patient is not clear. While renal impairment was present, this is not necessarily a contraindication. 6 The massive tumor area in the bladder probably presented a large absorptive area. However, the bleeding ceased promptly. The only other unusual feature in this patient was the fact that he had undergone alum irrigation 2 months before this episode. A mechanism to explain this increased absorption at the secondary exposure is not obvious but the possible association between retreatment and increased absorption should be considered in future clinical situations. In summary, while intravesical alum irrigation for intractable hematuria is generally safe and effective, we report a case of systemic aluminum toxicity and death after its use. Serum aluminum levels may prove to be useful in the monitoring of these patients, especially in the face of renal impairment, although the turnaround time for this test at most laboratories allows only for a retrospective diagnosis. The development of lethargy, confusion, seizures or metabolic acidosis in a patient receiving intravesical alum mandates cessation of treatment,

supportive measures and the selection of an alternative modality to control the hematuria. REFERENCES l. Kavoussi, L. R., Gelstein, L. D. and Andriole, G. L.: Encephalopathy

2. 3.

4. 5.


and an elevated serum aluminum level in a patient receiving intravesical alum irrigation for severe urinary hemorrhage. J. Urol., 136: 665, 1986. Ostroff, E. B. and Chenault, 0. W., Jr.: Alum irrigation for the control of massive bladder hemorrhage. J. Urol., 128: 929, 1982. Donahue, L. A. and Frank, I. N.: Intravesical formalin for hemorrhagic cystitis: analysis of therapy. J. Urol., 141: 809, 1989. Goel, A. K., Rao, M. S., Bhagwat, A.G., Vaidyanathan, S., Goswami, A. K. and Sen, T. K.: Intravesical irrigation with alum for the control of massive bladder hemorrhage. J. Urol., 133: 956, 1985. Mukamel, E., Lupu, A. and deKernion, J. B.: Alum irrigation for severe bladder hemorrhage. J. Urol., 135: 784, 1986. Modi, K. B. and Paterson, P. J.: Alum irrigation in massive bladder hemorrhage in severe renal failure. Amer. J. Kidney Dis., 12: 233, 1988.

0022-534 7 /92/14 73-0699$03.00/0 Vol. 147, 699-701, March 1992


Printed in U.S.A.



From the Departments of Urology and Pathology, Hartford Hospital, Hartford, and Division of Urology, University of Connecticut Health Center, Farmington, Connecticut


We report an additional case of primary adenoid cystic carcinoma of Cowper's gland in an otherwise healthy asymptomatic 66-year-old man. Based on a review of similar lesions presenting in the head and neck, our treatment plan entailed pelvic exenteration followed by radiation therapy. Whether adenoid cystic carcinoma of Cowper's gland behaves as aggressively as it does in the head and neck remains unclear. However, in view of the extensive local invasion in our patient and a previously successful outcome, a combined surgical and radiation approach appears to be the most appropriate therapy. KEY WORDS:

carcinoma, bulbourethral glands

Adenoid cystic carcinoma of Cowper's gland is an extremely rare entity, the only case being reported to our knowledge approximately 20 years ago. Since the behavior of this disease is unknown we believed it necessary to extrapolate data from adenoid cystic carcinoma occurring at nonurological sites in the treatment of our patient. Through careful literature review and personal correspondence with the author of the previous case report we formulated our treatment plan. CASE REPORT

A 66-year-old man had a rectal mass found by the primary care physician. Subsequent examination by a urologist and colorectal surgeon suggested that the mass was separate from the prostate gland. The patient suffered only mild obstructive voiding symptoms, notably a slight decrease in the force of the urinary stream along with some mild hesitancy. He denied any perinea! pain or discomfort, dysuria, hematuria, change in bowel habits or any recent weight loss. At physical examination the patient was moderately overAccepted for publication June 28, 1991.

weight and slightly hypertensive but otherwise healthy. Rectal examination revealed a soft, small prostate above a discrete, firm mass on the left side near the prostatic apex. The remainder of the physical. examination was unremarkableo Laboratory analysis revealed normal acid phosphatase (PAP) and prostate specific antigen levels, and normal kidney function tests. Urinalysis did not demonstrate any pyuria, hematuria or bacteriuria. Transrectal ultrasound revealed a large mass (3 to 4 cm.) extending inferiorly from the prostatic apex into the rectal wall. Hypoechoic areas were also noted at the left and right apexes of the prostate gland. An additional 1.5 cm. solid mass was noted within the bladder on the left trigone. Biopsies of the prostate and mass were interpreted as adenoid cystic carcinoma. Subsequent computerized tomography and magnetic resonance imaging (MRI) demonstrated a mass extending into the urogenital diaphragm and corpora cavernosa (fig. 1). A bone scan and chest x-ray were negative for metastatic disease. Pelvic exenteration was performed leaving the patient with a colostomy and an ilea! conduit. A polyglycolic acid mesh sling was placed to keep the peritoneal contents from the pelvis in anticipation of the possible need for radiation therapy postoperatively. lntraoperatively, the mass was clearly separate from



FIG. 1. MRI views. A, axial proton density image (repetition time 2,000, echo time 40) of mass (arrow) involving urogenital diaphragm, urethra and corpora cavernosa. B, Tl-weighted sagittal image (repetition time 700, echo time 20) of mass (arrow) involving urogenital diaphragm.

the prostate gland, which appeared to be small and unremarkable. The mass and the prostate measured approximately 3 to 4 cm. The prostate was soft and benign to palpation, while the mass was hard and extensively involved with the urogenital diaphragm. The mass also involved the left ischial tuberosity. Convalescence was complicated by a right lower extremity compartment syndrome on postoperative day 1 requiring urgent fasciotomies of the anterior and lateral compartments below the knee. A split thickness skin graft was eventually needed to cover the defect. The patient also suffered a prolonged ileus that resolved after several weeks of parenteral hyperalimentation and nasogastric suction. He was ultimately discharged from the hospital on postoperative day 45, ambulating well and tolerating a regular diet. After a 6-week convalescence the patient underwent radiation therapy. The low pelvis and perineum were treated with a 4-field technique with a total dose of 6,000 cGy. during 45 days. The patient tolerated the radiation well with no gastrointestinal symptomatology. Pathological examination of the specimen revealed a firm, white fibrous mass that was distinct from the prostate gland. The mass invaded the rectal wall anteriorly but did not penetrate the rectal mucosa. The mass encased the proximal urethra for a distance of approximately 3 cm. The cut section of the prostate showed diffuse involvement of all lobes by a firm yellowish process. Examination of the bladder demonstrated a 1.5 cm. papillary mass within the trigone on the left side. Histologically, the tumor had the appearance of an adenoid cystic carcinoma (fig. 2). Small uniform basaloid tumor cells grew in characteristic acini and cribriform patterns. In the latter case the tumor cells clustered around eosinophilic hyaline cylinders. The hyaline cylinders stained positively with periodic acid, Schiff and alcian blue stains at pH 2.5, consistent with basement membrane material showing mucinous degeneration. Immunostains for PSA and PAP were negative, while cytokeratins and S-100 protein immunostains were reactive. This immunophenotype is compatible with previous descriptions of adenoid cystic carcinoma. The tumor was believed to arise from the bulbourethral gland by virtue of the bulk of the tumor being outside the prostate with only minimal direct extension into the prostatic capsule and rectal wall. Perineural invasion was extremely prominent, and soft tissue and bony margins on the left side were positive. Although the tumor abutted the right lateral margins of resection, the margins were microscopically negative.

FIG. 2. Adenoid cystic carcinoma. Uniform population of small basaloid tumor cells grows in characteristic cribriform pattern with central hyaline cylinders. A, H & E, reduced from X75. B, H & E, reduced from X200. DISCUSSION

Cowper's glands or bulbourethral glands are diverticular outgrowths from the epithelial lining of the urogenital sinus. These pea-sized structures are embedded within the urogenital dia-


phragm and composed of several lobules containing epithelial lined acinio The glands secrete a clear mucus that constitutes a portion of the ejaculateo 10 2 Although Carpenter and Bernardo reported a case of primary adenoid cystic carcinoma of Cowper's gland in 1971, this tumor is more commonly found in the head and neck regiono 3 Also known as a cylindroma, a term coined by Bilroth for the manner in which the cells grow in tubes or cylinders, the tumor is usually slow growing, widely infiltrative and of moderate to low grade malignant potentiaL The tumor can also be found in the breast, cervix, trachea, esophagus and prostate gland" Grossly, the tumor has a grayish-white appearance, while microscopically there are varied patterns of small darkly staining cells with minimal cytoplasmo 4 The natural history of the disease varies but recurrences are frequent even after prolonged followup" The tumor is often locally aggressive, and can metastasize to the lung and braino 5 The natural history of adenoid cystic carcinoma involving the prostate gland is less well known" Only 6 cases have been reported, all with uniformly good outcomes within 1 to 6 yearso 6 Reported treatments include either transurethral resection of the prostate alone or in combination with radiation or a radical operation" The appropriate therapy of adenoid cystic carcinoma of Cowper's gland remains uncertain" Carpenter and Bernardo achieved a 13-year survival after local resection and adjuvant radiation therapy of 3,500 rad in the face of positive surgical margins" 30 7 The patient died of causes unrelated to the primary disease" Most articles discussing the management of adenoid cystic carcinoma involve treatment of the head and neck, particularly the major and minor salivary glands" For tumors in these regions the treatment options include an operation, radiation or a combined approach" Chemotherapy has not been shown to be effective" Complete excision appears to provide the best survivaL Adjuvant radiation therapy is usually administered only when complete surgical excision is not possible but does not appear to benefit patients who have significant residual disease at the surgical marginso 8 Distant metastasis can

occur despite local controL 9 Combined surgical and radiation approaches have achieved survival rates up to 70% ranging from 2 to 17 years after treatment" 10 Accordingly, we recommend a combined surgical and radiation approach for this rare entityo Drso Stephen Sussman and Andrew Salner, Departments of Radiology and Radiation Oncology, respectively, assisted in the preparation of this manuscripto REFERENCES

L Keen, Mo R, Golden, R Lo, Richardson, Jo Fo and Melicow, Mo Mo:


30 40 50 60

70 80

90 100

Carcinoma of Cowper's gland treated with chemotherapy" Jo UroL, 104: 854, 19700 Coffey, Do So: The biochemistry and physiology of the prostate and seminal vesicles" In: Campbell's Urology, 5th edo Edited by Po Co Walsh, R Fo Gittes, Ao Do Perlmutter and To Ao Stamey" Philadelphia: WO R Saunders Coo, voL 1, secto I, chapL 5, Po 235, 19860 Carpenter, Ao Ao and Bernardo, Jo R, Jro: Adenoid cystic carcinoma of Cowper's gland: case report. Jo UroL, 106: 701, 197L Frankel, K and Craig, Jo R: Adenoid cystic carcinoma of the prostate" Report of a case" Amero Jo Clino Path", 62: 639, 19740 Pellegrino, So Vo: Glossopyrosis due to adenoid cystic carcinoma" Oral Surgo, 43: 521, 19770 Young, RR, Frierson, R Fo, k, Mills, So K, Kaiser, Jo So, Talbot, WO Rand Bhan, Ao K: Adenoid cystic-like tumor of the prostate gland" A report of two cases and review of the literature on "adenoid cystic carcinoma" of the prostate" Amero Jo Clino Patho, 89: 49, 19880 Bernardo, Jo R: Personal correspondence" Shingaki, So, Saito, R, Kawasaki, To and Nakajima, To: Adenoid cystic carcinoma of the major and minor salivary glands" A clinicopathological study of 17 cases" Jo Maxillofaco Surgo, 14: 53, 19860 Matsuba, R Mo, Thawley, So K, Simpson, Jo R, Levine, L Ao and Mauney, Mo: Adenoid cystic carcinoma of major and minor salivary gland origin" Laryngoscope, 94: 1316, 19840 Shidnia, R, Hornback, No R, Hamaker, R and Lingeman, R: Carcinoma of major salivary glands" Cancer, 45: 693, 19800

0022-534 7/92/14 73-0701$03000/0 VoL 147, 701-703, March 1992


Printed in US.A.





From the Departments of Urology, University of California School of Medicine, San Francisco and Kaiser Foundation Medical Center, Oakland, California


Primary extranodal lymphoma of the female urethra is rare" We present case 10 and review the literature" Management in the past has varied markedly" Strategies have included simple excision, external beam and intracavitary radiation therapy, and multiple agent chemotherapy. The majority of patients have done well: 7 of 10 are alive with no evidence of disease" Newly diagnosed patients should undergo complete staging with the Ann Arbor staging system and the tumors should be classified histologically according to the Working Formulation for Clinical Usage" Therapy must be individualized but in most cases we recommend that patients be treated as if they have systemic illness, with multimodality therapy" KEY

WORDS: urethra, lymphoma

Accepted for publication August 2, 199L *Requests for reprints: Department of Urology, U-575, University of California, San Francisco, California 94143-07380

Lymphomas rarely present with a primary genitourinary complaint When they do, further evaluation commonly reveals evidence of widespread disease" Primary extranodal lymphoma

Adenoid cystic carcinoma of Cowper's gland.

We report an additional case of primary adenoid cystic carcinoma of Cowper's gland in an otherwise healthy asymptomatic 66-year-old man. Based on a re...
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