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Safety first? Necrotizing cystitis after autoerotic manipulation

To the best of our knowledge, this is the first reported case of  necrotising cystitis caused by the insertion of a foreign body.

 

Authors: S. Knipper, B. Feyerabend, A.J. Gross, T. Bach

Department of Urology, Asklepios Klinik Barmbek, Hamburg, Germany.

 
Corresponding Author: Sophie Knipper MD, Asklepios Klinik Barmbek, Department of Urology, Rübenkamp 220 , 22291 Hamburg/ Germany.   E-mail: [email protected]

 

Abstract
 
We present the case of a 72-year old man with necrotising cystitis. The patient was admitted after having been diagnosed with gross haematuria and an acute abdomen complicated by systemic inflammatory response syndrome (SIRS) and severe hyperglycemia in a peripheral hospital. Work-up included a CT scan that revealed a gas-forming infection of the lower abdomen, most likely caused by a foreign body which appeared to be within the urethra. The patient died within hours of multi-organ failure.
Emphysematous cystitis, in this case complicated by necrosis, is a rare clinical entity, most frequently seen in diabetic women. It can rapidly progress to severe sepsis in the absence of early therapeutic intervention. Conservative management leads to a full recovery in most cases, in some however, further treatment such as partial cystectomy, cystoprostatectomy or surgical debridement is required.

 

Introduction
 
Gas-forming (emphysematous) infection of the urinary tract is a potentially life-threatening condition (1). Emphysematous cystitis is a rare complication of  lower urinary tract infection, and is characterised by air within the bladder wall and lumen (2). Most commonly, it is found in middle-aged diabetic women. Risk factors for complicated urinary tract infections include the presence of an indwelling urethral catheter, urinary tract outlet obstruction, neurogenic bladder  or recurrent urinary tract infection (3). Pre-existing conditions such as diabetes, alcoholism and malnutrition are also described as possible risk factors for emphysematous cystitis (4).
To the best of our knowledge, this is the first reported case of  necrotising cystitis caused by the insertion of a foreign body..

 

Case report
 
A 72-year old mentally confused man was found in the street and was admitted to a peripheral hospital. He  presented with gross haematuria and an acute abdomen complicated by SIRS and severe hyperglycaemia. Initially, the patient was conscious and responsive. The complete blood cell count indicated a mild leucocytosis of 12.5/nl and a serum glucose level of 970 mg/dl. The patient underwent a CT scan for further evaluation. The scan showed a gas-forming infection in the lower abdomen with a necrotic bladder and partial necrosis of the prostate. A 6 cm long metal object appeared to be located within the urethra (Fig. 1 and 2).

 

Figure 1. CT-scan with the signs of an gas-forming infection.

 

 

Figure 2. CT-scan with a metal object in the urethra.

 

 

Approximately three hours after the primary admission he was transferred to our hospital, , in a deteriorating clinical condition.
Upon admission to our department, the patient was no longer responsive, and was hypotensive and tachycardic. The complete blood cell count now indicated a leucocytosis of 29.1/nl, his blood chemistry showed a creatinine of 4.7 mg/dl,  CRP of 305.3 mg/l and an persistent severe hyperglycaemia with a glucose level of 502 mg/dl.
In this terminal condition, the patient was no longer suitable for anaesthesia and we decided against an exploratory laparotomy. The patient died within hours.
Autopsy revealed extensive ulceration of the bladder mucosa with diffuse haemorrhagic changes in all layers and invasion into the prostate. In addition it showed the formation of air-filled cavities, within the bladder. Microbiology suggested an infection with a gram-positive organism (Enterococcus faecalis).
The cause of the necrotising cystitis was two safety pins, found in the bladder and urethra, which were most likely inserted for autoerotic purpose (Fig. 3 , 4 and 5).
 

Figure 3. Macroscopic image of  the urogenital system showing the kidneys and haemorrhagic bladder. The safety pin can be seen within the bladder.

 


 
Figure 4. Histological image of the emphysematous infection. Air-filled cavities can be seen.

 

 
 
Figure 5. Histological image of the bladder wall. Extensive ulceration of the bladder mucosa with diffuse haemorrhagic changes in all layers can be seen.

 


 
 
Discussion
 
Necrotising cystitis is a very rare entity in clinical practice. It is characterized as a emphysematous cystitis which can be complicated by complete necrosis of the bladder ( ).
In most cases, the course of disease is favourable. In the literature, it is stated that only about 10% of patients need medical and surgical treatment, with an overall mortality rate of 7% (2).
The pathogenesis of gas-forming infections of the urinary tract is not fully understood at present. It is thought to be due to a combination of the presence of gas-producing organisms, impaired tissue perfusion, and a high tissue glucose concentration; all are prerequisites that favour the development of emphysematous infections ( ). In most cases, the bacteria causing the infections are identified as E.coli, but Klebsiella pneumoniae, Clostridium species and Enterobacter species are also often found. In this case however, the basic cause of the infection was two foreign bodies which were inserted into urethra and bladder. This presumably happened for autoerotic purposes. However, since a detailed medical history could not be taken, , a traumatic cause can not be excluded.
The symptoms of emphysematous cystitis vary from a patient being almost asymptomatic, to presenting with an acute abdomen and severe sepsis. The most common clinical presentation is with lower abdominal pain, followed by dysuria, haematuria and sometimes pneumaturia (3). The infections occur more commonly in middle-aged woman, the main comorbidity is diabetes. Other risk factors include malnutrition or alcoholism.
Emphysematous or necrotising cystitis can be diagnosed radiologically by a simple plain film of the abdomen or on CT scanning, which can also differentiate vesicocolic fistula, intra-abdominal abscess, neoplastic diseases or the complication of an emphysematous pyelonephritis (2). In our case a foreign body could be seen in the CT scan.
Concerning the outcome, the most important issue is an early diagnostic and therapeutic approach. Depending on the course of the infection,  medical treatment with an intravenous antibiotic should be commenced. In some cases, additional treatment has to be considered. Severe infections might require partial cystectomy or even radical cystoprostatectomy.
In our case the patient deteriorated very rapidly. Since he presented in multi-organ failure caused by urosepsis and septic shock, no radical operation could have been considered life-prolonging. This underlines the need for  early diagnosis and treatment.
The authors declare that they have no competing interests.

 

References
 
1. Grayson DE, Abbott RM, Levy AD, Sherman PM. Emphysematous infections of the abdomen and pelvis: a pictorial review. Radiographics. 2002; 22:543-561.
2. Thomas AA, Lane BR, Thomas AZ, Remer EM, Campbell SC, Shoskes DA. Emphysematous cystitis: a review of 135 cases. BJU Int. 2007; 100:17-20.
3. Pérez Fentes D, Blanco Parra M, Lema Grille J, Toucedo Caamaño V, Novás Castro S, Lamas Cedrón P, Villar Núñez M. Emphysematous cystitis: case report. Arch Esp Urol. 2009; 62:392-395.
4. Perlemoine C, Neau D, Ragnaud JM, Gin H, Sahnoun A, Pariente JL, Rigalleau V. Emphysematous cystitis. Diabetes Metab. 2004;30:377-379.
5. Rindom AB, Gudnason HM, Thind PO. Emphysematous cystitis with total necrotization of the bladder. Ugeskr Laeger. 2008;170:3876.
6.  Huang JJ, Chen KW, Ruaan MK. Mixed acid fermentation of glucose as a mechanism of emphysematous urinary tract infection. J Urol. 1991;146:148-151.

 

Date added to bjui.org: 10/01/2012 


DOI: 10.1002/BJUIw-2011-114-web