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Benign Bladder Tumor in a Two Year, Nine Month Old Child1
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BENIGN BLADDER TUMOR IN A TWO YEAR, NINE MONTH OLD CHILD 1 GARDNER R. RIDLON
Bladder tumors in infancy are rare. Deming (1924) made an exhaustive research of the literature concerning this disease in the first decade of life, and discovered only 64 cases reported, the earliest in 1791. He added 2 cases of his own. Of these 66 cases only 18 patients were in their second year. In 1937, Rathbun published a paper which gave a resume of Deming's review, and added 1 case of his own with eight others during the past 13 years, making a total of 75 cases. To this series I wish to add 1 case of a benign tumor of the bladder in a child 2 years, 9 months old. Case report. In the latter part of September 1936, the mother of a 2 year, 9 months child was referred to me for advice. She gave an exact history of
her child's condition. The child had had a tonsillectomy, but, except for that, had been very healthy. The only relevant part of the family history was that the maternal grandmother had carcinoma of the breast. The present history was as follows: On May 30, 1936, while on a camping trip the child began having marked frequency about every half hour. From then on she began to wet the bed at night. Their family physician was consulted and medication was instituted. At this time no blood was found in the urine. While under treatment an improvement of the frequency was noted for short intervals, but the nocturia persisted. About 2 weeks after the onset the mother noticed that there was considerable straining during the act of urination. Nearly 2 months from the onset a gross hematuria with terminal dysuria was present. This became progressively worse even under treatment. About 2 weeks before I was consulted it was so bad that her physician instilled argyrol at midnight. During this period of treatment no pus cells were found in the urine but much blood was present in each specimen. Examination at the office, November 2, 1936, revealed a rosy-cheeked, robust, well nourished child who did not appear ill. Physical examination was negative throughout. Under intravenous anesthesia a child's cystoscope was passed and 60 cc. of bloody urine evacuated. The bladder was irrigated until the return· fl.ow was clear of blood. On inspection of the bladder a large tumor mass was seen in the region of the right ureter. As it was impossible to obtain a good view of this mass so a No. 18 cystoscope was carefully introduced. The tumor appeared very large, covering the right ureteral orifice and extend1 Read before the annual meeting of the Western Branch Society of the American Urological Association, Los Angeles, Calif., May 13, 1938. 173
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GARDNER R. RIDLON
ing over the right wall of the bladder. From this observation I gained the impression that this tumor was on a pedicle. A cystogram and airogram were taken. A tumor mass could be seen as a filling defect in both films. Being unable to get a biopsy I considered this tumor malignant, and advised open operation with fulguration and implantation of radium emanation seeds. Intravenous pyelograms were not taken at this time, although I advised that they be done. The patient entered Maynard Hospital November 5, 1936. Clinical laboratory :findings. Urine: albumin 11 per cent, many red blood cells, no white blood cells, no casts. Blood count: hemoglobin 80 per cent, erythrocytes 4,150,000, leucocytes 9,000, lymphocytes 20 per cent, neutrophiles 80; coagulation time 2½ minutes. Operation, November 6, under general anesthesia. With the radio knife a suprapubic incision was made to the aponeurosis. This was cut and the muscles separated. The bladder was filled with water and the peritoneum was retracted. The bladder was picked up with Allis clamps and opened with the radio knife. A large tumor was exposed which looked like a bunch of grapes lying over the right ureteral orifice and covering half of the right bladder wall. The base of the tumor was about the same size as the tumor itself. A number of pieces were removed from the tumor, and the remaining tissue was then thoroughly fulgurated. Nine 1.5 millicurie radium emanation seeds were implanted in such a manner that the entire base plus one-half a centimeter around it was irradiated. A Pezzer catheter was placed and the bladder closed with No. I plain gut sutures. A small Penrose drain was placed in the space of Retzius. The aponeurosis was closed with No. I chromic gut and the skin with silkworm sutures and skin clips. Pathological report (Dr. C. R. Jensen, pathologist at Maynard Hospital): Specimen consists of several pieces of tissue, altogether about 2 grams. Two of the pieces, the largest about 1.0 cm. in diameter, are soft and pink with a smooth, glistening surface. Other pieces appear to form part of a papillomatous mass, which has possessed many small villous-like projections 1 or 2 mm. in diameter; this tissue is firmer and darker red. One small piece of tissue is labeled as coming from the base and is sectioned separately. Frozen sections of one of the first described portions are interpreted as a non-malignant papilloma. "Sections are taken for examination from multiple portions of this tissue. Essentially the same changes are present in each piece. They consist of portions of some papillary formation composed of both stroma and of epithelial covering. Almost all of the tissue consists of stroma, and this dominates the histopathology of this neoplasm. It is very soft, moderately well vascularized, and formed by actively growing fibroblasts. In most places these are moderately compact but in many places the cells are widely separated by an inter-
BENIGN BLADDER TUMOR IN CHILD
175
cellular edema; this gives the appearance, in places, of a quasi-myxomatous texture. Where sections of the surface are available, it is covered by a thin layer of transitional epithelium in a few places, although in most places the growth is denuded and the surface is, in reality, formed by the stromal tissue with very widely dilated capillaries in these regions. In a few places there are narrow crypt-like extensions from the surface into the depth of the tissue, lined with the same type of epithelium; in a few places there are small enclosed tubule-like formations which represent the transected end of these crypts. In two of the sections made from this tissue there are small nests of hyperplastic epithelial cells which appear to be buried within the depth of the tissue; however, the tissue surrounding them is not the normal stroma but rather a disintegrated type of fibrous tissue and blood; so that I deduce from this that these formations do not represent true neoplastic invasion of tissue by the epithelial cells, but merely buds of epithelium which have been sectioned in a partially tangential manner so as to give the appearance of being imbedded within the depth of the tissue. From the sections available there is nothing malignant about the appearance of this neoplasm. The primary tissue concerned in the neoplastic formation is the stroma. Diagnosis: Benign fibro-papilloma of the urinary bladder. The child's convalescence was uneventful except for urethral tenesmus until the sixteenth post-operative day. Her temperature was 103 degrees, she had a slight chill and pain in the right back, showing symptoms of right renal blockage. She was somewhat toxic for a few days, but under forced fluids soon returned to normal. About 2 weeks later she had a similar attack at which time a transfusion of 250 cc of citrated blood was given. Since then she has had no acute attacks of pyelonephritis, but for about 9 months her temperature had been around 99.8 degrees daily. Many pus cells with clumps were found in her urine during this time. Small doses (20 grains daily) of sulfanilamide were given for about 2 weeks and since then her temperature and urine have been normal. She has passed seven of the nine radium seeds implanted, five were recovered. Intravenous pyelograms taken January 1937, November 1937 and April 1938 show a marked blockage of the right ureter which has gradually decreased. The irregularity of the right wall of the bladder was also apparent, probably due to scar formation. At the present time she often voids as much as 6 ounces and has no nocturia.
Symptomatology. Increasing frequency with nocturia, which had previously been under control, also dysuria and hematuria are the outstanding symptoms. Diagnosis. With our present armament, the cystoscope, x-ray and
176
GARDNER R. RIDLON
intravenous pyelograms, there is no necessity to fail in this diagnosis. Whenever anyone, regardless of age, complains of a gross hematuria, or after the findings show a microscopic hematuria, I believe a complete urinary investigation is indicated. No bladder tumors in infants had been diagnosed before operation previous to 1924. This probably is due to the fact that infant cystoscopes were not as common or as perfected as that time. Mortality. Of the 75 cases reported, only 7 patients have been reported cured. Of these seven cured patients, no follow-up has been recorded for a longer period than 2 years (2 cases). The pathological diagnoses of the cases cured were: Fibroma, 2 cases; fibrosarcoma, 1 case; fibromyoma, 1 case; polypoid, 1 case; sarcoma, 1 case; polyp, 1 case; telangiectatic fibrous polyp, 1 case. The mortality is high (90 per cent) since nearly all bladder tumors found in infancy are of a high type of malignancy. Pathology. The pathological diagnosis of the 75 tumors reported is as follows: sarcoma, 38 cases; myoma, 16 cases; myxosarcoma, 4 cases; neurogenic sarcoma, 1 case; fibroma, 5 cases; fibromyoma, 1 case; neurofibroma, 1 case; myoma, 1 case; rhabdomyoma, 2 cases; polyps (benign), 2 cases; dermoid, 1 case; papilloma, 1 case; hemanogimoma, 2 cases; fibropapilloma, 1 case. Treatment. Surgery is the only procedure for attacking this type of case; open operation is indicated. Children stand cystotomy very well. When a diagnosis of bladder tumor is made in a child I believe it should be considered malignant. If the tumor is operable my procedure would be to use both fulguration and implantation of radium emanation seeds. 1215 Fourth Ave., Seattle, Wash. REFERENCES DEMING, CLYDE L.: Tumors of bladder in first decade of life. Surg., Gynec. and Obst., 39: 432, 1924. Koss, I. H.: Neurofibromatosis of bladder. Am. J. Dis. Child., 44: 1040, 1934. RATHBUN, N. P.: Primary bladder tumors in infant and young children. Surg., Gynec and Obst., 64: 914, 1937.
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BENIGN BLADDER TUMOR IN A TWO YEAR, NINE MONTH OLD CHILD 1 GARDNER R. RIDLON
Bladder tumors in infancy are rare. Deming (1924) made an exhaustive research of the literature concerning this disease in the first decade of life, and discovered only 64 cases reported, the earliest in 1791. He added 2 cases of his own. Of these 66 cases only 18 patients were in their second year. In 1937, Rathbun published a paper which gave a resume of Deming's review, and added 1 case of his own with eight others during the past 13 years, making a total of 75 cases. To this series I wish to add 1 case of a benign tumor of the bladder in a child 2 years, 9 months old. Case report. In the latter part of September 1936, the mother of a 2 year, 9 months child was referred to me for advice. She gave an exact history of
her child's condition. The child had had a tonsillectomy, but, except for that, had been very healthy. The only relevant part of the family history was that the maternal grandmother had carcinoma of the breast. The present history was as follows: On May 30, 1936, while on a camping trip the child began having marked frequency about every half hour. From then on she began to wet the bed at night. Their family physician was consulted and medication was instituted. At this time no blood was found in the urine. While under treatment an improvement of the frequency was noted for short intervals, but the nocturia persisted. About 2 weeks after the onset the mother noticed that there was considerable straining during the act of urination. Nearly 2 months from the onset a gross hematuria with terminal dysuria was present. This became progressively worse even under treatment. About 2 weeks before I was consulted it was so bad that her physician instilled argyrol at midnight. During this period of treatment no pus cells were found in the urine but much blood was present in each specimen. Examination at the office, November 2, 1936, revealed a rosy-cheeked, robust, well nourished child who did not appear ill. Physical examination was negative throughout. Under intravenous anesthesia a child's cystoscope was passed and 60 cc. of bloody urine evacuated. The bladder was irrigated until the return· fl.ow was clear of blood. On inspection of the bladder a large tumor mass was seen in the region of the right ureter. As it was impossible to obtain a good view of this mass so a No. 18 cystoscope was carefully introduced. The tumor appeared very large, covering the right ureteral orifice and extend1 Read before the annual meeting of the Western Branch Society of the American Urological Association, Los Angeles, Calif., May 13, 1938. 173
174
GARDNER R. RIDLON
ing over the right wall of the bladder. From this observation I gained the impression that this tumor was on a pedicle. A cystogram and airogram were taken. A tumor mass could be seen as a filling defect in both films. Being unable to get a biopsy I considered this tumor malignant, and advised open operation with fulguration and implantation of radium emanation seeds. Intravenous pyelograms were not taken at this time, although I advised that they be done. The patient entered Maynard Hospital November 5, 1936. Clinical laboratory :findings. Urine: albumin 11 per cent, many red blood cells, no white blood cells, no casts. Blood count: hemoglobin 80 per cent, erythrocytes 4,150,000, leucocytes 9,000, lymphocytes 20 per cent, neutrophiles 80; coagulation time 2½ minutes. Operation, November 6, under general anesthesia. With the radio knife a suprapubic incision was made to the aponeurosis. This was cut and the muscles separated. The bladder was filled with water and the peritoneum was retracted. The bladder was picked up with Allis clamps and opened with the radio knife. A large tumor was exposed which looked like a bunch of grapes lying over the right ureteral orifice and covering half of the right bladder wall. The base of the tumor was about the same size as the tumor itself. A number of pieces were removed from the tumor, and the remaining tissue was then thoroughly fulgurated. Nine 1.5 millicurie radium emanation seeds were implanted in such a manner that the entire base plus one-half a centimeter around it was irradiated. A Pezzer catheter was placed and the bladder closed with No. I plain gut sutures. A small Penrose drain was placed in the space of Retzius. The aponeurosis was closed with No. I chromic gut and the skin with silkworm sutures and skin clips. Pathological report (Dr. C. R. Jensen, pathologist at Maynard Hospital): Specimen consists of several pieces of tissue, altogether about 2 grams. Two of the pieces, the largest about 1.0 cm. in diameter, are soft and pink with a smooth, glistening surface. Other pieces appear to form part of a papillomatous mass, which has possessed many small villous-like projections 1 or 2 mm. in diameter; this tissue is firmer and darker red. One small piece of tissue is labeled as coming from the base and is sectioned separately. Frozen sections of one of the first described portions are interpreted as a non-malignant papilloma. "Sections are taken for examination from multiple portions of this tissue. Essentially the same changes are present in each piece. They consist of portions of some papillary formation composed of both stroma and of epithelial covering. Almost all of the tissue consists of stroma, and this dominates the histopathology of this neoplasm. It is very soft, moderately well vascularized, and formed by actively growing fibroblasts. In most places these are moderately compact but in many places the cells are widely separated by an inter-
BENIGN BLADDER TUMOR IN CHILD
175
cellular edema; this gives the appearance, in places, of a quasi-myxomatous texture. Where sections of the surface are available, it is covered by a thin layer of transitional epithelium in a few places, although in most places the growth is denuded and the surface is, in reality, formed by the stromal tissue with very widely dilated capillaries in these regions. In a few places there are narrow crypt-like extensions from the surface into the depth of the tissue, lined with the same type of epithelium; in a few places there are small enclosed tubule-like formations which represent the transected end of these crypts. In two of the sections made from this tissue there are small nests of hyperplastic epithelial cells which appear to be buried within the depth of the tissue; however, the tissue surrounding them is not the normal stroma but rather a disintegrated type of fibrous tissue and blood; so that I deduce from this that these formations do not represent true neoplastic invasion of tissue by the epithelial cells, but merely buds of epithelium which have been sectioned in a partially tangential manner so as to give the appearance of being imbedded within the depth of the tissue. From the sections available there is nothing malignant about the appearance of this neoplasm. The primary tissue concerned in the neoplastic formation is the stroma. Diagnosis: Benign fibro-papilloma of the urinary bladder. The child's convalescence was uneventful except for urethral tenesmus until the sixteenth post-operative day. Her temperature was 103 degrees, she had a slight chill and pain in the right back, showing symptoms of right renal blockage. She was somewhat toxic for a few days, but under forced fluids soon returned to normal. About 2 weeks later she had a similar attack at which time a transfusion of 250 cc of citrated blood was given. Since then she has had no acute attacks of pyelonephritis, but for about 9 months her temperature had been around 99.8 degrees daily. Many pus cells with clumps were found in her urine during this time. Small doses (20 grains daily) of sulfanilamide were given for about 2 weeks and since then her temperature and urine have been normal. She has passed seven of the nine radium seeds implanted, five were recovered. Intravenous pyelograms taken January 1937, November 1937 and April 1938 show a marked blockage of the right ureter which has gradually decreased. The irregularity of the right wall of the bladder was also apparent, probably due to scar formation. At the present time she often voids as much as 6 ounces and has no nocturia.
Symptomatology. Increasing frequency with nocturia, which had previously been under control, also dysuria and hematuria are the outstanding symptoms. Diagnosis. With our present armament, the cystoscope, x-ray and
176
GARDNER R. RIDLON
intravenous pyelograms, there is no necessity to fail in this diagnosis. Whenever anyone, regardless of age, complains of a gross hematuria, or after the findings show a microscopic hematuria, I believe a complete urinary investigation is indicated. No bladder tumors in infants had been diagnosed before operation previous to 1924. This probably is due to the fact that infant cystoscopes were not as common or as perfected as that time. Mortality. Of the 75 cases reported, only 7 patients have been reported cured. Of these seven cured patients, no follow-up has been recorded for a longer period than 2 years (2 cases). The pathological diagnoses of the cases cured were: Fibroma, 2 cases; fibrosarcoma, 1 case; fibromyoma, 1 case; polypoid, 1 case; sarcoma, 1 case; polyp, 1 case; telangiectatic fibrous polyp, 1 case. The mortality is high (90 per cent) since nearly all bladder tumors found in infancy are of a high type of malignancy. Pathology. The pathological diagnosis of the 75 tumors reported is as follows: sarcoma, 38 cases; myoma, 16 cases; myxosarcoma, 4 cases; neurogenic sarcoma, 1 case; fibroma, 5 cases; fibromyoma, 1 case; neurofibroma, 1 case; myoma, 1 case; rhabdomyoma, 2 cases; polyps (benign), 2 cases; dermoid, 1 case; papilloma, 1 case; hemanogimoma, 2 cases; fibropapilloma, 1 case. Treatment. Surgery is the only procedure for attacking this type of case; open operation is indicated. Children stand cystotomy very well. When a diagnosis of bladder tumor is made in a child I believe it should be considered malignant. If the tumor is operable my procedure would be to use both fulguration and implantation of radium emanation seeds. 1215 Fourth Ave., Seattle, Wash. REFERENCES DEMING, CLYDE L.: Tumors of bladder in first decade of life. Surg., Gynec. and Obst., 39: 432, 1924. Koss, I. H.: Neurofibromatosis of bladder. Am. J. Dis. Child., 44: 1040, 1934. RATHBUN, N. P.: Primary bladder tumors in infant and young children. Surg., Gynec and Obst., 64: 914, 1937.