Ala-Opas M

Giordano S

Sankila A

Taskinen S

Scandinavian Journal of Urology and Nephrology

English

UTUPub

CASE REPORTAdenocarcinoma in isolated rectal bladder after treatment ofbladder exstrophySALVATORE GIORDANO1, SEPPO TASKINEN1, ANNA SANKILA2 &MARTTI ALA-OPAS3Divisions of 1Pediatric Surgery, 2Pathology and 3Urology, Helsinki University Hospital, Helsinki, FinlandAbstractWe report the first Finnish patient with carcinoma in an augmented intestinal bladder, where urine and stools are not incontact. The patient had undergone rectal bladder reconstruction at the age of 2 years because of bladder exstrophy. Whenthe patient was aged 46 years, a 2-cm, papillar, well-differentiated adenocarcinoma was detected and removed, preservingthe rectal bladder.Key Words: Carcinoma, bladder exstrophy, bladder augmentation, bladder replacement, enterocystoplasty, rectal bladderIntroductionStones, metabolic acidosis, infections, perforationsand carcinoma are well-recognized complications ofan augmented intestinal bladder [1]. However, therisk of carcinoma is not yet known and we feel it is ofutmost importance to report all cases of carcinomain intestinal bladders. Herein we report the firstpatient in Finland to develop carcinoma in anintestinal bladder, in which urine and stools arenot in contact.Case reportAt the age of 2 years, the bladder of a male patientwas removed and the rectal bladder reconstructedbecause of bladder exstrophy. During the operationthe ureters were connected to the rectum. Therectum itself was divided and was left as a urinarybladder. The distal end of the sigma was brought tothe ventral side of the anus through the commonsphincter mechanism [2]. At the end of the opera-tion the patient had two anal orifices: the ventral onefor stools and the dorsal one for urine. Stools andurine were not in contact. The patient experiencedoccasional bacteriuria, but apparently he did nothave symptomatic urinary tract infections duringchildhood. The patient was satisfied with his urinarycontinence and with the ability to void sponta-neously. Because of the anal stricture he neededoccasional enemas in order to defecate.At the age of 42 years the patient started toexperience recurrent pyelonephritis with left flankpain. Scintigraphy revealed that the function dis-tribution was 70% on the right side and 30% on theleft. An obstruction was observed in the left recto-ureteral junction and was treated with antegradeballoon dilatation. No endoscopy of the rectalbladder was performed on that occasion.At the age of 46 years the patient was admittedfrom another part of the country to the Hospital forChildren and Adolescents, Helsinki, where theoriginal operations were performed, to discuss theprimary diagnosis and the childhood operations. Itappeared that the patient had undergone no endo-scopic follow-up of the rectal bladder. During thesame visit, endoscopy of the rectal bladder revealed a2-cm papillary tumor. At biopsy a well-differentiatedadenocarcinoma was detected, but there was noevidence of tumor invasion or metastases (Figure 1).The tumor was removed in the adult urology unitwith rectum-preserving electroresection.Correspondence: Seppo Taskinen, MD, PhD, Hospital for Children and Adolescents, University of Helsinki, Stenbäckinkatu 11, 00290 Helsinki, Finland. Tel:358 50 4272542. Fax: 358 9 47175314. E-mail: seppo.taskinen@hus.fiScandinavian Journal of Urology and Nephrology, 2008; 42: 392394(Received 5 September 2007; accepted 7 December 2007)ISSN 0036-5599 print/ISSN 1651-2065 online # 2008 Informa UK Ltd. (Informa Healthcare, Taylor & Francis AS)DOI: 10.1080/00365590701871575Scand J Urol Nephrol Downloaded from informahealthcare.com by Tykslab on 01/13/15For personal use only.During a 3-year follow-up period, in addition to acouple of CT scans, the patient underwent endo-scopy of the rectal bladder, urine cytology andplasma carcinoembryonic antigen controls every 4months during the first year and every 6 monthsthereafter. At the last follow-up session in June 2007there was no evidence of tumor recurrence, noincontinence and the patient was still able to voidspontaneously.DiscussionThe reported risk of carcinoma after ureterosigmoi-dostomy (in which stools and urine mix) variesbetween 6% and 29% and the average intervalbetween ureterocolostomy and the development oftumor is 2026 years [3]. Carcinoma may resultfrom the nitrosoamides produced by bacterial actionin the mixture of urine and stools [4] or from activeoxygen radicals that are produced by phagocytic cellsas a result of inflammation at the anastomotic site[5].During the last 30 years, intestinal bladderaugmentation has largely replaced ureterosigmoi-dostomy. One reason for this has been to preventurine and stools from mixing in the hope ofreducing the risk of carcinoma. However, carcino-mas have also been reported after intestinal bladderaugmentation. The suggested risk of carcinoma inan augmented bladder is 3.6% after 25 years [6]. Todate 30 cases of carcinoma have been published,consisting mostly of adenocarcinomas or transitionalcell carcinomas [6]. Because of the long latencyperiod for the development of cancer the riskremains uncertain. Stools and urine were not mixingin our patient and the situation was very similar tothat after bladder augmentation or replacementnowadays.In 1944 the incidence of bladder carcinoma inbladder exstrophy was estimated to be 4% [7]. Itseems that successful operations to close the exstro-phied bladder at an early age prevent carcinoma. Inrecent decades, bladder carcinoma in bladder ex-strophy has become rare. Bladder reconstruction,however, is not always successful enough to preventincontinence and 070% of children need bladderaugmentation or replacement [8].In our case, the patient was satisfied with hisurinary continence and with the ability to voidspontaneously. He was against complete resectionof the rectal bladder and did not like the idea of anew intestinal bladder with a continent stoma.Because there was no sign of invasion and theadenocarcinoma was well differentiated, organ-spar-ing surgery was considered sufficient [9]. During a3-year follow-up period there has been no sign oftumor recurrence.Reported tumor cases have been rare after intest-inal bladder augmentation or replacement, but theirincidence is probably increasing. Currently, directvisualization by means of endoscopy is the onlyreliable method for finding carcinoma inside theintestinal bladder. A lifetime annual surveillanceprogram, including cystoscopy and cytology, isrecommended, starting 810 years after the aug-mentation [3,10]. Currently, in our institution westart cystoscopy controls earlier, so as to alsomonitor benign conditions such as stones.References[1] Gough DCS. Enterocystoplasty. BJU Int 2001;/88:/73943.[2] Barnes RW, Hill MR Sr, Hill MR Jr, Hill JT. Urinarydiversion through an isolated rectal bladder with intrasphinc-teric (anal) colostomy. Dis Colon Rectum 1958;/1:/48590.Figure 1. (A) Low-magnification image showing the polypoidarchitecture of the tumor. Histology is that of an adenocarcinomaof intestinal type. (B) High-magnification image showing markedatypia of the neoplastic epithelial cells and numerous mitoses.Adenocarcinoma in rectal bladder 393Scand J Urol Nephrol Downloaded from informahealthcare.com by Tykslab on 01/13/15For personal use only.[3] Azimuddin K, Khubchandani IT, Stasik JJ, Rosen L, ReitherRD. Neoplasia after ureterosigmoidostomy. Dis ColonRectum 1999;/42:/16328.[4] Husmann DA, Spence HM. Current status of tumor of thebowel following ureterosigmoidostomy; a review. J Urol1990;/144:/60710.[5] Weitzman SA, Weitberg AB, Clark EP, Stossel TP. Phago-cytes as carcinogens: malignant transformation produced byhuman neutrophils. Science 1985;/227:/12313.[6] Soergel TM, Cain MP, Misseri R, Gardner TA, Koch MO,Rink RC. Transitional cell carcinoma of the bladder follow-ing augmentation cystoplasty for the neuropathic bladder. JUrol 2004;/172:/164952.[7] Wattenberg CA, Beare JB, Tormey AR. Exstrophy of theurinary bladder complicated by adenocarcinoma. J Urol1956;/75:/58393.[8] Woodhouse CRJ, North AC, Gearhart JP. Standing the testof time: long-term outcome of reconstruction of the ex-strophy bladder. World J Urol 2006;/24:/2449.[9] Rossi H, Rothenberger DA. Surgical treatment of coloncancer. Surg Oncol Clin N Am 2006;/15:/10927.[10] Metcalfe PD, Cain MP, Kaefer M, Gilley DA, Meldrum KK,Misseri R, et al. What is the need for additional bladdersurgery after bladder augmentation in childhood? J Urol2006;/176:/18015.394 S. Giordano et al.Scand J Urol Nephrol Downloaded from informahealthcare.com by Tykslab on 01/13/15For personal use only.

Adenocarcinoma in isolated rectal bladder after treatment of bladder exstrophy

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Adenocarcinoma in isolated rectal bladder after treatment of bladder exstrophy

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