Ci:A SREE P O R T S
GIANT SCLEROSING LEIOMYOMA OF BLADDER PRESENTING AS CHRONIC RENAL FAILURE B. DELAHUNT, F.R.C.EA. J. N. NACEY, F.R.A.C.S. A. F. FERGUSON, ER.A.C.S., F.R.C.S. From the Departments of Pathology and Urology, Wellington School of Medicine, University of Otago, Wellington, New Zealand
ABSTRACT--The clinical and pathologic features of a case of giant sclerosing leiomyoma of ii~ bladder in a male Polynesian patient are reported. The presenting complication of chronic r ~ failure due to tumor envelopment of both ureters and consequent bilateral renal obstructioff ~ pears to be unique in the literature.
Leiomyoma is a rare variety of benign tumor of the bladder.1 This type of neoplasm is usually associated with such presenting symptoms as urinary frequency, abdominal pain or swelling, or decreased urinary flow secondary to ball valve blockage of the proximal urethra. 2 Previous reports deseribe unilateral ureterie obstruction due to compression by tumor 3 or displaeement of the ureterie orifiee. 4,~ The ease described herein appears to be the first reeorded instance where a leiomyoma has eneroaehed on and enveloped both ureters resulting in ureterie obstruetion w i t h c o n s e q u e n t bilateral hydroureter and hydronephrosis. Case Report The patient, a forty-two-year-old male Samoan taro planter, was referred from Western Samoa with a fifteen-month history of right loin pain associated with weight loss and increasing lethargy. An intravenous pyelogram taken on presentation fifteen months previously, had shown a nonfunetioning right kidney and hydronephrosis of the left kidney with ureterie dilatation. No further investigations were carried out until he was transferred to New Zealand with evidence of progressive renal failure. On admission he was noted to have a bladder palpable to the umbilicus with an enlarged left
270
kidney. Hematologi. pronounced anemia mild hypercaleemi~ 2.33 mmol/L, and I; L. An ultrasound ex~ hydronephrosis and l renal cortex on bot grossly enlarged ant completely filled wit A cystogram showe from the bladder w posterior wall, and any urethral obstru~ the mass, eomputer: formed whieh show mor beyond the bla. g a t i o n also eol~ hydronephrosis (Fig. adenopathy. Trucut : specimen showed s( and sheets of mature sue. The patient unc a laparotomy was p, At operation a la found arising from Both ureterie orifice the tumor, and the b] tended over the tum, easily enucleated alo and was reseeted alo
UROLOGY
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MARCH 1991
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VOLUME XXXVII,
F I c u ~ 3. Bisected surgical specimen shows encapsulated tumor consisting of interlacing whorls of connective tissue. Needle track is seen within specimen on leCt (magnification bar = 5 cm).
FmuaE 4. Photomicrograph of leiomyoma showing sheets of smooth muscle cells with dense collagenous stroma (hematoxylin and eosin; original magnification x 400). Ladder. The two ureters -he t u m o r and required The patient m a d e an te continues on hemoi renal transplantation. ,'n was a multiloculated ss weighing 1,840 g and Laximum extent (Fig. 3). t u m o r showed sheets of ranged in fascicles and bands of collagen (Fig. m a l degree of nuclear otic figures were not obas invested by a thick
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M A R C H 1991
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pseudocapsule, and there was no evidence of capsule invasion. The features were those of a selerosing leiomyoma. Comment L e i o m y o m a of the bladder is a rare t u m o r with less t h a n 200 eases being reported in the literature. These are usually 5 cm or less in diameter at time of presentation, with tumors greater t h a n 10 em in m a x i m u m extent being only rarely recorded. 5,6 The tumors m a y be subserosal, submueosal, or intramural in location and are usually situated in the vicinity of the trigone. 1 Approximately 60 percent of tumors are found to project into the bladder lumen
V O L U M E XXXVII, N U M B E R 3
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forming a space-occupying lesion. 1 Presenting symptoms are variable with symptoms of bladder instability or flank pain being most frequently reported. Occasionally the endovesieal portion of the tumor is peduneulated or dumbbell-shaped, and may project into the proximal urethra producing bladder neck outlet obstruction and consequent hydronephrosis. 2 Ureterie obstruction also has been reported with the tumor extending up to envelop the ureterie orifice. 3 In those eases where adequate clinical and pathologic details are reported, it appears that the ureterie obstruction is unilateral, and to our knowledge no ease of bilateral involvement of the ureters has been described. Previous reports have varied as to the most suitable method of treatment for vesieal leiomyoma. 1 The general consensus would suggest that surgical excision with reconstruction of the bladder is the treatment of choice. As these tumors usually present in a much smaller size than the example recorded in this report, enueleation is often a simple procedure and total or partial eysteetomy, as necessitated in the few reported eases of giant leiomyoma, is avoided. It has been suggested that since these tumors follow a benign course and no instance of malignant change within the tumor has been found, it is appropriate to treat these eases conservatively, v In eases where surgical excision was difficult, it was thought that adequate followup would be sufficient following needle biopsy to confirm the benign nature of the lesion. While there is no evidence for the development of sareomatous transformation, there is no doubt that leiomyomas gradually increase in size. The degree of sclerosis noted in this ease is evidence of the antiquity of giant leiomyomas, since this form of degeneration is frequently seen in large uterine leiomyomas and is thought to occur as the tumor slowly outgrows its blood supply, s Benign tumors are characteristically
272
encapsulated with the capsu pressure atrophy and fibrosis tures. 9 As the tumors increase phy and fibrosis may involve t ing in displacement or obstruc be distinguished from direct i ureters as is frequently see smooth muscle tumors. The development of urinary trae volving either the ureters dire, tion of the tumor into the urett sufficient indication for surgie these cases. Continued followcarried out since recurrence fe total tumor removal has been Wellington Wellin:
(
References 1. Vargas A.D, and Mendez R: Leiomy, 21:308 (1983). 2. Petraeeo S, et ah Considerations re~ oma of the urinary bladder, Int Urol Ne 3. Bornstein I, Charboneau JW, a n d oma of the bladder: sonographic and uro~ sound Med 5:407 (1"986). 4. Illescas FF, Baker ME, and Weine: oma: advantages of sonography over corn ttadiol 8:216 (1986). 5. Bramwell SP, Pitts J, Goudie SE leiomyoma of the bladder, Br J Urol 60: 6. Campbell EW, and Gislason GJ: Bc of the urinary bladder: review of the lite ease of leiomyoma, J Urol 70:733 (1953[ 7. Belis JA, Post GJ, Rochman SC, tourinary leiomyomas, Urology 13:424 8. Persaud V, and Arjoon PD: Uterine degenerative change and a correlatio~ Obstet Gyneeol 35:432 (1970). 9. Anderson J1q: Muir's Textbook of P~ don, Edward Arnold, 1985, p 12.9. 10. Chavaz CA, and Nero M: Multi urinary bladder, J Kans Med See 85: 29i 11. Lake MH, Kossow AS, and Bokins bladder and urethra, J UroI 125:742 (1981).
oLocYJ
1991,
FOLUM
XX
VII
GIANT SCLEROSING LEIOMYOMA OF BLADDER PRESENTING AS CHRONIC RENAL FAILURE B. DELAHUNT, F.R.C.EA. J. N. NACEY, F.R.A.C.S. A. F. FERGUSON, ER.A.C.S., F.R.C.S. From the Departments of Pathology and Urology, Wellington School of Medicine, University of Otago, Wellington, New Zealand
ABSTRACT--The clinical and pathologic features of a case of giant sclerosing leiomyoma of ii~ bladder in a male Polynesian patient are reported. The presenting complication of chronic r ~ failure due to tumor envelopment of both ureters and consequent bilateral renal obstructioff ~ pears to be unique in the literature.
Leiomyoma is a rare variety of benign tumor of the bladder.1 This type of neoplasm is usually associated with such presenting symptoms as urinary frequency, abdominal pain or swelling, or decreased urinary flow secondary to ball valve blockage of the proximal urethra. 2 Previous reports deseribe unilateral ureterie obstruction due to compression by tumor 3 or displaeement of the ureterie orifiee. 4,~ The ease described herein appears to be the first reeorded instance where a leiomyoma has eneroaehed on and enveloped both ureters resulting in ureterie obstruetion w i t h c o n s e q u e n t bilateral hydroureter and hydronephrosis. Case Report The patient, a forty-two-year-old male Samoan taro planter, was referred from Western Samoa with a fifteen-month history of right loin pain associated with weight loss and increasing lethargy. An intravenous pyelogram taken on presentation fifteen months previously, had shown a nonfunetioning right kidney and hydronephrosis of the left kidney with ureterie dilatation. No further investigations were carried out until he was transferred to New Zealand with evidence of progressive renal failure. On admission he was noted to have a bladder palpable to the umbilicus with an enlarged left
270
kidney. Hematologi. pronounced anemia mild hypercaleemi~ 2.33 mmol/L, and I; L. An ultrasound ex~ hydronephrosis and l renal cortex on bot grossly enlarged ant completely filled wit A cystogram showe from the bladder w posterior wall, and any urethral obstru~ the mass, eomputer: formed whieh show mor beyond the bla. g a t i o n also eol~ hydronephrosis (Fig. adenopathy. Trucut : specimen showed s( and sheets of mature sue. The patient unc a laparotomy was p, At operation a la found arising from Both ureterie orifice the tumor, and the b] tended over the tum, easily enucleated alo and was reseeted alo
UROLOGY
/
MARCH 1991
/
VOLUME XXXVII,
F I c u ~ 3. Bisected surgical specimen shows encapsulated tumor consisting of interlacing whorls of connective tissue. Needle track is seen within specimen on leCt (magnification bar = 5 cm).
FmuaE 4. Photomicrograph of leiomyoma showing sheets of smooth muscle cells with dense collagenous stroma (hematoxylin and eosin; original magnification x 400). Ladder. The two ureters -he t u m o r and required The patient m a d e an te continues on hemoi renal transplantation. ,'n was a multiloculated ss weighing 1,840 g and Laximum extent (Fig. 3). t u m o r showed sheets of ranged in fascicles and bands of collagen (Fig. m a l degree of nuclear otic figures were not obas invested by a thick
/
M A R C H 1991
/
pseudocapsule, and there was no evidence of capsule invasion. The features were those of a selerosing leiomyoma. Comment L e i o m y o m a of the bladder is a rare t u m o r with less t h a n 200 eases being reported in the literature. These are usually 5 cm or less in diameter at time of presentation, with tumors greater t h a n 10 em in m a x i m u m extent being only rarely recorded. 5,6 The tumors m a y be subserosal, submueosal, or intramural in location and are usually situated in the vicinity of the trigone. 1 Approximately 60 percent of tumors are found to project into the bladder lumen
V O L U M E XXXVII, N U M B E R 3
271
forming a space-occupying lesion. 1 Presenting symptoms are variable with symptoms of bladder instability or flank pain being most frequently reported. Occasionally the endovesieal portion of the tumor is peduneulated or dumbbell-shaped, and may project into the proximal urethra producing bladder neck outlet obstruction and consequent hydronephrosis. 2 Ureterie obstruction also has been reported with the tumor extending up to envelop the ureterie orifice. 3 In those eases where adequate clinical and pathologic details are reported, it appears that the ureterie obstruction is unilateral, and to our knowledge no ease of bilateral involvement of the ureters has been described. Previous reports have varied as to the most suitable method of treatment for vesieal leiomyoma. 1 The general consensus would suggest that surgical excision with reconstruction of the bladder is the treatment of choice. As these tumors usually present in a much smaller size than the example recorded in this report, enueleation is often a simple procedure and total or partial eysteetomy, as necessitated in the few reported eases of giant leiomyoma, is avoided. It has been suggested that since these tumors follow a benign course and no instance of malignant change within the tumor has been found, it is appropriate to treat these eases conservatively, v In eases where surgical excision was difficult, it was thought that adequate followup would be sufficient following needle biopsy to confirm the benign nature of the lesion. While there is no evidence for the development of sareomatous transformation, there is no doubt that leiomyomas gradually increase in size. The degree of sclerosis noted in this ease is evidence of the antiquity of giant leiomyomas, since this form of degeneration is frequently seen in large uterine leiomyomas and is thought to occur as the tumor slowly outgrows its blood supply, s Benign tumors are characteristically
272
encapsulated with the capsu pressure atrophy and fibrosis tures. 9 As the tumors increase phy and fibrosis may involve t ing in displacement or obstruc be distinguished from direct i ureters as is frequently see smooth muscle tumors. The development of urinary trae volving either the ureters dire, tion of the tumor into the urett sufficient indication for surgie these cases. Continued followcarried out since recurrence fe total tumor removal has been Wellington Wellin:
(
References 1. Vargas A.D, and Mendez R: Leiomy, 21:308 (1983). 2. Petraeeo S, et ah Considerations re~ oma of the urinary bladder, Int Urol Ne 3. Bornstein I, Charboneau JW, a n d oma of the bladder: sonographic and uro~ sound Med 5:407 (1"986). 4. Illescas FF, Baker ME, and Weine: oma: advantages of sonography over corn ttadiol 8:216 (1986). 5. Bramwell SP, Pitts J, Goudie SE leiomyoma of the bladder, Br J Urol 60: 6. Campbell EW, and Gislason GJ: Bc of the urinary bladder: review of the lite ease of leiomyoma, J Urol 70:733 (1953[ 7. Belis JA, Post GJ, Rochman SC, tourinary leiomyomas, Urology 13:424 8. Persaud V, and Arjoon PD: Uterine degenerative change and a correlatio~ Obstet Gyneeol 35:432 (1970). 9. Anderson J1q: Muir's Textbook of P~ don, Edward Arnold, 1985, p 12.9. 10. Chavaz CA, and Nero M: Multi urinary bladder, J Kans Med See 85: 29i 11. Lake MH, Kossow AS, and Bokins bladder and urethra, J UroI 125:742 (1981).
oLocYJ
1991,
FOLUM
XX
VII
