THB JOC'R~AL OF l,'R.OT,OGT
Vol. 70, ~u. 2, A. ugn:-;t Hl5:; fJ0>11tcd 1:n C.S ...:l
PRIAPISIV[ DFE TO CKriSUAL l\1ETARTASIS FROl\1 BLADDER CARCE\'"OMA: CASE HEPORT L. N. BERARD
AXD
JOH_\' E. BYRXE
Frmn the Departm.ent of C'rology, 81. Loicis Cn1:11ersi{y School of J1edu:inc, St. Low:s, .lfo
Scattered reports of priapism due to neoplastic infiltration of the penis have appeared in the British and American literature. 1,3.34, 0 Hinman 6 in a rm'te\Y of I 70 cases of priapism reported in 1914 found 2 cases to be due to _primary neoplastic infiltration; metastatic infiltration failed to account for the condition in any of the cases in his senes. Ikeda, Foley and Rosenow 7 reviewed the literature in 1943 on the subject of malignant priapism. They found 8 cases of priapism due to malignant .involvement of the penis by a primary neoplasm; to this number, the authors added a case of their own. In the group of cases in which the priaµism was due to secondary or metastatic tumor in the penis, they found a total of fifteen cases. The primary tumor in these cases was found to be: prostate, ti r-asn;; bladder, 4 cases; test,icle, 2 cases; rectum, 2 cases; kidney, 1 case; and liver) J case. In one of the previously mentioned cases, Kernel" reports a case of bladder carcinoma rnetastasing to the corpora cavernosa. In this case, the initial mYolvement occurred in the distal segment of the penis. Following the development of a single me!;astatic nodule iu the region of the glans penis, the entire peuile shaft \Vaci soon involved ill rapidly spreading tumor tissue. The spread of bladder carcinoma to the penis is nrmsnal. It seldom i11 voh cs the prostate, the seminal vesicles or the urethra by direct ext,ension.'3 It was originally felt that carcinoma of the bladder tends to remarn locaJizecl. As early as 1881 Albarran 9 demonstrated lymph node met.a,:;tasis from carcinoma of the bladder. Colston and Leadhetter 10 found distant 1netastasis in (il per cent of their cases. Spooner 11 found metastasis to the retroperitoneal lymph nodes, liver, lungs, and hone in 29 per cent of l G7 cases of bladder carcinoma that were said to ha Ye died from other causes than the malignancy. Baker 1~ rne11tio1rn 4 cases in which 1 Peacock, A. H ;\falignant priapism due to seconcLuy c11rcinonu1. of the corpor:1 cnvernosa. ,J. UroL, 49: 732-747, 1943. 2 Begg, R. C.: Persisten I, priapism due 1.o secondary carcinoma of the corpora t'.averno,s:t. Brit. M. J., 2: 10, HJ28. 3 Burrell, ,J. C.: Priapism due to metastatic bypernephrorna in the corpora cavernosa. J. lJrol., 60: fi36, Hl48. 1 Peters, C. N. and Huntress, R. L.: Priapisrn and ehordce due lo metastatic carcinumn of lhe penis, with prostate being the primary sour-en. J. l1rol., 49: 810, rn:,8. 5 Kessel, J. S .. Interesting case due to nrnliiple cflrcinomatous nodul(,s in the corporu. c·a.vornosa. J. Urol., 32: 213-216, 19;34_ "Hirnna11, F. · Priapism. Ann. Snrg., 60: fifl8, HJH. 7 lkec!a, K., Foley, F. E. B. ,we! Rosenow, J.: 1\lalignant pria.pism S(:condary to c,u. cinoma of the urethra J. Urol, 49: 732-747, H/4,l. 8 Ackerman, V. L. and de! Regata, ,T. A .. Cm,cer: Diagnosis, Treatment and Progrt
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L. N". BERARD AND JOHN E. BYRNE
bladder carcinoma developed penile metastasis follmving surgical treatment of the bladder tumor. All 4 cases had a different type of malignancy, and in all 4 cases the patients received some form of treatment to their obstructed bladder outlet, prior to, simultaneously, or shortly following surgical treatment of the bladder tumor. He points out the possibility of rapid penile metastasis following the invasion of recently operated prostatic tissue. CASE REPORT
A 68 year old white executive was first admitted to the urological service by one of us (L. N. B.) on August 15, 1949. His initial complaint was that of frequency of urination and nocturia of 8 years' duration. The patient was known to have had a residual urine in excess of 6 ounces for the past 4 years, but on many occasions he had denied permission for surgery. In the 3 months prior to hospitalization his frequency had increased to hourly voiding, and he was getting up 5 to 6 times during the night. Except for the foregoing, his genito-urinary history was negative. He had always been in good health, and there was no familial history of cancer. Physical examination disclosed a rather emotional, elderly white man who did not appear to be ill. The positive clinical findings revealed blood pressure 180/100 without signs of cardiomegaly or congestive heart failure. Examination of the abdomen was negative. Rectal examination disclosed grade 2 hypertrophy of the prostate without evidence of fixation or malignancy. The genitalia were negative, the penis was uncircumcised, and both testicles were in the scrotum free of enlargement or tenderness. The urethra was unobstructed to the passage of a 20F soft rubber catheter; 4 ounces of grossly clear residual urine. The initial urinary cultures were sterile. All of the laboratory examinations including the nonprotein nitrogen and the acid and alkaline phosphatase determinations were within normal limits. Cystoscopic examination on August 22, 1949 revealed median bar type of prostatic obstruction, and an irregular, granular area of necrotic tissue on the bladder wall just below the right ureteral orifice. Our initial impression was that of carcinoma of the bladder, type undetermined, and median bar prostatic obstruction. On August 26, 1949, a transurethral resection of the prostatic bar, and a biopsy of the suspicious bladder mucosa was performed. Bimanual rectal examination under anesthesia at this time failed to demonstrate a palpable lesion of the bladder. The pathological report on the resected tissue was that of benign prostatic hyperplasia and epidermoid carcinoma of the bladder, grade l. The patient was markedly relieved of his symptoms following surgery, and although informed of the malignancy he refused further surgery at this time. The patient was discharged from the hospital 10 days following operation. The patient was not seen again until November 1949, when he again returned complaining of increasing frequency and severe dysuria of three weeks' duration. He stated that he had noticed "tissue shreds" in his urine in the past 8 days. He gave no history of hematuria following the previous postoperative period. The patient was again hospitalized; his physical examination and laboratory determinations were unchanged from the first admission. He had 1 ounce of rather dirty
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residual urine. Culture revealed an E. coli infection. Excretory urograms revealed good function and apparently normal renal outline. Cystoscopic examination revealed the area of resected prostate to be well healed with an adequate bladder outlet. The previously noted tumor mass had changed little since the previous observation. It was a single broad base tumor, approximately 0.5 cm. in its greatest diameter, and surrounded by a low ridge of necrotic tissue. The patient was advised of the need for radical surgery, but again refused. On November 30, 1949 the tumor was resected transurethrally, and the base of the tumor fulgurated. At the time of surgery, bimanual rectal examination failed to reveal evidence of extension of the tumor through the wall of the bladder. The pathological report on the resected tissue was epidermoid carcinoma of the blad-
Fm. 1. A, section of bladder tumor removed transurethrally 3 months following initial biopsy. Again showing an epidermoid carcinoma, grade 1. B, section taken from indurated area in shaft of penis. Note extensive invasion of squamous tumor cells in architecture of penis. Further sections from this area showed involvement of corpora spongiosum as well as the cavernous bodies.
der (fig. 1, A). The postoperative period was uncomplicated, and the patient was discharged from the hospital on December 3, 1949. He was not seen again until April 1950, when he came to us complaining of severe dysuria, frequency, and periodic hematuria of 10 days' duration. He stated that for 3 days prior to consultation he had been experiencing persistent and painful erections. The patient was again hospitalized. Physical examination disclosed that he had failed miserably since the previous examination. He had lost 20 pounds in weight, was pale, and markedly dehydrated. Examination of the heart and lungs was not remarkable. The abdomen was soft, and no mass could be palpated. Examination of the genitalia revealed the penis to be semi-erect, and generally firm and tender (fig. 2). The penis was firm from a point just behind the corona, to the level of the suspensory ligament. In the midpoint of the shaft of the penis, there was noted
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L. K. BERARD AND JOHN K
BYRNE
an area of greater density than the remainder of the organ. This area was approximately 2 cm. in length, and extended through the entire cross-section diameter of the penis. Rectal examination disclosed that the prostate had changed little in size or position, although at this time it was quite tender to palpation. Tender areas of induration were present over each seminal vesicle. Laboratory examination revealed anemia (2~:2 million red blood cells with 7 grams of hemoglobin). The nonprotein nitrogen was 60 mg. per cent. Urinary culture revealed a moderate growth of E. coli. Excretory urograms revealed mild right hydronephrosis with delayed emptying of the dye on the right; the left side appeared normal. X-ray examination of the chest failed to reveal evidence of metastatic lesion. Supportive therapy was begun upon admission to the hospital. He was given three transfusions of 500 c:c each of whole blood with the correction of the anemia over a period of 5 days. Parenteral fluids were given with correction of the initial dehydration. Dihydrostreptomycin 0.5 gm. 4 times daily for 5 days produced sterile urinary cultures on the fifth day.
No Iumor
Frn. 2. Diagrammatic representation of localization of metastatic tumor in shaft of peniR.
Cystoscopy under anesthesia 7 days after admission revealed an extensive tumor involving the entire base of the bladder, and obstructing the right ureteral orifice. Rectal examination disclosed invasion of the prostate and seminal vesicles. The patient was requiring large doses of opiates for the relief of pain; in the face of the extensive local extension of the tumor we believed that we had little to offer him other than a palliative cystectomy and diversion of the urinary stream to the colon. The patient was prepared with oral urinary and gastro-intestinal antiseptics for 7 days. His appetite was good, he was no longer anemic:, and his hydration was excellent. On Niay 8, 1950, under cyclopropane anesthesia, a total cystectomy with removal of the prostate, seminal vesicles, and the regional lymph nodes -was carried out. The tumor had extended into virtually all of the pelvic structures, and the dissection was tedious and time-consuming. The ureters were anastomosed to the lower sigmoid in a mucosa to mucosa fashion, without splinting catheter,,. Both ureters appeared normal at the time of surgery. In the face of our preoperative impression of metastatic neoplasm in the penis, the organ was removed at its base.
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The patient tolerated surgery poorly and showed signs of shock during the! late of the opern.tion, and in the immediate postoperative period. Blood pressure stabilized following the administration of multiple whole blood transfusiom, and plasma. He was given both penicillin a11d streptornyciu in Lhera.peutic dosage and fluids were administered for the first 3 "Urinary out.put through a de Pezzer catheter inserted ill the rectum was 500 cc in the first 12 hours postoperatively. l:rinary output subsequently increased to an average 1500 ce and maintained itself at approximately that len-,l for the next 4 days. Ou trw fourth postoperatiYe day the patient had chills, fever, alld septic. He to the antibiotics on the seventh day and was temperature free. Little or no change occurred in the urinary output during the period of sepsis. After this period, the urinary output wa:,; illcreased to 2500 cc daily, the patient was ambulatory and toleratiug food and fluids weJl. He was e,·acuating the hmYel of urine every 2 to 3 hours duriug the day and ;:3 to 4 time,~ during the night. Pathological report on the surgical specimen revealed a 4 epidermoid carcinoma of the bladder, ,vith extension to the prostate and the c1emi11al vesicles. F'iftecn of twenty lymph nodes examined were i11nih·c-d th<· Lurnor. Serial sections through the penis revealed the interesting feature of this ca.se.
Sections taken from the base of the penis showed normal penile architPcture without e,·idence of tumor. Sections taken from the previously described area ;J cm. from the base of the penis revealed dense tumor infiltration of the corpora caver11osa and the corpus spongiosum . The area of infiltration exte11ded to\\'ard;-; the distal end of the for a distance of 2.5 cm. (fig. 1, Serial sed.iom, taken from the shaft of the penis distal to the above segment, and from the glan~ penis failed to reveal further eYidern:e of tumor. The patient had been doiug wel I except for periodic bouts of low-grade temperature elevation, when 011 his fifth nn.c•h,n,~pq he complained of suddeu sharp pain in hito chest, he exwithin ao minutec: of the onset of pain. An autop8y wa,~ performed, and evidence of a recent anterior myocardial infarction was found. Scattered areas of metastatic epidermoid ean-inoma ,vere present in the lung fields, but no otlwr evidence of tumor extension c:onld be demonstrated. The meterosigmoidostumy wm, intact, and the showed only moderate hydronephrnsis TherP mt,, evidence of chronic bilaterally. DISCFSSION
A case of priapism due to nwta:-;tasi:o of a bladder c:arcinoma to the penis is reported. This c:ase is unusual 1101 only in the pathological process, but e,·en more so in eon.~iderntion of the mechanism of the metastasis. In analyzing Lhe case all of the know11 rouies of metastatic spread were c011sidered. Tbe chance of direct implantatim1 of this tumor by urethral instrumentation rn mu minds, more than a theoretical corn,ideration. In view of the pulrnonary metastasis found at autopsy, the blood borne rnute of tumor spread would seem quite logically responsible for the penile le:-;wn This 011 two count,;. First, there was no previom, ,·ascular thromfJosis is not
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L. N. BERARD AND JOHN E. BYRNE
or obstrnction which could conceivably localize tumor emboli in a single segment of the penile sl:aft. Secondly, had the tumor been blood borne it would seem more likely for the involvement to have occurred in the terminal portion of the organ. The tumor emboli localized in the end vessels as ,vas shown in other reported cases. 5 The fact that the base of the penis was free of tumor would rule out the possibility of the bladder tumor extending into the resected prostate, and thence into the penis. Retrograde lymphatic extension although certainly a possibility does not seem likely in the absence of gross edema or microscopic evidence of lymphatic distention. To further the argument of direct implantation of the tumor is the fact that this patient had been instrumented many times with large caliber instruments (24~28F) since the demonstration of the original lesion. The likelihood of such instrumentation denuding the urethra and lacerating the mucosa is obvious. The lacerated urethral mucosa offers an excellent medium upon which the displaced squamous tumor cells could lodge and proliferate. The involvement of the corpus spongiosum as well as the corpora cavernosa lends more weight to the theory of direct implantation of the tumor. This pathological entity, although certainly a rarity, is of serious implications. Instrumentation of the urethra in any form is to be kept at a minimum in the face of a bladder neoplasm. Our thanks are expressed to Dr. ·w alter Rice of the Department of Pathology of the St. Louis University School of Medicine for his aid in the interpretation of the pathological sections. Our thanks also to Mr. Phillip Conrath of the Department of Medical Illustration of the St_ Louis University School of Medicine for his aid in the preparation of the illustrations.