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Saudi Journal of Kidney Diseases and Transplantation
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Table of Contents   
CASE REPORT  
Year : 2012  |  Volume : 23  |  Issue : 1  |  Page : 122-124
Squamous cell carcinoma in exstrophic unreconstructed urinary bladder in an adult


Department of Urology, Institute of Post- Graduate Medical Education and Research, Kolkata, West Bengal, India

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Date of Web Publication3-Jan-2012
 

   Abstract 

Bladder exstrophy is rare and associated with an increased incidence of bladder cancer. Unreconstructed bladder extrophy presenting in an adult is very rare as most of the patients undergo repair in childhood. Most of the cancers are adenocarcinomas. We report a rare case of squamous cell carcinoma occurring in exstrophic unreconstructed bladder in a 58-year-old male patient.

How to cite this article:
Bansal P, Gupta A, Mongha R, Kundu AK. Squamous cell carcinoma in exstrophic unreconstructed urinary bladder in an adult. Saudi J Kidney Dis Transpl 2012;23:122-4

How to cite this URL:
Bansal P, Gupta A, Mongha R, Kundu AK. Squamous cell carcinoma in exstrophic unreconstructed urinary bladder in an adult. Saudi J Kidney Dis Transpl [serial online] 2012 [cited 2019 Sep 21];23:122-4. Available from: http://www.sjkdt.org/text.asp?2012/23/1/122/91316

   Introduction Top


Bladder exstrophy occurs in approximately one in 35,000 live births and is associated with an increased incidence of bladder cancer. Because the vast majority of patients with bladder exstrophy now undergo reconstruction at a young age, unreconstructed bladder exstrophy in an adult patient is exceedingly rare. Only 113 cases of cancer occurring in unreconstructed exstrophic bladders have been reported. Approximately 90% were adenocarcinomas and 5% were squamous cell carcinomas. We report a rare case of squamous cell carcinoma, occurring in the unreconstructed exstrophic bladder in a 58-year-old man; he was treated with radical cystectomy. This case, to our knowledge, represents the oldest reported patient to present with squamous cell carcinoma of an unreconstructed exstrophic bladder.


   Case Report Top


A 58-year-old man with classic bladder exstrophy who had never undergone a reconstruction earlier presented with ulcerating lesion on the left lateral aspect of his bladder with involvement of the contiguous skin [Figure 1]. The patient was otherwise healthy. His renal function tests were normal. The intravenous pyelogram showed bilateral normal excretory kidneys with no cystogram phase. The characteristic skeletal defects of exstrophy complex such as widening of the pubic symphysis caused by malrotation of the innominate bones in relation to the sagittal plane of the body along both sacroiliac joints were present [Figure 2]. The metastatic work up was negative. Preoperative biopsy of the ulcerative lesion revealed it to be squamous cell carcinoma of of low grade.
Figure 1: Ulcerative lesion seen on the bladder wall along with exstrophy-epispadias complex.

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Figure 2: Widening of the pubic symphysis and malrotation of the innominate bones. Cystogram is not present.

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The patient underwent radical cystectomy and closure of the infra-umbelical defect with Cardiff repair with On-lay mesh repair. [1] Histopathological examination revealed squamous cell carcinoma of low grade with negative lymph nodes. The skin margins were not involved. The patient was not given any adjuvant therapy and is doing well at six months of follow-up.


   Discussion Top


Bladder exstrophy occurs in approximately 1 in 35,000 live births and is associated with an increased incidence of bladder cancer. The reported incidence among exstrophy cohorts varies from 3.3% to 7.5%, which is several times greater than in age-matched controls in the general population. [2],[3] The etiology of this increased incidence remains unclear. The environmental stimuli and chronic infection may cause bladder cells to undergo glandular metaplasia. [4] After excluding patients who underwent reconstruction, only around 113 cases of cancer occurring in exstrophic bladders have been reported thus far. Approximately 90% were adenocarcinomas and 5% were squamous cell carcinomas. [2],[3],[4] These cancers tend to be aggressive, and most patients undergo radical cystectomy as primary therapy. [2],[3]

Because the vast majority of patients with bladder exstrophy now undergo reconstruction at a young age, unreconstructed bladder exstrophy in an adult patient is exceedingly rare. This case also represents, to our knowledge, the oldest patient to present with primary squamous cell carcinoma of an unreconstructed exstrophic bladder. Because the patient had not undergone bladder reconstruction, an exposed, chronically irritated, chronically infected urothelium in direct continuity with squamous epithelium may have predisposed him to the development of squamous carcinoma rather than adenocarcinoma. Squamous metaplasia occurring at the borders of untreated exstrophic bladders has been previously documented.

The orientation of the tumor on final clinical and pathologic examination in our patient showed that the ulcerative lesion was mainly present over the bladder mucosa, thus strongly suggesting the bladder as the primary source; another possible explanation is that the squamous cell carcinoma in such patients represents a skin neoplasm infiltrating the bladder. Chronic inflammation is associated with squamous cell carcinoma of the skin. [5] Chronic irritation and inflammation induced by adjacent, exposed urothelium may be a potential pathogenic stimulus for epidermal carcinogenesis. It is possible that some of the cases of squamous carcinoma previously reported in patients with exstrophy may have been malignancies arising from the skin bordering the bladder.

 
   References Top

1.Shukla VK, Mongha R, Gupta N, Chauhan VS. Incisional hernia-comparison of mesh repair with Cardiff repair: An university hospital experience. Hernia 2005;9(3):238-41.  Back to cited text no. 1
    
2.Smeulders N, Woodhouse CR. Neoplasia in adult exstrophy patients. BJU Int 2001;87(7): 623-8.  Back to cited text no. 2
    
3.Paulhac P, Maisonnette F, Bourg S, Dumas JP, Colombeau P. Adenocarcinomain the exstrophic bladder. Urology 1999;54(4):744.  Back to cited text no. 3
    
4.McIntosh JF, Worley G. Adenocarcinoma arising in exstrophy of the bladder: Report of two cases and review of the literature. J Urol 1955;73(5):820-9.  Back to cited text no. 4
    
5.Gur E, Neligan PC, Shafir R, Reznick R, Cohen M, Shpitzer T. Squamous cell carcinoma in perineal inflammatory disease. Ann Plast Surg 1997;38(6):653-7.  Back to cited text no. 5
    

Top
Correspondence Address:
Anup Kumar Kundu
Head of Department, Department of Urology, IPGMER & SSKM Hospital, Kolkata
India
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PMID: 22237233

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    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures
 

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