETHRAL DIVERTICULUM
719
wall was sutured. A catheter was left indwelling for 6 days postoperatively. The wound healed by primary intention; and the patient returned home 11 days after operation, free of symptoms. The calculus was large, measuring 3.9 by 2.6 by 2.5 cm., and was smooth, hard, and laminated with a soft center. Chemical analysis showed it to be composed of calcium, phosphates, carbonates, and a trace of ammonia. Microscopic section of the diverticulum wall disclosed it to be lined with squamous epithelium. COMMENT
1Jndoubtedly, the urethraldiverticulum in this case ,vas of the acquired type; this is deduced from the history of the absence of urinary symptoms until infection appeared late in life. The exact nature of the infection is not evident; there may have been an abscess of a periurethral gland which produced by its rupture a shallow pouch, thus laying the groundwork for the subsequent development and enlargement of the diverticulum in which later calculus formation occurred. The sequence of events in this case may be assumed to have been: (1) infection, as indicated by the history of antecedent urinary symptoms; (2) lapse of time in which the diverticulum enlarged progressively and urinary salts precipitated in the stagnant urine leading to calculus formation--the laminated character of the calculus evidences such an event; (3) recurrent urinary symptoms due to the presence of the calculus-containing diverticulum. Surgery consisted of removal of the calculus and simultaneous excision of the diverticulum; diversion of the urinary stream ,vas accomplished by means of an indwelling catheter. These measures resulted in complete cure. SUMMARY
The thirty-ninth reported case of calculus formation in a urethral diverticulum in a woman is presented. The calculus probably formed in situ from precipitation of urinary salts in the presence of urinary stasis. The urethra in women should not be overlooked as a cause of urinary symptoms. REFERENCES ANGLADA, E.: Diverticular calculi of female urethra with report of cases. Vida Nueva., 48: 277-282, 1941 Brnus, B.: Zur Klinik der Harnrohrensteine beim Weibe. Zeitschr. f. Urol., 31: 473-479, 1937. DE SousA, E., AND CAMPOS DE PAz, JR.: Urethral calculus in female. Rec. de gynec. e d'obst., 1: 421, 1939. EDWARDS, F. D., AND DEDDENS, L. E. L.: Calculus in diverticulum of female urethra; report of case. J. A. M. Georgia, 28: 449-451, 1939. EISENDRATH, D. N., AND RoLNICK, H. C.: Textbook of Urology. Philadelphia: J. B. Lippincott Co., 1938. FOLSOM, A. I.: The female urethra; clinical and pathological study. J. A. M. A., 97: 1345-1351, 1931. GASTON, E. A., AND FERRUCCI, J.: Calculus formation in urethral diverticulum in woman. New Eng. J. Med., 221: 379-383, (Sept.) 1939. HINMAN F.: Principles and Practice of Urology. Philadelphia: W. B. Saunders Co., 1935. LowsLE.;,., 0. S., AND KrnwIN, T. J. : Clinical Urology. Baltimore: Williams & Wilkins Co., 1940. PARMENTER, F. J.: Diverticulum of female urethra. J. Urol., 45: 476-496, 1941. YouNG, HuGH H., AND DAVIS, D. M.: Young's Practice of Urology. Philadelphia: W. B. Saunders Co., 1926.
AND
PHILIP R. ROEN
From the Cleveland Clinic, Cleveland
Diseases of the urethra are frequently overlooked as a cause of urinary symptoms in women. Folsom was one of the first to emphasize this fact and to focus the attention of the urologist on this small but very important anatomical structure. He pointed out the significance of the presence of glandular elements in the posterior urethra, which are productive of urinary symptoms after infection occurs. It is the purpose of this report to cite an unusual cause of such urinary symptoms in a woman, in which the lesion may have arisen from such a gland of the posterior urethra, and to call attention to the female urethra as the possible source of urinary complaints. Urethral diverticulum in women is rather uncommon. Although exact statistics as to the frequency of this lesion are not available, 19 such cases were found in a IO-year period at the Mayo Clinic (cited by Parmenter). At the Cleveland Clinic only 7 cases including the one herein reported have been observed in the 22 years since 1921. Calculus formation in a diverticulum of the urethra is extremely rare. In a review of the literature prior to 1939, Gaston and Ferrucci were able to collect 33 cases and added one of their own. We have been able to find published reports of 4 additional cases which makes a total of 39 cases in the literature including the one presented here. Standard textbooks leave much to be desired in describing this condition. In the basic work of Young, diverticulum of the female urethra is not mentioned. More modern textbooks devote but a few lines (Hinman) or fail to describe the lesion (Eisendrath and Rolnick); however, the latest comprehensive text on urology (Lowsley and Kirwin) describes the condition in detail. Nevertheless, it is evident that the literature gives scant consideration to this entity. ORIGIN
Urethral diverticulum may be defined as a pouch formed by dilatation of a portion of the urethrovaginal septum, and communicating with the urethral canal. The pouch may communicate widely with the urethra, or it may have a narrow or tubular opening into it. The etiology of urethral diverticulum itself is unsettled, both the congenital and acquired theories having been quoted as explanation for its origin. Probably a urethral diverticulum occasionally does arise as a result of anomalous development, but it is more frequently acquired. It is quite likely that the true congenital diverticulum of the urethra more often presents features that simulate cystocele than do those sacs which arise secondarily. There are two principal causes of the acquired type: (1) Trauma may produce a weakened point in the urethral wall, and later, 715
716
CHARLES C. HIGGINS AND PHILIP R. ROEN
herniation may occur through this area. Trauma may be in the form of passage of a calculus, instrumentation, or childbirth. Undoubtedly, the last is the most frequent causative factor. Instrumentation is not so often the cause of this intraurethral injury in the female as it is in the longer pendulous urethra of the male. (2) Infection: An inflammatory process in the periurethral tissues may serve to produce a diverticulum. Inflammatory occlusion of a urethral duct may produce a retention cyst of its associated gland, and subsequent suppuration and rupture into the urethral lumen serves by repetition of these events to produce an increasingly larger sac. If the mouth of the diverticulum is wide, it is doubtful whether calculus formation can occur, for stone formation in such a diverticulum is probably due to local urinary stasis with infection which allows for the precipitation of urinary salts. Thus, theoretically the presence of a calculus in a diverticulum would presuppose a narrow opening between the urethra and the diverticulum. Such was the finding in our case. The rarity of the finding of such calculi is probably result of the periodic emptying of the diverticulum with coitus, changes of posture, and so forth. No doubt in an occasional case it is entirely conceivable that a stone descending from the upper urinary tract may become lodged in a preformed urethral diverticulum, where it increases in size by accretion. Bibus has indicated that in such cases multiple stones are to be found, whereas, a single stone is usually present when calculus formation occurs primarily within the diverticulum. Symptoms of urethral diverticulum, with or without calculus, are those of lower urinary tract infection including frequency and dysuria, nocturia, pyuria, and rarely hematuria, and in instances in which the calculus is large, urethral obstruction may occur. Local examination reveals a smooth, spherical, visible, and palpable swelling of the anterior vaginal well, just posterior to the external urethral meatus. This must not be confused with cystocele. In the presence of a calculus, this swelling is hard. Urethroscopic inspection and x-ray, especially a cystogram with urethral catheter in place, confirm the diagnosis. The treatment of urethral diverticulum with or without stone is entirely surgical. The sac of the diverticulum is readily approached through incision in the anterior vaginal wall. Resection of the sac with resuture of the urethral and vaginal walls has given universally satisfactory results. Diversion of the urinary stream for approximately a week is a necessary feature of the treatment. In most cases this may be accomplished by an inlying catheter; rarely, suprapubic cystotomy may be required. In all reported cases healing has occurred promptly without the formation of urethrovaginal fistula.
a
REPORT OF A CASE
A 66 year old woman registered at the Cleveland Clinic on September 24, 1942 complaining of dysuria, frequency, difficulty in voiding, and slight terminal hematuria. The past history was irrelevant except for the fact that the patient had experienced a severe attack of "cystitis" which had subsided in a month's time some 3 to 4 years ago. The present urinary symptoms had appeared about
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FIG. 1
FIG. 2
Fm. 1. X-ray film of the pelvis showing the large calculus Fw. 2. The calculus and its relation to the bladder; cystogram obtained by intravenous dye.
-:r I-'
--..1
718
CHARLES C. HIGGINS AND PHILIP R. ROEN
8 months prior to her visit to the Clinic. The symptoms had been prcgressive, especially the difficulty in voiding. Physical examination revealed a somewhat obese, elderly woman who showed no abnormal findings except for the presence of a hard, bulging mass in the anterior vaginal wall. This was clinically diagnosed as a calculus ·within a diverticulum of the urethra.
DIVERTICULU M Fm. 3. Schematic drawing illustrating a large diverticulum of the urethra
The red blood study showed a very mild hypochromic anemia; the white blood count was normal. The blood urea and sugar were normal. The complement fixation test was negative. X-ray study disclosed a large calculus below the level of the bladder (figs. 1 and 2). The urogram showed a normal upper urinary tract. Urinalysis revealed a number of white blood cells and red blood cells. The urine culture showed B. coli. At operation the anterior vaginal wall was incised longitudinally, the diverticulum sac was opened, and the calculus extracted. The sac (fig. 3) was then resected, and the orifice into the urethra closed by purse-string suture. This orifice was quite narrow with a diameter of approximately 5 mm. The vaginal
CALCULUS-CONTAINING URETHRAL DIVERTICULUM
719
wall was sutured. A catheter was left indwelling for 6 days postoperatively. The wound healed by primary intention; and the patient returned home 11 days after operation, free of symptoms. The calculus was large, measuring 3.9 by 2.6 by 2.5 cm., and was smooth, hard, and laminated with a soft center. Chemical analysis showed it to be composed of calcium, phosphates, carbonates, and a trace of ammonia. Microscopic section of the diverticulum wall disclosed it to be lined with squamous epithelium. COMMENT
1Jndoubtedly, the urethraldiverticulum in this case ,vas of the acquired type; this is deduced from the history of the absence of urinary symptoms until infection appeared late in life. The exact nature of the infection is not evident; there may have been an abscess of a periurethral gland which produced by its rupture a shallow pouch, thus laying the groundwork for the subsequent development and enlargement of the diverticulum in which later calculus formation occurred. The sequence of events in this case may be assumed to have been: (1) infection, as indicated by the history of antecedent urinary symptoms; (2) lapse of time in which the diverticulum enlarged progressively and urinary salts precipitated in the stagnant urine leading to calculus formation--the laminated character of the calculus evidences such an event; (3) recurrent urinary symptoms due to the presence of the calculus-containing diverticulum. Surgery consisted of removal of the calculus and simultaneous excision of the diverticulum; diversion of the urinary stream ,vas accomplished by means of an indwelling catheter. These measures resulted in complete cure. SUMMARY
The thirty-ninth reported case of calculus formation in a urethral diverticulum in a woman is presented. The calculus probably formed in situ from precipitation of urinary salts in the presence of urinary stasis. The urethra in women should not be overlooked as a cause of urinary symptoms. REFERENCES ANGLADA, E.: Diverticular calculi of female urethra with report of cases. Vida Nueva., 48: 277-282, 1941 Brnus, B.: Zur Klinik der Harnrohrensteine beim Weibe. Zeitschr. f. Urol., 31: 473-479, 1937. DE SousA, E., AND CAMPOS DE PAz, JR.: Urethral calculus in female. Rec. de gynec. e d'obst., 1: 421, 1939. EDWARDS, F. D., AND DEDDENS, L. E. L.: Calculus in diverticulum of female urethra; report of case. J. A. M. Georgia, 28: 449-451, 1939. EISENDRATH, D. N., AND RoLNICK, H. C.: Textbook of Urology. Philadelphia: J. B. Lippincott Co., 1938. FOLSOM, A. I.: The female urethra; clinical and pathological study. J. A. M. A., 97: 1345-1351, 1931. GASTON, E. A., AND FERRUCCI, J.: Calculus formation in urethral diverticulum in woman. New Eng. J. Med., 221: 379-383, (Sept.) 1939. HINMAN F.: Principles and Practice of Urology. Philadelphia: W. B. Saunders Co., 1935. LowsLE.;,., 0. S., AND KrnwIN, T. J. : Clinical Urology. Baltimore: Williams & Wilkins Co., 1940. PARMENTER, F. J.: Diverticulum of female urethra. J. Urol., 45: 476-496, 1941. YouNG, HuGH H., AND DAVIS, D. M.: Young's Practice of Urology. Philadelphia: W. B. Saunders Co., 1926.