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Ganglioneuroma of Bladder: Report of a Case1
THE JOURNAL OF UROLOGY
Vol. 63, No. 3, Marchl950
Printed in U.S.A.
GANGLIONEUROMA OF BLADDER: REPORT OF A CASE 1 HUGHE. WYMAN, BUFORD S. CHAPPELL JONES, JR.
AND
WILLIAM RUSSELL
From the Department of Urology, Veterans Administration Hospital, Columbia, S. C.
Ganglioneuroma is a very rare tumor, composed of adult nerve cells and fibers. It is more often found in the peripheral ganglion than in the central nervous system and is most commonly found in the retroperitoneal tissues along the aorta and in the pelvis. The tumors are moderately firm, elastic, with a distinct capsule, glistening, translucent and yellowish pink. They are divided into lobules by connective tissue septa and on microscopic examination, bundles of nerves embedded in connective tissue stroma with numerous ganglion cells. The ganglion cells contain one or more processes and occasionally contain two nuclei and with Nissl substance in the cytoplasm. Ganglioneuromas are occasionally multiple and may be associated with other sympathetic nervous system tumors. No functional activity has been attributed to these tu~ors and they cause symptoms by pressure. Stout in 1947 found available reports of 233 cases of ganglioneuromas in the literature to which he added 10 previously unreported cases. Of the cases, 199 were suitable for analysis and on this basis, he was able to devise a classification of the tumors into 3 groups: 1) with fully differentiated cells and do not metastasize. This group includes a majority of the tumors; 2) partly differentiated group with occasional metastasis; 3) a small but complex group of fully differentiated and fully malignant sympathicablastomatous nodules and capable of metastasis. Further analysis by Stout revealed that 60 per cent of the cases occurred before the age of 20 years. Surgery could not be evaluated because only 6 of the cases were operated on and only 4 of those not operated on had been followed for more than 3 years. While it is generally conceded that this type of tumor is most commonly located along the great chains of the sympathetic ganglia, they occur in other locations, notably the adrenal medulla and posterior mediastinum. The gastrointestinal canal, appendix, uterus and other viscera have occasionally been the site of this tumor. We believe ganglioneuroma of the bladder to be unique and a case is therefore reported. CASE REPORT
A 17 year old white man entered this hospital on May 8, 1944, complaining of dysuria, pain in right flank and fever. These symptoms had been present for about 8 days. Past history revealed that patient had had the usual childhood diseases and was said to have had malaria 7 years ago. He had also suffered with tonsillitis and pneumonia about 10 years prior to admission and on one occasion 1 Sponsored by the Veterans Administration and published with the approval of the Chief Medical Director. The statements and conclusions published by the authors are a result of their own study and do not necessarily reflect the opinion or policy of the Veterans Administration. 526
GANGLIONEUROMA OF BLADDER
527
was thought to have had hookworm infestation. The military history was brief, patient having served 2 months as Pvt. USMC with no overseas service. The patient was acutely ill. He was of somewhat nervous temperament. The temperature -was 99, pulse 110, respirations 22. Positive physical findings showed mild generalized abdominal tenderness with well healed appendectomy scar. The blood pressure was 128/84. The pulse was rapid and regular. Several small verruca were present on the dorsum of the hands. Laboratory examinations: Complement fixation and precipitation tests for syphilis were negative. Blood: Hemoglobin 14.2 gm., 88 per cent; white blood cells 11,100, polymorphonuclears 70 per cent, lymphocytes 25 per cent, monocytes 3 per cent, eosinophils, 2 per cent. Plasmodium malariae~none found. Urinalysis: cloudy, stra-w colored; alkaline; specific gravity 1.016; albumin 1 plus; sugar negative; white blood cells many; red blood cells 3; Casts none. Crystals, triple phosphate. Culture of stools negative. N onprotein nitrogen 37 mg. per cent. Complete urological study revealed an obstruction at the vesical neck due to hypertrophy of the median lobe of the prostate gland. Bilateral hydro-ureter and left hydronephrosis were present as well. Obstructing tissue at the vesical neck was resected transurethrally May 30, 1944. Tissue removed was reported as fibromuscular hyperplasia of the prostate gland by the pathologist. On July 12 a partial resection of the lower pole of the left kidney was done from which patient made an uneventful convalescence. Preoperative retrograde study of the left kidney had shown a shadow suggestive of calculus in the lower pole of the kidney. No calculus was present at operation. Tissue removed from the kidney shmved changes compatible with hydronephrosis and chronic pyelonephritis. The patient was discharged in an improved condition August 25, 1944. The second admission was on October 6, 1944, the patient complaining of dysuria, frequency and pain in the left flank. Examination showed some enlargement of the median lobe of the prostate gland and "persistent left hydronephrosis and hydroureter." Transurethral resection was performed again from which patient made an uneventful recovery. Prostatic tissue was again reported as fibromuscular hyperplasia by the hospital pathologist. At discharge on N ovember 20, 1944, it was believed that a left nephrectomy would ultimately be required although function of the left kidney (as determined by phthalein output) was fair. Following second admission to the hospital the patient apparently did well until June 11, 1945, when he was seized with sudden intermittent pain in the left loin radiating to the left flank and groin accompanied by severe shaking chills, fever and sweating. Frequency of urination, nocturia and dysuria were severe. Examination on admission, in addition to the findings previously noted, revealed the patient to be acutely ill and in pain. The temperature was elevated to 101 F, pulse 110, and respiration 24. There was marked tenderness over the left costovertebral angle and left lumbar region. Slight tenderness was noted over the right costovertebral angle as well. Positive laboratory findings showed elevation of leukocyte count to 10,500 with 78 per cent polymorphonuclears. The voided urine was loaded with white
528
H. E. WYMAN, B. S. CHAPPELL AND W. R. JONES
blood cells and acid in reaction. The nonprotein nitrogen was 39 mg. per cent. Following a course of sulfadiazine therapy, sedation and bed rest, he showed progressive improvement. Cystoscopy and retrograde study demonstrated practical loss of function of the left kidney with persistence of infection. There was a marked degree of pyelectasis and ureterectasis on the left with moderate dilatation of the right ureter as well. The renal pelvis and calyces were normal on the right side (fig. 1). Left nephrectomy was done on August 15, 1945, without complication, the patient being discharged again on September 8, 1945. Little or no obstruction
Fm. 1. A, bilateral ureterectasis incident to obstructive uropathy. Left kidney did not fill on injection of skiodan and function is lost. B, recent intravenous urogram. Calyceal pattern of remaining right kidney is relatively good. Ureterectasis persists. Left nephroureterectomy and removal of vesical growth has been done.
had been noted at the vesical neck on cystoscopy during this period of hospitalization. The patient's fourth admission to this hospital was in December 1946, when he returned complaining of dysuria, pain in right side and "pus in urine." General condition on this admission was good. Further transurethral resection, the third, was performed since the obstruction at the vesical neck had returned. A small amount of tissue, again reported as fibromuscular hyperplasia of the prostate gland, was resected relieving the obstruction. During this hospital stay patient underwent an operation for pterygium and was discharged in an improved condition on March 3, 1947. During the period between April 1947 and December of the same year, the patient was admitted 5 times for several days on each occasion for cystoscopy and dilatation of the ureters. Streptococcus
GANGLIONEUROMA OF BLADDER
529
faecalis and Proteus were cultured from the urine on several occasions. He continued to have intermittent pain in the right loin accompanied by bouts of frequency and dysuria. An additional admission followed in January 1948, when patient was admitted in a very intoxicated state with a stab wound of the right chest. He was treated expectantly for this, making a complete recovery from his chest injury in 10 days. Urinary symptoms persisted in spite of repeated cystoscopy and dilatations of the right ureter. Accordingly, it was decided to explore the bladder suprapubically on March 17, 1948. At operation there was a large, oval-shaped mass approximately 4.5 by 2.5 cm. lying in the left lateral wall of the bladder just anterior to the region of the left ureteral orifice. This mass was apparently covered by vesical mucosa and to effect its removal the entire thickness of the bladder
s-:iE-:>:, ..
Fm. 2. Surgical specimen, showing dilated left ureter adherent to tumor mass and penetrating bladder wall. Growth is of retrovesical origin.
had to be traversed. This was done by blunt and sharp dissection, the mass being enucleated principally with the finger. It was soon appreciated that the mass involving the bladder wall was intimately associated with the termination of the left ureter which had the configuration of a long sausage-like mass (fig. 2). This portion of the ureter was inadvertently opened and drained a small amount of yellowish pus. A second incision, a left pararectus incision, was then made through which the entire left ureter was removed extraperitoneally to the site of previous nephrectomy. The ureter was thickened and approximated the diameter of the normal ileum. The bladder wall was sutured in layers with catgut and a mushroom catheter was placed into the vesical cavity for drainage. Aside from a transient febrile response, patient made an uneventful convalescence following operation and was discharged April 16 with all wounds healed and voiding well. It was believed that all of the tumor which involved the bladder was removed at this operation. Pathological report: S-48-200, March 17, 1948. "Gross: An irregular encapsu-
530
H. E. WYMAN, B. S. CHAPPELL AND W. R. JONES
lated mass of tissue which measures 4.5 by 2.5 cm. On section it is composed of firm, pale gray tissue, which has a rubbery consistency. A section of ureter which is 18 cm. long and averages 2.8 cm. in diameter. The outer surface is rough and grayish-purple. On section the wall is thickened and in the lumen there is a small amount of brownish-yellow purulent material. "Microscopic: The mass is made up of large sheets of loosely arranged, elongated cells which have rather small, spindle-shaped, vesicular nuclei. Their cystoplasm is indistinct and scant. These cells are separated by a moderate amount of fibrillar material having an amphoteric tint. There is a pronounced tendency of the cells to be arranged in whorls and nuclear pallisading is easily demonstrated. In many areas spherical or oval masses of tissue are surrounded by a small amount
Fm. 3. A, spherical and oval masses of tissue surrounded by fibrous tissue resembling nerve fibers. (Low power.) B, note large oval cells reminiscent of ganglion cells of sympathetic trunk. Ganglioneuroma.
of fibrous connective tissue so that they have the appearance of nerve fibers. Scattered here and there, sometimes occurring singly and sometimes in groups, are very large cells which vary from oval to round. They have a granular eosinophilic cytoplasm and occasional cytoplasmic processes. Their nuclei vary from one to two in number and are generally eccentric in position. The nuclear membrane is distinct and the nucleolus prominent. Elsewhere the nuclear chromatin is scant. Throughout the sections there are a few scattered eosinophilic leukocytes. Diagnosis: Ganglioneuroma." The tissue removed by resectoscope a year later from nodule on trigone showed: "S-49-185, March 10, 1949. Gross: Three small sections of pale gray tissue (from trigone of bladder). Microscopic: The sections are similar to the previous specimen, being made up of numerous nerve cells and fibers. Diagnosis: Ganglioneuroma." See figure 3. At examination on March 10, 1949, patient was found to have slight dysuria
GANGLIONEUROMA OF BLADDER
531
and occsional pain in the right costovertebral angle, although his general condition was good. Cystoscopic examination, however, revealed a return of the growth at the vesical neck extending into the bladder (fig. 4). The prostatic fossa was of a uniform, slightly oval character such as might be found after a
Frn. 4. View at vesical neck obtained by panendoscope March 10, 1949. Growth has recurred and is covered by bladder mucosa. Note previously resected vesical neck.
Frn. 5. Diagram illustrating findings on bimanual examination under spinal anesthesia.
transurethral resection. Nodular irregularity of the floor of the bladder, including the trigone, the region of the right ureteral orifice, and the left bladder wall, were noted. The former site of the left ureteral orifice could be identified by a gutter along the floor of the bladder. Bimanual palpation under spinal anesthesia showed the outlines of the prostate
532
H. E. WYMAN, B. S. CHAPPELL A.ND W. R. JONES
to be identifiable. There were small, rather firm nodules located under the trigone of the bladder. The seminal vesicles were encompassed in the fibro-elastic field and could not be identified (fig. 5). Biopsies were taken, first from the nodular irregularities over the floor of the bladder and then the small nodule on the left wall of the bladder was biopsied and a third biopsy was taken from the vesical neck itself. Pathological study of the tissue removed revealed findings characteristic of ganglioneuroma. The patient was last examined in June 1949 and there has been great increase in the intravesical tumor and progression of the right hydro-ureter since the examination in March 1949. COMMENT
A 5 year period of observation afforded unusual opportunity in the study of this patient and the case report is given at some length. Of particular interest was the presentation of the patient with obstructive symptomatology referable to bladder and to ureters at the vesico-ureteral junction, almost 4 years before a positive diagnosis of ganglioneuroma could be made. The exact origin of the tumor must remain a point of conjecture but there is certainly ample evidence to show that the tumor eventually invaded the bladder and biopsies readily demonstrating this tumor were obtained by relatively shallow bites into the tumor on the trigone (fig. 5) with the resectoscope loop. Less than a year elapsed after surgical removal of all demonstrable tumor before reappearance of a tumor mass equal in size to the one removed surgically. No evidence of metastasis was found but pressure of the tumor has severely damaged his urinary tract with loss of one kidney and damage to the remaining kidney. Impairment of sexual ability is a feature not fully explained. SUMMARY
A case report of a ganglioneuroma of the bladder in a patient 22 years of age is reported for purposes of record. The patient was closely observed for the 5 year period since onset of symptoms. Symptoms were present for 4 years before diagnosis was made. Surgical removal of tumor transvesically did not effect a cure. Severe and progressive damage to the urinary tract resulted from pressure obstruction by the tumor. Acknowledgments: The authors wish to express appreciation to Dr. Kenneth M. Lynch of the Medical College of the State of South Carolina and to Dr. E. S. Cardwell for the study and diagnosis of the tissue removed surgically. We express our thanks to Dr. Theodore M. Yates of the Department of Urology, Veterans Administration Hospital, Columbia, South Carolina, for helpful suggestions and continued interest. REFERENCES ACKERMAN, L. V. AND DEL REGATO, J. A.: Cancer. St. Louis: C. V. Mosby Co., 1947, pp. 482 and 780. BoYD, W"llr.: A Textbook of Pathology. Philadelphia: Lea and Febiger, 1947, 5th ed., pp. 935. MooRE, R. A.: A Textbook of Pathology. Philadelphia: W. B. Saunders Co., 1945. STOUT, A. P.: Ganglioneuroma of sympathetic nervous system. Surg., Gynec. & Obst., 84: 101-110, 1947. YouNG, H. H. AND DAVIS, D. M.: Young's Practice of Urology. Philadelphia: W. B. Saunders Co., 1926, vol. 1.
Vol. 63, No. 3, Marchl950
Printed in U.S.A.
GANGLIONEUROMA OF BLADDER: REPORT OF A CASE 1 HUGHE. WYMAN, BUFORD S. CHAPPELL JONES, JR.
AND
WILLIAM RUSSELL
From the Department of Urology, Veterans Administration Hospital, Columbia, S. C.
Ganglioneuroma is a very rare tumor, composed of adult nerve cells and fibers. It is more often found in the peripheral ganglion than in the central nervous system and is most commonly found in the retroperitoneal tissues along the aorta and in the pelvis. The tumors are moderately firm, elastic, with a distinct capsule, glistening, translucent and yellowish pink. They are divided into lobules by connective tissue septa and on microscopic examination, bundles of nerves embedded in connective tissue stroma with numerous ganglion cells. The ganglion cells contain one or more processes and occasionally contain two nuclei and with Nissl substance in the cytoplasm. Ganglioneuromas are occasionally multiple and may be associated with other sympathetic nervous system tumors. No functional activity has been attributed to these tu~ors and they cause symptoms by pressure. Stout in 1947 found available reports of 233 cases of ganglioneuromas in the literature to which he added 10 previously unreported cases. Of the cases, 199 were suitable for analysis and on this basis, he was able to devise a classification of the tumors into 3 groups: 1) with fully differentiated cells and do not metastasize. This group includes a majority of the tumors; 2) partly differentiated group with occasional metastasis; 3) a small but complex group of fully differentiated and fully malignant sympathicablastomatous nodules and capable of metastasis. Further analysis by Stout revealed that 60 per cent of the cases occurred before the age of 20 years. Surgery could not be evaluated because only 6 of the cases were operated on and only 4 of those not operated on had been followed for more than 3 years. While it is generally conceded that this type of tumor is most commonly located along the great chains of the sympathetic ganglia, they occur in other locations, notably the adrenal medulla and posterior mediastinum. The gastrointestinal canal, appendix, uterus and other viscera have occasionally been the site of this tumor. We believe ganglioneuroma of the bladder to be unique and a case is therefore reported. CASE REPORT
A 17 year old white man entered this hospital on May 8, 1944, complaining of dysuria, pain in right flank and fever. These symptoms had been present for about 8 days. Past history revealed that patient had had the usual childhood diseases and was said to have had malaria 7 years ago. He had also suffered with tonsillitis and pneumonia about 10 years prior to admission and on one occasion 1 Sponsored by the Veterans Administration and published with the approval of the Chief Medical Director. The statements and conclusions published by the authors are a result of their own study and do not necessarily reflect the opinion or policy of the Veterans Administration. 526
GANGLIONEUROMA OF BLADDER
527
was thought to have had hookworm infestation. The military history was brief, patient having served 2 months as Pvt. USMC with no overseas service. The patient was acutely ill. He was of somewhat nervous temperament. The temperature -was 99, pulse 110, respirations 22. Positive physical findings showed mild generalized abdominal tenderness with well healed appendectomy scar. The blood pressure was 128/84. The pulse was rapid and regular. Several small verruca were present on the dorsum of the hands. Laboratory examinations: Complement fixation and precipitation tests for syphilis were negative. Blood: Hemoglobin 14.2 gm., 88 per cent; white blood cells 11,100, polymorphonuclears 70 per cent, lymphocytes 25 per cent, monocytes 3 per cent, eosinophils, 2 per cent. Plasmodium malariae~none found. Urinalysis: cloudy, stra-w colored; alkaline; specific gravity 1.016; albumin 1 plus; sugar negative; white blood cells many; red blood cells 3; Casts none. Crystals, triple phosphate. Culture of stools negative. N onprotein nitrogen 37 mg. per cent. Complete urological study revealed an obstruction at the vesical neck due to hypertrophy of the median lobe of the prostate gland. Bilateral hydro-ureter and left hydronephrosis were present as well. Obstructing tissue at the vesical neck was resected transurethrally May 30, 1944. Tissue removed was reported as fibromuscular hyperplasia of the prostate gland by the pathologist. On July 12 a partial resection of the lower pole of the left kidney was done from which patient made an uneventful convalescence. Preoperative retrograde study of the left kidney had shown a shadow suggestive of calculus in the lower pole of the kidney. No calculus was present at operation. Tissue removed from the kidney shmved changes compatible with hydronephrosis and chronic pyelonephritis. The patient was discharged in an improved condition August 25, 1944. The second admission was on October 6, 1944, the patient complaining of dysuria, frequency and pain in the left flank. Examination showed some enlargement of the median lobe of the prostate gland and "persistent left hydronephrosis and hydroureter." Transurethral resection was performed again from which patient made an uneventful recovery. Prostatic tissue was again reported as fibromuscular hyperplasia by the hospital pathologist. At discharge on N ovember 20, 1944, it was believed that a left nephrectomy would ultimately be required although function of the left kidney (as determined by phthalein output) was fair. Following second admission to the hospital the patient apparently did well until June 11, 1945, when he was seized with sudden intermittent pain in the left loin radiating to the left flank and groin accompanied by severe shaking chills, fever and sweating. Frequency of urination, nocturia and dysuria were severe. Examination on admission, in addition to the findings previously noted, revealed the patient to be acutely ill and in pain. The temperature was elevated to 101 F, pulse 110, and respiration 24. There was marked tenderness over the left costovertebral angle and left lumbar region. Slight tenderness was noted over the right costovertebral angle as well. Positive laboratory findings showed elevation of leukocyte count to 10,500 with 78 per cent polymorphonuclears. The voided urine was loaded with white
528
H. E. WYMAN, B. S. CHAPPELL AND W. R. JONES
blood cells and acid in reaction. The nonprotein nitrogen was 39 mg. per cent. Following a course of sulfadiazine therapy, sedation and bed rest, he showed progressive improvement. Cystoscopy and retrograde study demonstrated practical loss of function of the left kidney with persistence of infection. There was a marked degree of pyelectasis and ureterectasis on the left with moderate dilatation of the right ureter as well. The renal pelvis and calyces were normal on the right side (fig. 1). Left nephrectomy was done on August 15, 1945, without complication, the patient being discharged again on September 8, 1945. Little or no obstruction
Fm. 1. A, bilateral ureterectasis incident to obstructive uropathy. Left kidney did not fill on injection of skiodan and function is lost. B, recent intravenous urogram. Calyceal pattern of remaining right kidney is relatively good. Ureterectasis persists. Left nephroureterectomy and removal of vesical growth has been done.
had been noted at the vesical neck on cystoscopy during this period of hospitalization. The patient's fourth admission to this hospital was in December 1946, when he returned complaining of dysuria, pain in right side and "pus in urine." General condition on this admission was good. Further transurethral resection, the third, was performed since the obstruction at the vesical neck had returned. A small amount of tissue, again reported as fibromuscular hyperplasia of the prostate gland, was resected relieving the obstruction. During this hospital stay patient underwent an operation for pterygium and was discharged in an improved condition on March 3, 1947. During the period between April 1947 and December of the same year, the patient was admitted 5 times for several days on each occasion for cystoscopy and dilatation of the ureters. Streptococcus
GANGLIONEUROMA OF BLADDER
529
faecalis and Proteus were cultured from the urine on several occasions. He continued to have intermittent pain in the right loin accompanied by bouts of frequency and dysuria. An additional admission followed in January 1948, when patient was admitted in a very intoxicated state with a stab wound of the right chest. He was treated expectantly for this, making a complete recovery from his chest injury in 10 days. Urinary symptoms persisted in spite of repeated cystoscopy and dilatations of the right ureter. Accordingly, it was decided to explore the bladder suprapubically on March 17, 1948. At operation there was a large, oval-shaped mass approximately 4.5 by 2.5 cm. lying in the left lateral wall of the bladder just anterior to the region of the left ureteral orifice. This mass was apparently covered by vesical mucosa and to effect its removal the entire thickness of the bladder
s-:iE-:>:, ..
Fm. 2. Surgical specimen, showing dilated left ureter adherent to tumor mass and penetrating bladder wall. Growth is of retrovesical origin.
had to be traversed. This was done by blunt and sharp dissection, the mass being enucleated principally with the finger. It was soon appreciated that the mass involving the bladder wall was intimately associated with the termination of the left ureter which had the configuration of a long sausage-like mass (fig. 2). This portion of the ureter was inadvertently opened and drained a small amount of yellowish pus. A second incision, a left pararectus incision, was then made through which the entire left ureter was removed extraperitoneally to the site of previous nephrectomy. The ureter was thickened and approximated the diameter of the normal ileum. The bladder wall was sutured in layers with catgut and a mushroom catheter was placed into the vesical cavity for drainage. Aside from a transient febrile response, patient made an uneventful convalescence following operation and was discharged April 16 with all wounds healed and voiding well. It was believed that all of the tumor which involved the bladder was removed at this operation. Pathological report: S-48-200, March 17, 1948. "Gross: An irregular encapsu-
530
H. E. WYMAN, B. S. CHAPPELL AND W. R. JONES
lated mass of tissue which measures 4.5 by 2.5 cm. On section it is composed of firm, pale gray tissue, which has a rubbery consistency. A section of ureter which is 18 cm. long and averages 2.8 cm. in diameter. The outer surface is rough and grayish-purple. On section the wall is thickened and in the lumen there is a small amount of brownish-yellow purulent material. "Microscopic: The mass is made up of large sheets of loosely arranged, elongated cells which have rather small, spindle-shaped, vesicular nuclei. Their cystoplasm is indistinct and scant. These cells are separated by a moderate amount of fibrillar material having an amphoteric tint. There is a pronounced tendency of the cells to be arranged in whorls and nuclear pallisading is easily demonstrated. In many areas spherical or oval masses of tissue are surrounded by a small amount
Fm. 3. A, spherical and oval masses of tissue surrounded by fibrous tissue resembling nerve fibers. (Low power.) B, note large oval cells reminiscent of ganglion cells of sympathetic trunk. Ganglioneuroma.
of fibrous connective tissue so that they have the appearance of nerve fibers. Scattered here and there, sometimes occurring singly and sometimes in groups, are very large cells which vary from oval to round. They have a granular eosinophilic cytoplasm and occasional cytoplasmic processes. Their nuclei vary from one to two in number and are generally eccentric in position. The nuclear membrane is distinct and the nucleolus prominent. Elsewhere the nuclear chromatin is scant. Throughout the sections there are a few scattered eosinophilic leukocytes. Diagnosis: Ganglioneuroma." The tissue removed by resectoscope a year later from nodule on trigone showed: "S-49-185, March 10, 1949. Gross: Three small sections of pale gray tissue (from trigone of bladder). Microscopic: The sections are similar to the previous specimen, being made up of numerous nerve cells and fibers. Diagnosis: Ganglioneuroma." See figure 3. At examination on March 10, 1949, patient was found to have slight dysuria
GANGLIONEUROMA OF BLADDER
531
and occsional pain in the right costovertebral angle, although his general condition was good. Cystoscopic examination, however, revealed a return of the growth at the vesical neck extending into the bladder (fig. 4). The prostatic fossa was of a uniform, slightly oval character such as might be found after a
Frn. 4. View at vesical neck obtained by panendoscope March 10, 1949. Growth has recurred and is covered by bladder mucosa. Note previously resected vesical neck.
Frn. 5. Diagram illustrating findings on bimanual examination under spinal anesthesia.
transurethral resection. Nodular irregularity of the floor of the bladder, including the trigone, the region of the right ureteral orifice, and the left bladder wall, were noted. The former site of the left ureteral orifice could be identified by a gutter along the floor of the bladder. Bimanual palpation under spinal anesthesia showed the outlines of the prostate
532
H. E. WYMAN, B. S. CHAPPELL A.ND W. R. JONES
to be identifiable. There were small, rather firm nodules located under the trigone of the bladder. The seminal vesicles were encompassed in the fibro-elastic field and could not be identified (fig. 5). Biopsies were taken, first from the nodular irregularities over the floor of the bladder and then the small nodule on the left wall of the bladder was biopsied and a third biopsy was taken from the vesical neck itself. Pathological study of the tissue removed revealed findings characteristic of ganglioneuroma. The patient was last examined in June 1949 and there has been great increase in the intravesical tumor and progression of the right hydro-ureter since the examination in March 1949. COMMENT
A 5 year period of observation afforded unusual opportunity in the study of this patient and the case report is given at some length. Of particular interest was the presentation of the patient with obstructive symptomatology referable to bladder and to ureters at the vesico-ureteral junction, almost 4 years before a positive diagnosis of ganglioneuroma could be made. The exact origin of the tumor must remain a point of conjecture but there is certainly ample evidence to show that the tumor eventually invaded the bladder and biopsies readily demonstrating this tumor were obtained by relatively shallow bites into the tumor on the trigone (fig. 5) with the resectoscope loop. Less than a year elapsed after surgical removal of all demonstrable tumor before reappearance of a tumor mass equal in size to the one removed surgically. No evidence of metastasis was found but pressure of the tumor has severely damaged his urinary tract with loss of one kidney and damage to the remaining kidney. Impairment of sexual ability is a feature not fully explained. SUMMARY
A case report of a ganglioneuroma of the bladder in a patient 22 years of age is reported for purposes of record. The patient was closely observed for the 5 year period since onset of symptoms. Symptoms were present for 4 years before diagnosis was made. Surgical removal of tumor transvesically did not effect a cure. Severe and progressive damage to the urinary tract resulted from pressure obstruction by the tumor. Acknowledgments: The authors wish to express appreciation to Dr. Kenneth M. Lynch of the Medical College of the State of South Carolina and to Dr. E. S. Cardwell for the study and diagnosis of the tissue removed surgically. We express our thanks to Dr. Theodore M. Yates of the Department of Urology, Veterans Administration Hospital, Columbia, South Carolina, for helpful suggestions and continued interest. REFERENCES ACKERMAN, L. V. AND DEL REGATO, J. A.: Cancer. St. Louis: C. V. Mosby Co., 1947, pp. 482 and 780. BoYD, W"llr.: A Textbook of Pathology. Philadelphia: Lea and Febiger, 1947, 5th ed., pp. 935. MooRE, R. A.: A Textbook of Pathology. Philadelphia: W. B. Saunders Co., 1945. STOUT, A. P.: Ganglioneuroma of sympathetic nervous system. Surg., Gynec. & Obst., 84: 101-110, 1947. YouNG, H. H. AND DAVIS, D. M.: Young's Practice of Urology. Philadelphia: W. B. Saunders Co., 1926, vol. 1.