Retrovesical Cysts in Boys

& \Villeins Cc.

RETROVESICAL CYSTS IN BOYS SUBIR K. CHATTERJEE

AND

SUSANTA K. SARKAR

From the Pediatric Surgery Unit and Department of Pathology, Nilratan Sarkar Medical College and Hospital, Calcutta, India

Problems associated with cystic swelling behind and below the bladder base in boys have received little attention in standard textbooks of urology, pediatric surgery and even pediatric urology. Our experience with this condition in 2 patients seen within 1 ½ years is described herein. CASE REPORTS

Case 1. J. N ., a 3-year-old boy, presented on February 3, 1970 with gradually increasing dysuria and constipation 2 years in duration. There were occasional 24-hour periods of urinary retention with abdominal pain, which were relieved by medication. The patient weighed 10 kg. and was 83 cm. in height. A mass resembling a distended bladder was

side. A cystogram confirmed the distortion of the bladder by a spherical mass pushing into it from below and behind and on the right. Barium enema showed displacement of the rectum and colon upward, backward and to the left (fig. 1, B). All other clinical and laboratory findings were normal. Laparotomy was carried out on February 20 using a transverse subumbilical incision. A huge cystic mass was found arising from the pelvic floor, displacing the gut upward, backward and to the left and displacing the bladder upward, forward and to the left. The right ureter and the vasa deferentia were stretched tightly over the mass. The ureter above was dilated and led to a markedly hydronephrotic right kidney. The cyst was aspirated and

Fm. 1. Case 1. A, IVP shows non-functioning right kidney and displacement of bladder. B, barium meal discloses displacement of sigmoid colon and rectum and also coils of small gut. C, postoperative IVP reveals return of function in right kidney and normal bladder appearance. seen and palpated in the lower abdomen extending to the umbilicus. Rectal palpation and bimanual palpation of the abdomen revealed the mass to be cystic. The mass filled the pelvis and protruded into the hypogastrium and lower umbilical region. ·when a catheter was per urethram, a small amount of urine was drained but the mass remained unchanged. Excretory urography (IVP) revealed no function on the right side (fig. 1, A). The left kidney showed mild hydronephrotic changes and the left ureter was deviated laterally by a soft tissue mass. The bladder was crescentic in shape owing to indentation by a rounded mass inferiorly and on the right Accepted for publication June 2, 1972. 107

thin turbid chocolate-colored fluid was found. It was separated from the rectum and bladder with some difficulty. The right ureter and the vasa deferentia were transected. Finally the cyst was peeled off the pelvic floor and removed. Right ureteroneocystostomy was carried out using the PolitanoLeadbetter technique. The bladder was drained a suprapubic tube. An indwelling polyvinyl ureteral catheter was led out through the same opening. The pelvic peritoneum was closed, the anterior abdomen was repaired in layers and a Penrose drain was inserted. Convalescence was complicated by intestinal obstruction owing to prolapse of a loop of small gut through the repaired pelvic peritoneum. This condition was corrected by an operation on February 25. Convalescence was uneventful.

108

CHATTERJEE AND SARKAR

Postoperative IVP showed that the left kidney and ureter were almost normal and that function had returned to the large hydronephrotic right kidney (fig. 1, C). Case 2. P. P., a 2-year-old boy, presented on June 4, 1971 with dysuria 5 months in duration and recurrent attacks of urinary retention. An indwelling urethral catheter was used to relieve distress. The patient weighed 10 kg. and was 68 cm. in height. Clinical findings were similar to those in case 1 except that the mass was smaller and extended 5 cm. above the pubis. However, this patient was febrile and blood urea nitrogen (BUN) was 25 mg. per 100 ml. Temperature and BUN returned to normal after antibiotic therapy. IVP revealed non-function on the left side and a mild hydronephrosis and hydrometer on the right side (fig. 2, A). The filling defect in the bladder was almost identical to that in case 1. It was difficult to fill the bladder for cystography (fig. 2, B). Barium enema demonstrated displacement of the gut upward, backward and to the right (fig. 2, C). At laparotomy on August 13, the findings were almost identical to those in case 1. Since the cyst was smaller, preliminary aspiration was not done. The left ureter and left vas deferens were transected but the right vas deferens was saved

and the cyst was removed easily (fig. 2, D). It contained clear fluid. The left kidney was extremely hypoplastic and the left ureter showed multiple strictures. Nevertheless, ureteroneocystostomy was done as in case 1 and the wound was closed. On postoperative IVP the left kidney did not excrete any dye. Convalescence was uneventful. HISTOPATHOLOGY

In case 1 the cyst contained tubules lined with cuboidal or columnar cells containing hyperchroma tic vesicular nuclei with nucleoli (fig. 3, A). Individual cell outlines were indistinct and syncytial masses of variable thickness projected onto the lumen (fig. 3, B). These projections appeared as fringes in some tubules. Cytoplasmic projections from the cells traversed the lumen of some tubules. The basement membrane was absent around the tubules and the tubular lining cells were juxtaposed to similar cells in the stroma. Periodic acid Schiff positive granules and droplets were present in the tubular lining cells as well as in the stromal cells. Aggregates of clear cells were present in the wall of the cyst. Some tubules were irregular spaces lined with flattened or low cuboidal cells (fig. 3, A). They simulated the "macro-folliculoid" pattern of an adenomatoid

Fm. 2. Case 2. A, IVP demonstrates no renal function on left side and displacement of bladder. B, cystogram (lateral view) reveals displacement of bladder. C, barium enema shows displacement of rectum and sigmoid colon. D, photograph of operative findings shows retrovesical cyst and bladder.

109

RETROVESICAL CYSTS IN BOYS

FrG. 3. Case 1. A, photomicrograph shows row of tubular spaces in superficial part of cyst wall; some tubules have adenomatoid appearance. H & E, reduced from X140. B, photomicrograph shows tubular spaces with epithelial fringes; processes from cells traverse lumen and individual cell outline cannot be discerned; one space contains miniature corpora amylacea. H & E, reduced from X165.

Fm. 4. Case 2. A, photomicrograph shows invaginations and projections in cyst wall. H & E, reduced from X300. B, photomicrograph shows gland-like spaces in superficial part and smooth muscle in deeper part of cyst wall. Mallory's stain, reduced from X300. tumor described by Golden and Ash. 1 The stroma contained fibrous connective tissue and smooth muscle, the latter in variable amount in different parts of the cyst wall. In case 2 the lining of the cyst wall was flattened at places and cuboidal elsewhere and contained hyperchromatic vesicular nuclei with nucleoli. The epithelium showed gland-like invaginations and papillary projections that produced a fringed appearance (fig. 4, A). The stroma was fibrous and also contained smooth muscle, which was abundant in the deeper part of the wall and formed interlacing fascicles (fig. 4, B). 1

Golden, A. and Ash, J.E.: Amer. J. Path., 21: 63,

1945.

DISCUSSION

We have found only one report of similar cysts in boys. McKenna and Kieffer described a 3½-year-old boy with dysuria owing to a cyst in the pelvis. 2 At laparotomy a strictly midline cyst 5 cm. in diameter was removed. The peritoneum, bladder and urethra were opened and the vasa deferentia were divided; the ureters were uninvolved. The condition was labeled mullerian duct cyst. 2 McKenna, C. M. and Kieffer, J. H.: Cited by R.R. Landes and C. L. Ransom. 9

110

CHATTERJEE AND SARKAR HISTOGENESIS

Congenital retrovesical cysts in the male subject may originate in the seminal vesicle, prostate, prostatic utricle wolffian vestige or mullerian vestige. These cysts' are easy to distinguish in adults, in whom most of the reported cases occur. However, in children even with clinical examination, radiology, endosco~y, operative findings and histology, it is difficult to distinguish these cysts. Congenital seminal vesicle cysts are small and thin-walled and are rare. They may simulate mullerian duct cysts histologically 3 but the microscopic features of the seminal vesicle are often present. It has been reported that many cysts thought to arise in the seminal vesicle or prostate are in fact cysts of the prostatic utricle; the diagnostic .error is a~tributed to insufficient attention to this embryomc vestige. 4 The embryological origin of the prostatic utricle is unknown. It is believed to arise from the mullerian duct urogenital sinus or both; the consensus is that it arises in the mullerian duct. Mullerian duct cysts of the present type have been described. 2 • s-s Cystic change in the prostatic utricle is the most common mullerian duct cyst observed clinically 9 and at autopsy. 10 • 11 The epithelial lining in our cases showed "fringes" simulating fallopian tube mucosa in miniature. This was especially true in case 2. The appendix testis, a mullerian vestige, has a similar appearance. 4 Smoo.th muscle was present in variable amount and its abundance and arrangement in case 2 simulated the musculature derived from the caudal end of the mullerian mesenchyma in the female subject. The adenomatoid appearance of some tubular spaces in case 1 has been noted in adenomatoid tumors believed to have originated from the mullerian vestige. 4 Miniature corpora amylacea are found in the prostatic utricle.11 In investigations on an 8 mm. human embryo, Gruenwald observed the proximity of mullerian and wolffian ducts and dispensed with the concept of their independent growth. 12 The caudal ends of the wolffian and mullerian ducts are in close proximity with no basal membrane separating their cells. 12 3

Sundarasivarao, D.: J. Path. Bact., 66: 417, 1953. Coppridge, W. M.: Cited ):>YD. Sundarasivarao. 4 s Deming, C. L. and Berne1ke, R.R.: J. Urol., 61:

4 5

563, 1944. 8

Hennessey, R. A.: J. Urol., 42: 1042, 1939. Howard, F. S.: Surg., Gynec. & Obst., 86: 307,

1948. 9

DIAGNOSIS

The presenting symptom in both cases was dysuria and occasional urinary retention. The presenting sign was cystic swelling that simulated a distended bladder. Hence, the first clinical diagnosis was urethral valves. When a urethral catheter failed to reduce the swelling, it was suspected that the catheter had not entered the bladder. However, the diagnosis of retrovesical cyst was clearly established radiologically: IVP and ascending cystography confirmed that a large extravesical cystic swelling displaced the bladder forward, upward and to the opposite side and that renal function was absent on the side of the swelling. Similarly, a barium enema established that the swelling displaced the large gut backward, upward and to the opposite side. TREATMENT

Excision of the cyst produced symptomatic relief, that is micturition and defecation are normal. However, renal function on the side of the lesion has not improved in case 2 and is unlikely to become completely normal in case 1. While renal changes secondary to compression by the cyst could have been prevented or more easily reversed by earlier diagnosis and treatment, it is believed that compression was not the only factor producing a non-functioning kidney in case 2. It is certain that excision could not have been carried out without the sacrifice of the ipsilateral vas deferens. While the contralateral vas was cut in case 1, it was saved in case 2. With increasing awareness of this condition, the vas is likely to be preserved in subsequent cases. In order to save the adjoining genital structures the less radical operation of marsupialization has been suggested13 but the morbidity associated with this operation would not be acceptable to most surgeons. SUMMARY

Stewart, B. L. and Nicoll, G. A.: J. Urol., 62: 189,

1949.

1

Hence, they may give rise to similar cysts in that region. In previously described cases cysts of the prostatic utricle were midline cysts while in our cases they were not. It has been suggested that cysts may arise at any site in the mullerian duct in male subjects where obliteration is not complete. This may explain the unilateral location of the cyst in our cases. 9

Landes, R.R. and Ransom, C. L.: J. Urol., 61:

1089, 1949.

10 Springer, C.: Cited by R. R. Landes and C. L. Ransom. 9 u Moore, R. A.: Arch. Path., 23: 517, 1937. 12 Gruenwald, P.: Anat. Rec., 81: 1, 1941.

Bladder outlet obstruction in boys may sometimes be caused by cystic space-occupying lesions. An apparently distended bladder may prove to be a cyst in the pelvis unconnected with the bladder. Cysts pose a threat to renal function and fertility. Prompt diagnosis and treatment of these cysts improve the possibility of preserving normal renal and genital function. Two children with this condition are described. 13

Moore, V. and Howe, G. E.: J. Urol., 70: 781,

1953.