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Cystic Pelvic Mass
0022-534 7/86/1353-0550$02.00/0
Vol. 135, March Printed in U.S.A.
THE JOURNAL OF UROLOGY
Copyright © 1986 by The Williams & Wilkins Co. ·
Clinicopathological Conference CYSTIC PELVIC MASS A. RICHARD KENDALL,* BARRY S. STEIN,t FRANCIS J. SHEA,+ ROBERT 0. PETERSENII
AND
BRUCE SENAY§ From the Departments of Urology, Radiology and Pathology, Temple University School of Medicine, Philadelphia, Pennsylvania
infertility or impotence may result, or that urinary/fecal diversion may be necessary.
PRESENTATION OF CASE1
Dr. Bruce Senay. A 23-year-old man was hospitalized elsewhere in December 1980 for evaluation of left testicular pain 3 months in duration, at which time the prostate gland was described as being greater than 100 gm. in size. Urinalysis, prostatic acid phosphatase, cystourethroscopy and excretory urography (IVP) all were normal. Because of the symptomatology and the clinically huge prostate, a perineal prostatic biopsy was performed, which revealed normal prostatic tissue. The patient was lost to followup until October 1981, when he returned with left flank and scrotal pain accompanied by nausea and vomiting. A repeat IVP revealed an elevated bladder pushed anteriorly by a grapefruit-sized mass. Computerized tomography (CT) revealed a septated cystic mass arising from the pelvis without any detectable adenopathy. Chest x-ray and a bone scan were negative. Following complete evaluation, needle biopsy of the mass via a perineal and transrectal approach was performed. Little tissue but large amounts of thick fluid compatible with old blood were obtained. Pathological and cytological studies were negative for tumor, and exploration of the pelvis revealed a thick-walled cystic mass between the bladder and rectum. More than 1,000 cc old blood-type fluid were aspirated and multiple biopsies were obtained. During this procedure the right ureter was compromised and reimplanted. Pathological study did not reveal any evidence of malignancy and the patient was discharged from the hospital without symptoms. In March 1982 the patient presented to our hospital with a 3-week history of left flank, hip and leg pain requiring the administration of large amounts of narcotics. At that time there was no history of anorexia, weight loss, or gastrointestinal or genitourinary symptomatology. Physical examination revealed the previous midline incision to be well healed and there was a significant fullness in the lower abdomen. All routine laboratory studies were within normal limits and a CT scan was essentially unchanged from the previous study. Pelvic angiography and a barium enema were performed. The patient adamantly refused total extirpation of the mass because of concerns of impotence and infertility. However, because of the severe pain decompression was attempted by opening of numerous cystic structures, and attempted marsupialization to the anterior abdominal wall in the hope of being able to catheterize the cyst intermittently. Drains and packing were removed during a 6-week interval and the patient remained fairly comfortable until September, when chronic pelvic pain and hourly urinary frequency developed. The condition became intolerable and the patient then consented to a definitive operation in October, recognizing that
RADIOLOGICAL STUDIES
Dr. Francis J. Shea. The initial study at our hospital was a CT examination of the abdomen and retroperitoneum (fig. 1). A 14 X 10 cm. cystic mass in the pelvis displaced the bladder anteriorly. A pelvic arteriogram performed the following day (fig. 2, A) via a right transfemoral approach demonstrated a large soft tissue mass displacing the bladder anteriorly and superiorly. The mass appeared to be supplied by the obturator, pudendal and vesicular arteries arising from the internal iliac vessels. No tumor vessels were identified. An IVP approximately 2 months after the initial CT scan showed a large pelvic mass causing moderate obstruction of both ureters not detected previously (fig. 2, B). A barium enema and cystogram also were performed later in the same day, and a lateral film of the pelvis illustrated significant anterior displacement of the bladder and significant posterior compression of the rectosigmoid (fig. 3, A).
A repeat CT scan of the pelvis approximately 6½ months after the initial CT examination again demonstrated the cystic mass, which had increased slightly in size, and again caused marked anterior displacement of the bladder and posterior displacement of the rectosigmoid (fig. 3, B). An ultrasound examination at the time of ultrasound-guided biopsy 8 months after the initial CT examination also demonstrated a large multiloculated cystic mass in the pelvis. DIFFERENTIAL DIAGNOSIS
Dr. Barry S. Stein. The differential diagnosis in this 23-yearold man presenting with a large prostatic or periprostatic cystic mass includes miillerian duct cyst, cystic dilatation of the utricle, seminal vesicle cyst, cysts of the prostate, sarcoma of
* Professor and Chairman, Department of Urology.
t Associate Professor, Department of Urology. :j: Professor, Department of Radiology. II Clinical Associate Professor, Department of Pathology. § Chief Resident, Department of Urology. 'll Case presented at Urological Radiology Conference, February 14, 1983.
FIG. 1. CT scan shows cystic mass within pelvis displacing bladder anteriorly.
550
fi51
CYST:1C PELVIC l\r1A8S
FIG. 2. A, right transfemoral anteriogram demonstrates mass displacing bladder anteriorly and superiorly. B, IVP shows ureteral obstruction
FIG. 3. A, combination barium enema and cystogram show mass to be anterior to rectosigmoid and posterior to bladder. B, repeat CT examination shows cystic nature of mass and evidence of slight enlargement during 6½ months.
the prostate and seminal vesicle carcinoma or leiomyoma. Most pelvic cysts in male patients are of mullerian duct origin and all are exceedingly rare. If the mi.illerian duct were present in the male patient it would follow the course of the vas deferens and both sides would join at the utricle. Cysts can occur anywhere along the tract but they are reported most commonly in the deep pelvis between the bladder and rectum. The most common presentation of a mullerian duct cyst is in a young man between 20 and 40 years old with signs and symptoms of urinary obstruction, dysuria, hematuria, suprapubic or rectal and/or a lower abdominal mass. Most of these cysts are midline and symmetrical, and appear to arise from the upper margin of the prostate. The fluid in these cysts does not contain sperm and the cyst may be extremely adherent to the posterior prostatic capsule and, thus, may be difficult to excise. Cysts or dilatations of the utricle usually are associated with hypospadias and incomplete testicular descent. Most of these patients present in the first 2 decades of life, earlier than patients with mi.illerian duct cysts. The cysts usually are not adherent to the prostate or rectum and, therefore, are excised easily. Seminal vesicle cysts are rarely evident clinically. Such patients may present with irritative urinary complaints, such as dysuria, frequency and urgency. Pain is rare but has been reported as being pelvic, urethral or testicular in nature. Surprisingly, hematospermia does not appear to be a significant presenting complaint. The cysts usually are monolocular and located on both sides of the midline, and generally are smaller than mullerian duct cysts. The association of an absent kidney
ipsilaterally is helpful in the diagnosis. A further diagnostic differentiation between seminal vesicle and mullerian duct cysts is that seminal vesicle cysts usually contain sperm, while mullerian duct cysts do not. Cysts of the prostate are extremely rare and usually are incidental autopsy findings. When present clinically they are small and to one side of the midline in continuity with the prostate. Sarcoma of the prostate represents less thah 0.1 per cent of all prostatic malignancies. Of these patients 30 per cent present before the first year of life and 75 per cent before they are 40 years old. In children most are rhabdomyosarcomas, whereas leiomyosarcomas and fibrosarcomas are seen frequently in adults. These tumors grow rapidly and may encroach upon the rectum, with symptomatology being either difficulty or change in bowel habits, or urinary tract obstruction. Seminal vesicle carcinoma is exceedingly rare and usually occurs in men more than 50 years old. The lesion usually is more solid, although some have been papillary. Considering the size and location of this mass, plus the age of the patient my clinical diagnosis is mullerian duct cyst. Dr. A. R. Kendall. Doctor Stein, if needle aspiration had revealed sperm, would this have changed your diagnosis? Doctor Stein. Under those circumstances I might be more prone to consider a cyst of the seminal vesicle. OPERATIVE DESCRIPTION
Doctor Kendall. Before definitive exploration, cystoscopic examination revealed the urethra to be normal but the bladder
552
KENDALL AND ASSOCIATES
was pushed markedly anteriorly by a huge palpable pelvic mass making ureteral catheterization impossible. The pelvis was explored through the previous vertical incision and the bladder was freed from multiple perivesical adhesions. Eventually, a plane was able to be achieved with some difficulty between the anterior wall of the pelvic mass and the posterior wall of the bladder. The mass appeared to be markedly adherent to the rectum but it could be separated from this structure because of a thick capsule. The left ureter was involved intimately with the mass and was transected as far distal as possible. The bladder then was delivered anteriorly almost out of the abdomen as dissection was continued distally, and eventually the entire grapefruit-sized mass was excised. The most distal aspect of the mass did not seem to be attached to any structure, and peeled out easily after being freed from the bladder anteriorly and the rectum posteriorly. A small opening in the anterior wall of the rectum was repaired and the left ureter was reimplanted in the usual fashion. Convalescence was relatively uncomplicated and the patient was discharged from the hospital with a Foley catheter in place, which was removed 20 days postoperatively. Doctor Shea. X-rays demonstrated the postoperative findings. A followup IVP obtained approximately 9 months postoperatively revealed satisfactorily functioning upper tracts without significant obstruction, and minimal postoperative deformity of the bladder without any evidence of recurrence of the mass (fig. 4).
sia (fig. 6, inset). The identification of the cystic structures as prostatic in origin is accomplished by observing typical prostatic ductal epithelial cells lining the cysts (fig. 7). Supportive evidence of the prostatic origin of this cystic mass is the positive
PATHOLOGICAL FINDINGS
FIG. 5. Seminal vesicle (specimen 1). Wall of seminal vesicle is composed of fibromuscular tissue with typical papillary configuration of lining mucosa. Lipofuscin is present in cytoplasm of seminal vesicle epithelium. H & E, reduced from X63.
Dr. R. 0. Petersen. Specimen 1 was the biopsy obtained at the initial exploration. All 4 specimens have identical histological features and are interpreted as portions of normal seminal vesicles (fig. 5). There is no evidence of cystic enlargement in the sections available for review. The characteristic feature of lipofuscin pigment accumulation is observed in the cytoplasm of the seminal vesicle epithelium. This epithelium shows numerous typical bridging papillae with interspersed smaller papillary projections. The lining epithelium is cuboidal to low columnar and cell borders a:re indistinct. The surrounding stroma is composed of smooth muscle in interweaving bundles characteristic of the seminal vesicle. Specimens 2 and 3 were obtained from the mass excised in October 1982. The slides from both specimens show an essentially identical histological picture. They contain fragments of prostatic tissue. The prostatic ductal tributaries show variable dilatation with innumerable stellate-shaped ducts interspersed between enlarged smooth-contoured, cystically dilated ducts (fig. 6). The lining ductal mucosa shows a spectrum of epithelial alterations. The ductal cells are flat to low columnar and mucous secreting, or show clear evidence of squamous metapla-
FIG. 6. Prostate (specimen 2). Duct lining epithelium shows variability including foci of squamous metaplasia (inset). H & Estain.
FIG. 4. Followup studies 9 months postoperatively. A, IVP shows resolution of previously described obstruction of upper tracts. B, excretory cystogram reveals minimal deformity of bladder and no evidence of mass.
553
CYSTIC PELVIC MASS
immunoperoxidase staining for prostatic specific antigen (fig. 8). No cystic structures lined by seminal vesicle epithelium are identified in any of the slides available for review. The stroma intervening between the dilated ducts also shows noteworthy changes. Condensed collagenous fibrous tissue with minimal evidence of smooth muscle surrounds the largest cysts (fig. 7). This tissue makes a gradual transition to the typical fibromuscular stroma of the prostate. In addition, numerous stellate-shaped ducts appear to have an enveloping edematous, hypocellular, primitive stroma somewhat reminiscent of the breast lesion, cystosarcoma phyllodes (fig. 9). This histological appearance is present in only focal locations. The differential diagnosis of cystic lesions in this region in the male pelvis includes mullerian duct cyst, seminal vesicle cyst, prostatic cyst and cyst development in hyperplasia in order of decreasing frequency. 1- 12 The histological features supported by the immunoperoxidase staining identify the lesion in this case as prostatic in origin. Referable to prostatic lesions, 2 diagnostic possibilities were considered, namely a cystic hamartomatous proliferation and a phyllodes-type hyperplastic proliferation of the prostate with cystic change. The relatively young age of the patient, cystic structure, and unusual stromal and ductal proliferative pattern supported the former diagnostic possibility. However, I could find no documented precedent in the literature or reference to such a lesion in the available textbooks on urological pathological conditions. Support for the alternative diagnostic possibility rested on 2 cases reported in the literature. In 1976 Attah and Nkposong described a phyllodes-like hyperplasia in the prostate of a 31year-old man. 13 The histological features of the ductal and
FIG. 9. Prostate (specimen 3). Periductal stroma shows mild edema and relative hypocellularity compared to adjacent stroma of prostate gland. Ducts are dilated and irregular in outline. Focal ductal hyperplasia is present. H & E stain.
stromal proliferation reported by these investigators bear a striking resemblance to our case. However, the lesion was not cystic as in this case. The following year Attah and Powell reported 3 cases of prostatic hyperplasia with atypical stromal proliferation. 14 Case 3 in that report also is similar to our case. I finally concluded that the lesion represented a cystic hamartoma of the prostate despite the absence of recorded precedent. Dr. F. Kash Mostofi, at the Armed Forces Institute of Pathology, reviewed the case and indicated that he previously had studied a similar case, also in a young man, and believed that both represented examples of a phyllodes-type variant ofprostatic hyperplasia. The previous case to which he referred had multiple local recurrences of the lesion. The cystic component of our case was regarded as uncommon but it has been observed in hyperplasia and is independent of the phyllodes pattern. I deferred to his diagnosis. FINAL DIAGNOSIS
Phyllodes-type variant of prostatic hyperplasia. Doctor Kendall. The patient has been followed for 2½ years. There is no evidence of recurrence and he is entirely asymptomatic except for lack of ejaculate. Urinalysis following sexual activity has failed to reveal sperm on several occasions but there is no erectile dysfunction. REFERENCES
FIG. 7. Prostate (specimen 3). Prostatic cyst wall with lining epithelium similar to that present in prostatic ducts.
FIG. 8. Prostate (specimen 3). Immunoperoxidase staining for prostatic specific antigen in prostate cyst wall epithelium. H & E counterstain.
1. Emmett, J. L. and Braasch, W. F.: Cysts of the prostate gland. J. Urol., 36: 236, 1936. 2. Dees, J. E.: Congenital cyst of prostate communicating with right vas and rudimentary right ureter: case report. J. Urol., 57: 304, 1947. 3. Rieser, C. and Griffin, T. L.: Cysts of the prostate. J. Urol., 91: 282, 1964. 4. Kirkland, K. L. and Bale, P. M.: A cystic adenoma of the prostate. J. Urol., 97: 324, 1967. 5. Culbertson, L. R.: Mullerian duct cyst. J. Urol., 58: 134, 1947. 6. Moore, V. and Howe, G. E.: Miillerian duct remnants in the male. J. Urol., 70: 781, 1953.
7. Schuhrke, T. D. and Kaplan, G. W.: Prostatic utricle cysts (miillerian duct cysts). J. Urol., 119: 765, 1978. 8. Eickhoff, J. H.: Miillerian duct cyst. Report of a case and review of the literature. Scand. J. Urol. Nephrol., 12: 89, 1978. 9. Hart, J. B.: A case of cyst or hydrops of the seminal vesicle. J. Urol., 86: 137, 1961. 10. Heetderks, D. R., Jr. and Delambre, L. C.: Cyst of the seminal vesicle. J. Urol., 93: 725, 1965. 11. Reddy, Y. N. and Winter, C. C.: Cyst of the seminal vesicle: a case report and review of the literature. J. Urol., 108: 134, 1972. 12. Ogreid, P. and Hatteland, K.: Cyst of seminal vesicle associated with ipsilateral renal agenesis. A report on four cases. Scand. J. Urol. Nephrol., 13: 113, 1979. 13. Attah, E. B. and Nkposong, E. 0.: Phyllodes type of atypical prostatic hyperplasia. J. Urol., 115: 762, 1976. 14. Attah, E. B. and Powell, M. E. A.: Atypical stromal hyperplasia of the prostate gland. Amer. J. Clin. Path., 67: 324, 1977.
Vol. 135, March Printed in U.S.A.
THE JOURNAL OF UROLOGY
Copyright © 1986 by The Williams & Wilkins Co. ·
Clinicopathological Conference CYSTIC PELVIC MASS A. RICHARD KENDALL,* BARRY S. STEIN,t FRANCIS J. SHEA,+ ROBERT 0. PETERSENII
AND
BRUCE SENAY§ From the Departments of Urology, Radiology and Pathology, Temple University School of Medicine, Philadelphia, Pennsylvania
infertility or impotence may result, or that urinary/fecal diversion may be necessary.
PRESENTATION OF CASE1
Dr. Bruce Senay. A 23-year-old man was hospitalized elsewhere in December 1980 for evaluation of left testicular pain 3 months in duration, at which time the prostate gland was described as being greater than 100 gm. in size. Urinalysis, prostatic acid phosphatase, cystourethroscopy and excretory urography (IVP) all were normal. Because of the symptomatology and the clinically huge prostate, a perineal prostatic biopsy was performed, which revealed normal prostatic tissue. The patient was lost to followup until October 1981, when he returned with left flank and scrotal pain accompanied by nausea and vomiting. A repeat IVP revealed an elevated bladder pushed anteriorly by a grapefruit-sized mass. Computerized tomography (CT) revealed a septated cystic mass arising from the pelvis without any detectable adenopathy. Chest x-ray and a bone scan were negative. Following complete evaluation, needle biopsy of the mass via a perineal and transrectal approach was performed. Little tissue but large amounts of thick fluid compatible with old blood were obtained. Pathological and cytological studies were negative for tumor, and exploration of the pelvis revealed a thick-walled cystic mass between the bladder and rectum. More than 1,000 cc old blood-type fluid were aspirated and multiple biopsies were obtained. During this procedure the right ureter was compromised and reimplanted. Pathological study did not reveal any evidence of malignancy and the patient was discharged from the hospital without symptoms. In March 1982 the patient presented to our hospital with a 3-week history of left flank, hip and leg pain requiring the administration of large amounts of narcotics. At that time there was no history of anorexia, weight loss, or gastrointestinal or genitourinary symptomatology. Physical examination revealed the previous midline incision to be well healed and there was a significant fullness in the lower abdomen. All routine laboratory studies were within normal limits and a CT scan was essentially unchanged from the previous study. Pelvic angiography and a barium enema were performed. The patient adamantly refused total extirpation of the mass because of concerns of impotence and infertility. However, because of the severe pain decompression was attempted by opening of numerous cystic structures, and attempted marsupialization to the anterior abdominal wall in the hope of being able to catheterize the cyst intermittently. Drains and packing were removed during a 6-week interval and the patient remained fairly comfortable until September, when chronic pelvic pain and hourly urinary frequency developed. The condition became intolerable and the patient then consented to a definitive operation in October, recognizing that
RADIOLOGICAL STUDIES
Dr. Francis J. Shea. The initial study at our hospital was a CT examination of the abdomen and retroperitoneum (fig. 1). A 14 X 10 cm. cystic mass in the pelvis displaced the bladder anteriorly. A pelvic arteriogram performed the following day (fig. 2, A) via a right transfemoral approach demonstrated a large soft tissue mass displacing the bladder anteriorly and superiorly. The mass appeared to be supplied by the obturator, pudendal and vesicular arteries arising from the internal iliac vessels. No tumor vessels were identified. An IVP approximately 2 months after the initial CT scan showed a large pelvic mass causing moderate obstruction of both ureters not detected previously (fig. 2, B). A barium enema and cystogram also were performed later in the same day, and a lateral film of the pelvis illustrated significant anterior displacement of the bladder and significant posterior compression of the rectosigmoid (fig. 3, A).
A repeat CT scan of the pelvis approximately 6½ months after the initial CT examination again demonstrated the cystic mass, which had increased slightly in size, and again caused marked anterior displacement of the bladder and posterior displacement of the rectosigmoid (fig. 3, B). An ultrasound examination at the time of ultrasound-guided biopsy 8 months after the initial CT examination also demonstrated a large multiloculated cystic mass in the pelvis. DIFFERENTIAL DIAGNOSIS
Dr. Barry S. Stein. The differential diagnosis in this 23-yearold man presenting with a large prostatic or periprostatic cystic mass includes miillerian duct cyst, cystic dilatation of the utricle, seminal vesicle cyst, cysts of the prostate, sarcoma of
* Professor and Chairman, Department of Urology.
t Associate Professor, Department of Urology. :j: Professor, Department of Radiology. II Clinical Associate Professor, Department of Pathology. § Chief Resident, Department of Urology. 'll Case presented at Urological Radiology Conference, February 14, 1983.
FIG. 1. CT scan shows cystic mass within pelvis displacing bladder anteriorly.
550
fi51
CYST:1C PELVIC l\r1A8S
FIG. 2. A, right transfemoral anteriogram demonstrates mass displacing bladder anteriorly and superiorly. B, IVP shows ureteral obstruction
FIG. 3. A, combination barium enema and cystogram show mass to be anterior to rectosigmoid and posterior to bladder. B, repeat CT examination shows cystic nature of mass and evidence of slight enlargement during 6½ months.
the prostate and seminal vesicle carcinoma or leiomyoma. Most pelvic cysts in male patients are of mullerian duct origin and all are exceedingly rare. If the mi.illerian duct were present in the male patient it would follow the course of the vas deferens and both sides would join at the utricle. Cysts can occur anywhere along the tract but they are reported most commonly in the deep pelvis between the bladder and rectum. The most common presentation of a mullerian duct cyst is in a young man between 20 and 40 years old with signs and symptoms of urinary obstruction, dysuria, hematuria, suprapubic or rectal and/or a lower abdominal mass. Most of these cysts are midline and symmetrical, and appear to arise from the upper margin of the prostate. The fluid in these cysts does not contain sperm and the cyst may be extremely adherent to the posterior prostatic capsule and, thus, may be difficult to excise. Cysts or dilatations of the utricle usually are associated with hypospadias and incomplete testicular descent. Most of these patients present in the first 2 decades of life, earlier than patients with mi.illerian duct cysts. The cysts usually are not adherent to the prostate or rectum and, therefore, are excised easily. Seminal vesicle cysts are rarely evident clinically. Such patients may present with irritative urinary complaints, such as dysuria, frequency and urgency. Pain is rare but has been reported as being pelvic, urethral or testicular in nature. Surprisingly, hematospermia does not appear to be a significant presenting complaint. The cysts usually are monolocular and located on both sides of the midline, and generally are smaller than mullerian duct cysts. The association of an absent kidney
ipsilaterally is helpful in the diagnosis. A further diagnostic differentiation between seminal vesicle and mullerian duct cysts is that seminal vesicle cysts usually contain sperm, while mullerian duct cysts do not. Cysts of the prostate are extremely rare and usually are incidental autopsy findings. When present clinically they are small and to one side of the midline in continuity with the prostate. Sarcoma of the prostate represents less thah 0.1 per cent of all prostatic malignancies. Of these patients 30 per cent present before the first year of life and 75 per cent before they are 40 years old. In children most are rhabdomyosarcomas, whereas leiomyosarcomas and fibrosarcomas are seen frequently in adults. These tumors grow rapidly and may encroach upon the rectum, with symptomatology being either difficulty or change in bowel habits, or urinary tract obstruction. Seminal vesicle carcinoma is exceedingly rare and usually occurs in men more than 50 years old. The lesion usually is more solid, although some have been papillary. Considering the size and location of this mass, plus the age of the patient my clinical diagnosis is mullerian duct cyst. Dr. A. R. Kendall. Doctor Stein, if needle aspiration had revealed sperm, would this have changed your diagnosis? Doctor Stein. Under those circumstances I might be more prone to consider a cyst of the seminal vesicle. OPERATIVE DESCRIPTION
Doctor Kendall. Before definitive exploration, cystoscopic examination revealed the urethra to be normal but the bladder
552
KENDALL AND ASSOCIATES
was pushed markedly anteriorly by a huge palpable pelvic mass making ureteral catheterization impossible. The pelvis was explored through the previous vertical incision and the bladder was freed from multiple perivesical adhesions. Eventually, a plane was able to be achieved with some difficulty between the anterior wall of the pelvic mass and the posterior wall of the bladder. The mass appeared to be markedly adherent to the rectum but it could be separated from this structure because of a thick capsule. The left ureter was involved intimately with the mass and was transected as far distal as possible. The bladder then was delivered anteriorly almost out of the abdomen as dissection was continued distally, and eventually the entire grapefruit-sized mass was excised. The most distal aspect of the mass did not seem to be attached to any structure, and peeled out easily after being freed from the bladder anteriorly and the rectum posteriorly. A small opening in the anterior wall of the rectum was repaired and the left ureter was reimplanted in the usual fashion. Convalescence was relatively uncomplicated and the patient was discharged from the hospital with a Foley catheter in place, which was removed 20 days postoperatively. Doctor Shea. X-rays demonstrated the postoperative findings. A followup IVP obtained approximately 9 months postoperatively revealed satisfactorily functioning upper tracts without significant obstruction, and minimal postoperative deformity of the bladder without any evidence of recurrence of the mass (fig. 4).
sia (fig. 6, inset). The identification of the cystic structures as prostatic in origin is accomplished by observing typical prostatic ductal epithelial cells lining the cysts (fig. 7). Supportive evidence of the prostatic origin of this cystic mass is the positive
PATHOLOGICAL FINDINGS
FIG. 5. Seminal vesicle (specimen 1). Wall of seminal vesicle is composed of fibromuscular tissue with typical papillary configuration of lining mucosa. Lipofuscin is present in cytoplasm of seminal vesicle epithelium. H & E, reduced from X63.
Dr. R. 0. Petersen. Specimen 1 was the biopsy obtained at the initial exploration. All 4 specimens have identical histological features and are interpreted as portions of normal seminal vesicles (fig. 5). There is no evidence of cystic enlargement in the sections available for review. The characteristic feature of lipofuscin pigment accumulation is observed in the cytoplasm of the seminal vesicle epithelium. This epithelium shows numerous typical bridging papillae with interspersed smaller papillary projections. The lining epithelium is cuboidal to low columnar and cell borders a:re indistinct. The surrounding stroma is composed of smooth muscle in interweaving bundles characteristic of the seminal vesicle. Specimens 2 and 3 were obtained from the mass excised in October 1982. The slides from both specimens show an essentially identical histological picture. They contain fragments of prostatic tissue. The prostatic ductal tributaries show variable dilatation with innumerable stellate-shaped ducts interspersed between enlarged smooth-contoured, cystically dilated ducts (fig. 6). The lining ductal mucosa shows a spectrum of epithelial alterations. The ductal cells are flat to low columnar and mucous secreting, or show clear evidence of squamous metapla-
FIG. 6. Prostate (specimen 2). Duct lining epithelium shows variability including foci of squamous metaplasia (inset). H & Estain.
FIG. 4. Followup studies 9 months postoperatively. A, IVP shows resolution of previously described obstruction of upper tracts. B, excretory cystogram reveals minimal deformity of bladder and no evidence of mass.
553
CYSTIC PELVIC MASS
immunoperoxidase staining for prostatic specific antigen (fig. 8). No cystic structures lined by seminal vesicle epithelium are identified in any of the slides available for review. The stroma intervening between the dilated ducts also shows noteworthy changes. Condensed collagenous fibrous tissue with minimal evidence of smooth muscle surrounds the largest cysts (fig. 7). This tissue makes a gradual transition to the typical fibromuscular stroma of the prostate. In addition, numerous stellate-shaped ducts appear to have an enveloping edematous, hypocellular, primitive stroma somewhat reminiscent of the breast lesion, cystosarcoma phyllodes (fig. 9). This histological appearance is present in only focal locations. The differential diagnosis of cystic lesions in this region in the male pelvis includes mullerian duct cyst, seminal vesicle cyst, prostatic cyst and cyst development in hyperplasia in order of decreasing frequency. 1- 12 The histological features supported by the immunoperoxidase staining identify the lesion in this case as prostatic in origin. Referable to prostatic lesions, 2 diagnostic possibilities were considered, namely a cystic hamartomatous proliferation and a phyllodes-type hyperplastic proliferation of the prostate with cystic change. The relatively young age of the patient, cystic structure, and unusual stromal and ductal proliferative pattern supported the former diagnostic possibility. However, I could find no documented precedent in the literature or reference to such a lesion in the available textbooks on urological pathological conditions. Support for the alternative diagnostic possibility rested on 2 cases reported in the literature. In 1976 Attah and Nkposong described a phyllodes-like hyperplasia in the prostate of a 31year-old man. 13 The histological features of the ductal and
FIG. 9. Prostate (specimen 3). Periductal stroma shows mild edema and relative hypocellularity compared to adjacent stroma of prostate gland. Ducts are dilated and irregular in outline. Focal ductal hyperplasia is present. H & E stain.
stromal proliferation reported by these investigators bear a striking resemblance to our case. However, the lesion was not cystic as in this case. The following year Attah and Powell reported 3 cases of prostatic hyperplasia with atypical stromal proliferation. 14 Case 3 in that report also is similar to our case. I finally concluded that the lesion represented a cystic hamartoma of the prostate despite the absence of recorded precedent. Dr. F. Kash Mostofi, at the Armed Forces Institute of Pathology, reviewed the case and indicated that he previously had studied a similar case, also in a young man, and believed that both represented examples of a phyllodes-type variant ofprostatic hyperplasia. The previous case to which he referred had multiple local recurrences of the lesion. The cystic component of our case was regarded as uncommon but it has been observed in hyperplasia and is independent of the phyllodes pattern. I deferred to his diagnosis. FINAL DIAGNOSIS
Phyllodes-type variant of prostatic hyperplasia. Doctor Kendall. The patient has been followed for 2½ years. There is no evidence of recurrence and he is entirely asymptomatic except for lack of ejaculate. Urinalysis following sexual activity has failed to reveal sperm on several occasions but there is no erectile dysfunction. REFERENCES
FIG. 7. Prostate (specimen 3). Prostatic cyst wall with lining epithelium similar to that present in prostatic ducts.
FIG. 8. Prostate (specimen 3). Immunoperoxidase staining for prostatic specific antigen in prostate cyst wall epithelium. H & E counterstain.
1. Emmett, J. L. and Braasch, W. F.: Cysts of the prostate gland. J. Urol., 36: 236, 1936. 2. Dees, J. E.: Congenital cyst of prostate communicating with right vas and rudimentary right ureter: case report. J. Urol., 57: 304, 1947. 3. Rieser, C. and Griffin, T. L.: Cysts of the prostate. J. Urol., 91: 282, 1964. 4. Kirkland, K. L. and Bale, P. M.: A cystic adenoma of the prostate. J. Urol., 97: 324, 1967. 5. Culbertson, L. R.: Mullerian duct cyst. J. Urol., 58: 134, 1947. 6. Moore, V. and Howe, G. E.: Miillerian duct remnants in the male. J. Urol., 70: 781, 1953.
7. Schuhrke, T. D. and Kaplan, G. W.: Prostatic utricle cysts (miillerian duct cysts). J. Urol., 119: 765, 1978. 8. Eickhoff, J. H.: Miillerian duct cyst. Report of a case and review of the literature. Scand. J. Urol. Nephrol., 12: 89, 1978. 9. Hart, J. B.: A case of cyst or hydrops of the seminal vesicle. J. Urol., 86: 137, 1961. 10. Heetderks, D. R., Jr. and Delambre, L. C.: Cyst of the seminal vesicle. J. Urol., 93: 725, 1965. 11. Reddy, Y. N. and Winter, C. C.: Cyst of the seminal vesicle: a case report and review of the literature. J. Urol., 108: 134, 1972. 12. Ogreid, P. and Hatteland, K.: Cyst of seminal vesicle associated with ipsilateral renal agenesis. A report on four cases. Scand. J. Urol. Nephrol., 13: 113, 1979. 13. Attah, E. B. and Nkposong, E. 0.: Phyllodes type of atypical prostatic hyperplasia. J. Urol., 115: 762, 1976. 14. Attah, E. B. and Powell, M. E. A.: Atypical stromal hyperplasia of the prostate gland. Amer. J. Clin. Path., 67: 324, 1977.