YU AND ASSOCIATES
the right side. The entire right hemiscrotum, including the arteriovenous malformation, was excised painstakingly by the technique of incising the skin and dermis for a short segment, placing the hemostats on either side of the incision and dividing between them. Three large arterial feeding vessels (1 cm. in diameter) were encountered, and they were doubly ligated and divided. The testis and spermatic cord were entirely free of the arteriovenous malformation and separated easily from it (fig. 3). After division of all 3 feeding vessels the skin bleeding slowed dramatically and meticulous hemostasis was achieved. A Jackson-Pratt drain was placed in the depth of the wound. The left scrotal skin was brought over to cover the right testis and spermatic cord. Because of the looseness of the left scrotal skin no skin grafts were necessary. The scrotal skin was reap Pl'O�_Illated with 2:Zfil'_Oan_d 3-zero p_olyglactin sutures. At the end of the procedure, over-all, a good cosmetic result was obtained.
malformations who presented with impotence presumably sec ondary to arterial steal phenomena. Interestingly, even though our patient had a significant contribution to the malformation by the left internal iliac artery he did not complain of erectile dysfunction. Because of the fear of inducing impotence we elected not to embolize the feeder from the left internal iliac artery. The surgeon who embarks upon the task of excising an arteriovenous malformation should be prepared to encounter profuse bleeding. The blood vessels in the skin should be clamped before cutting through them, as described. Ligation of feeding vessels seems to slow the bleeding significantly. If the feeding vessels are embolized as a prelude to surgical excision, then an operation should follow soon before the collateral vessels open. We suggest that the possibility of a scrotal arte riov_enous_malformation-�nd----hemangiomaS-should-be-entei, tained in cases of unilateral scrotal swelling.
1. Laurian, C., Leclef, Y., Gigou, F., Alzaoubi, I., Riche, M.-C., Melki, J.-P. and Cormier, J.-M.: Pelvic arteriovenous fistulas: therapeu tic strategy in five cases. Ann. Vase. Surg., 4: 1, 1990. 2. Dhabuwala, C. B., Kumar, A. B., Kerkar, P. D., Bhutawala, A. and Pierce, J.: Patterns of Doppler recordings and its relationship to varicocele in infertile men. Int. J. Androl., 12: 430, 1989. 3. Guz, B. V., Ziegelbaum, M. and Pontes, J. E.: Arteriovenous mal formation of spermatic cord. Urology, 33: 427, 1989. 4. Bezirdjian, D.R., Reznikov, P. D. and Tisnado, J.: Transcatheter embolization of an arteriovenous malformation of the scrotum. Cardiovasc. Intervent. Rad., 12: 267, 1989. 5. Zorgniotti, A. W., Shaw, W. W., Padula, G. and Rossi, G.: Impo tence associated with pudenda! arteriovenous malformation. J. Urol., 132: 128, 1984. 6. Khoury, M. B. and Contractor, F. M.: Impotence caused by trau matic pelvic arteriovenous communication-a case report. An giology, 39: 849, 1988.
Arteriovenous fistulas involving the pelvis have been re ported by several physicians. 1 However, the majority of these cases are traumatic in origin. Congenital arteriovenous malfor mations involving the spermatic vessels have also been re ported.3 To date there is only 1 report of arteriovenous malfor mations involving the scrotal skin and underlying soft tissue.4 Extreme pain, ulceration and bleeding made an operation im perative. An interesting feature of this malformation was the extensive and varied arterial supply, which presented a unique management challenge. The preoperative embolization of 2 feeding arteries failed to alleviate the symptoms of the patient for any significant interval. Zorgniotti et al,5 and Khoury and Contractor6 have inde pendently reported on patients with pudenda! arteriovenous
0022-5347/92/1471-0162$03.00/0 THE JOURNAL OF UROLOGY Copyright© 1992 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Vol. 147, 162-165, January 1992 Printed in U.S.A.
PAPILLARY CYSTADENOCARCINOMA OF THE EPIDIDYMIS: A CASE REPORT AND REVIEW OF THE LITERATURE CHIA-CHENG YU, JONG-KHING HUANG,* HUNG CHIANG, MING-TSUN CHEN LUKE S. CHANG
From the Division of Urology, Department of Surgery, Department of Pathology, Veterans General Hospital-Kaohsiung and Taipei, and National Yang-Ming Medical College, Taipei, Taiwan, Republic of China
Primary epididymal carcinoma is rare. We report a case of papillary cystadenocarcinoma of the epididymis presenting with a painful scrotal mass. The clinical course and histopathological features are reviewed. KEY WORDS: adenocarcinoma, papillary; cystadenocarcinoma; epididymis
Tumors of the epididymis are rare, comprising only 5% of the intrascrotal neoplasms. 1 Of 341 tumors of the epididymis reported by Beccia et al 75% were benign and 25% were malignant.2 According to Mostofi and Price adenomatoid tu mors are the most frequent of the paratesticular structures and correspond to 32% of all tumors involving these tissues.3 We report on a patient with a painful scrotal mass. Pathological Accepted for publication May 17, 1991. * Requests for reprints: 386 Ta-chung 1st Rd., Kaohsiung, Taiwan 813, Republic of China.
examination revealed papillary cystadenocarcinoma of the ep ididymis with right testicular invasion and right paraureteral metastasis causing nonfunction of the right kidney. CASE REPORT
A 27-year-old single man was hospitalized with a painful right scrotal swelling 1 year in duration. The patient had noticed unusual enlargement of the right scrotum for 15 years with acute exacerbation within the last 3 years. Intermittent pain was present for 1 year. No previous medical or surgical
PAPILLARY CYSTADENOCARCINOMA OF EPIDIDYl\HS
problems were noted. The patient also denied a history of testicular maldescent, infection or trauma. Physical examination was unremarkable except for a cystic hard right scrotal mass measuring 14 X 13 X 8 cm. with nodulation and tenderness (fig. 1). The mass did not transmit light. The left testis was normal. No inguinal lymphadenopathy was palpated. Serum carcinoembryonic antigen, /3-human cho rionic gonadotropin and a-fetoprotein levels were normal, as were the complete blood count, SMAC test, urinalysis and chest radiography. Tuberculosis smear, culture and cytological ex amination of the scrotal aspirate and urine were negative. Excretory urography revealed nonvisualization of the right kidney. Cystoscopic retrograde pyelography demonstrated com plete obstruction near the lower portion of the upper third of the right ureter (fig. 2). Ultrasound and computerized tomog raphy (CT) showed marked hydroureteronephrosis of the right upper collecting system and multiloculated cystic deformity of the right scrotum (fig. 3). The comprehensive renal function test results were right kidney O ml. per minute (normal greater than 225 ml. per minute), left kidney 307.7 ml. per minute and excretion index 1.09 (normal 0.84 to 1.12). No other distant metastases were demonstrated by negative bone, liver and spleen scans. The patient refused exploration of the right ureteral lesion initially. Right orchiectomy was performed through an inguinal approach with scrotal extension because of the huge scrotal mass, the expectation of severe adhesions within the scrotum and a great possibility of right ureteral metastasis. The entire scrotal mass was composed of multiloculated cystic structures containing a copious amount of brownish fluid. Multiple cau liflower papillary tumors were visible on the right epididymis adjacent to the testis. The cyst walls were irregular (5 to 6 mm. thick) and exuberant papillary excrescences were present. The left testis was grossly intact by palpation. Microscopically, the cyst walls were lined with a variable pattern of cuboidal or columnar epithelial cells. In most areas the tumor consisted of an irregular papillary structure composed of a rather uniform cuboidal epithelial lining with large vesicular nuclei and coarse nucleoli, displaying atypia and occasional mitotic figures. Strama! invasion was also demon strated (fig. 4, A and B). Sections of the cyst showed evidence of inflammatory reaction, hemosiderin-containing macro phages and fibrosis. Psammoma bodies were conspicuous at many foci (fig. 4, C). Invasion to the adjacent testis and tunica vaginalis was found. Most of the seminiferous tubules near the cystic tumor were atrophic and hyalinized. The lesion extended to the efferent ductules and was believed to be papillary cystadenocarcinoma with testicular invasion arising from the epididymis. Adjuvant chemotherapy with cisplatin, etoposide and bleo mycin was given for 6 courseso Followup chest roentgenography, whole abdominal CT scan and a bone scan were all within normal limits except for a previous right ureteral lesion. Ret roperitoneaJ. exploration 6 months later revealed an induration at the right paraureteral area resulting in ureteral stricture and proximal hydroureteronephrosis. Segmental resection of the ureter with end-to-end anastomosis was done. Pathological examination revealed metastatic adenocarcinoma similar to the epididymal tumor (fig. 4, D). Postoperative local irradiation with 5,000 rad was given. The patient was free of disease after 2 years of followup.
FIG. 1. Gross huge scrotal mass. Arrowhead shows site of needle aspiration.
The epididymis is rarely the seat of a neoplasm. Benign tumors predominate over malignant growths in the proportion of 3:1. The most common of these lesions are the adenomatoid tumors, which account for approximately a third of all epi didymal tumors. Sarcomas are the most common primary ma lignant tumors in the epididymis, followed in descending order
FIG. 2. Retrograde pyelogram reveals complete obstruction near lower portion of right upper third of ureter (arrow).
YU AND ASSOCIATES
Fm. 3. A, multiloculated cystic deformity ofright scrotum with cauliflower papillary protrusions within it.Band C, arrow indicates induration over right paraureter, which resulted in ureteral stricture and marked dilatation of right renal pelvis (P).
Fm. 4. A, papillary structure of epididymal tumor. B, conspicuous stromal invasion (arrows). C, prominent psammoma bodies (arrows). D, nests of papillary tumor (arrows) infiltrating paraureteral soft tissue. L, ureteral lumen. A and B, H & E, reduced from X264. C and D, H & E, reduced from X132.
of frequency by carcinoma, seminoma and teratoma.4 Met astatic tumors of the epididymis are even more uncommon.5• 6 According to Salm, 14 cases of epididymal carcinoma were reported with several different histological types: solid trabec ular in 4, squamoid in 2, columnar cell in 1, anaplastic in 1, adenocarcinoma in 4 and papillary adenocarcinoma in 2.4 A slight left preponderance was observed (left-to-right side ratio 8:6). The majority of the tumors measured less than 5 cm. in diameter. Patient age ranged from 22 to 78 years, with an average age of 47 years. Duration of symptoms varied from a few weeks to several months. In 6 cases a hydrocele was associated with the growth. The prognosis has been invariably poor with the majority of patients dying within 18 months. Broth et al reviewed 20 epididymal carcinomas with poor his tological documentation. 7 An extensive survey of the literature was done by Beccia et al.2 The results were similar to those of Broth et al. 7 Approxi mately a fourth of the epididymal tumors were malignant. They suggested that when one is confronted with a painless epi-
didymal mass one should orient clinical management according to the age of the patient. Any child with a nontesticular mass should undergo inguinal exploration because of the high inci dence of paratesticular rhabdomyosarcoma. Since people more than 50 years old were likely to have either a benign lesion, such as adenomatoid tumor, or a metastasis from another primary site in which cure was not achievable, scrotal explo ration will be sufficient. In the 20 to 50-year-old age group, in which malignancy is common, inguinal exploration should be done unless the tumor is proved to be benign. According to the long history of our patient, low grade malignancy with slow progression or malignant transformation from the benign papillary cystadenoma seems to be reasonable. It is commonly believed that if malignancy is found on the epididymal mass, radical inguinal epididymo-orchiectomy should be the treatment of choice followed by retroperitoneal lymph node dissection and adjuvant chemotherapy or radio therapy.2 Early and aggressive surgical intervention appears to result in the best prognosis. In view of the small numbers
ECTOPIC PROSTATIC TISSUE OF ANAL CANAL
observed, the effect of chemotherapy or irradiation cannot be evaluated to date. Long-term foliowup is necessary. REFERENCES 1. Elsasser, E.: Tumors of the epididymis. Recent Results fcancer Res., 60: 163, 1977. 2. Beccia, D. J., Krane, R. J. and Olsson, C. A.: Clinical management of non-testicular intrascrotal tumors. J. Urol., 116: 476, 1976. 3. Mostofi, F. K. and Price, E. B., Jr.: Tumors of the male genital system. In: Atlas of Tumor Pathology. Washington, D. C.: Armed
Forces Institute of 2nd series, fasc. 8, p. 151, 1973. 4. Salm, R.: Papillary carcinoma the epididymis. J. Path., 97: 253, 1969. 5. Powell, B. L., Craig, J.B. and Muss, H.B.: Secondary malignancies of the penis and epididymis: a case report and review of the literature. J. Clin. Oncol., 3: 110, 1985. 6. Willis, R. A.: The Spread of Tumors in the Human Body, 3rd ed. London: Butterworths & Co. Ltd., pp. 284-285, 1973. 7. Broth, G., Bullock, W. K. and Marrow, J.: Epididymal tumors: 1. Report of 15 new cases including review of literature. 2. Histo chemical study of the so-called adenomatoid tumor. J. Urol., 100: 530, 1968.
0022-5347/92/1471-0165$03.00/0 THE JOURNAL OF UROLOGY Copyright© 1992 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Vol. 147, 165-166, January 1992 Printed in U.S.A.
ECTOPIC PROSTATIC TISSUE OF THE ANAL CANAL MICHAEL B. MORGAN* From the Departments of Pathology, University of South Florida, College of Medicine and James A. Haley Veterans Hospital, Tampa, Florida
A unique case is reported of ectopic prostatic tissue present within the anal submucosa from a routine ischial decubitus resection specimen. Although ectopic prostatic tissue has been reported at loci adjoining the prostate, most notably within the urethra, this case is noteworthy by merit of its disparate location. KEY WORDS: prostate, ectopic tissue, choristoma, anus
Prostatic heterotopia has been previously described to occur in the urethra and at adjoining locations. A unique case of prostatic tissue occurring within the anal submucosa is re ported. CASE REPORT
A 65-year-old white man with a history of paraplegia second ary to a motor vehicle accident presented for care of an ischial decubitus pressure sore. The patient had no history of urological manipulation, surgery or malignancy. At examination a grade 4, 2 x 2 cm. decubitus was found and localized surgical debride ment was done. Representative sections were fixed in 10% neutral buffered formaldehyde solution. Tissue blocks were processed in paraffin, and sections were stained with hematox ylin and eosin. Immunohistochemical studies were performed with the avidin-biotin peroxidase complex method with appro priately diluted rabbit antihuman antibodies to prostate spe cific antigen (PSA, 1:100 dilution) and prostatic acid phospha tase (PAP, 1:200 dilution) as primary antibodieso RESULTS
The debridement specimen consisted of a 4 cm. in diameter rim of devitalized perinea! tissue, including portions of the anal verge and necrotic ulcer base extending into soft tissue and skeletal muscle. Microscopic sections revealed an ulcer with a necrotic base extending into the subcutaneous fat and muscle with adjoining anal epithelium. Situated below the anal epithe lium were several distinct glandular foci strongly reminiscent of prostatic tissue (fig. 1). The glands were comprised of pseu dostratified cuboidal epithelium with a typical basal layer of cells surrounded by fibroelastic and smooth muscle septa (fig. 2). Occasional glands contained corpora amylacea (fig. 3, A). Immunohistochemical staining with PAP and PSA confirmed prostatic origin (fig. 3, B ). Accepted for publication May 17, 1991. * Requests for reprints: 15403 Lake Magdalene Blvd., Tampa, Flor ida 33613.
FIG. 1. Anal epithelium with submucosal prostatic glandular foci. H & E, reduced from X40. DISCUSSION
Several theories regarding the pathogenesis of prostatic het erotopia have been formulated. Some physicians contend that