Jun 10, 2005 - adult woman requiring radical surgery for progressive relentless disease. Estrogen deficiency associated with a possible allergic etiology could ...
Blackwell Publishing AsiaMelbourne, AustraliaIJUInternational Journal of Urology0919-81722006 Blackwell Publishing Asia Pty Ltd???200613811321133Eosinophilic cystitis and anorexia nervosa J Philip et al.
International Journal of Urology (2006) 13, 1132–1133
Case Report
Severe eosinophilic cystitis in a woman with anorexia nervosa JOE PHILIP,1 FAISAL SM ALI,2 HANI D ZAKHOUR2 AND NIGEL J PARR1 Departments of 1Urology and 2Histopathology, Wirral Hospitals NHS Trust, Wirral, UK Abstract Eosinophilic cystitis is a rare inflammatory disorder. It is considered to be self limiting necessitating only supportive therapy. Surgical intervention is unusual. We report here an association between eosinophilic cystitis and anorexia nervosa in an adult woman requiring radical surgery for progressive relentless disease. Estrogen deficiency associated with a possible allergic etiology could explain this association. Key words anorexia nervosa, cystectomy, eosinophilic cystitis.
Introduction Eosinophilic cystitis (EC) is a rare inflammatory disorder, described in elderly men with recent instrumentation and also in young adults. Symptoms include suprapubic pain, frequency, dysuria and hematuria. Radiological findings include bladder mass lesions and upper tract dilatation.1 Symptomatic control is advocated, as EC is considered self-limiting.2 Surgical intervention is unusual. EC has been reported in association with transitional cell bladder carcinoma, respiratory diseases, autoimmune disorders, parasitic infestations and eosinophilic enteritis.3 Case report A 22-year-old woman, with a body mass index (BMI) of 23 and suffering from anorexia nervosa, presented with right loin pain, irritative bladder symptoms and a history of recurrent urinary tract infection. Her daytime frequency ranged between 10 and 30 times with nocturia of up to 5 times. An i.v. urogram showed a dilated right collecting system and bilateral vesico-ureteric reflux on micturating cysto-urethrogram. Cystoureteroscopy was normal. She underwent a right subureteric injection of macroplastique. However, she then attended elsewhere with ongoing right loin pain and open right renal denervation was performed, which relieved symptoms for 2 months. She returned to our hospital with recurrent pain and hematuria. Repeat cystoureteroscopy revealed an inflamed bladder with patchy involvement of the right ureter and renal pelvis. Biopsy showed edematous papillary urothelium with marked eosinophil infiltrate. Investigations excluded peripheral eosinophilia, eosinophilic gastroenteritis, chlamydia and toxocara. Intravesical sodium pentosan polysulfate, predCorrespondence: J. Philip MRCSI, 1 Catkin Road, Halewood village, Liverpool L26 7XJ, UK. Email: indianajoe@gmail. com Received 10 June 2005; accepted 14 December 2005.
nisolone (given p.o) and steroid instillations had only transient benefit. After each modality of treatment, her symptoms showed temporary marginal resolution for periods ranging from 2 weeks to 3 months. Her BMI had fallen to 15 over a 3-year period. Four years later, persistent suprapubic pain requiring an analgesic infusion pump, high dose opiates and intense nocturia necessitated urinary diversion. A year afterwards, progression of symptoms required cystourethrectomy. Histology showed mucosal ulceration with predominantly eosinophil permeation of the underlying stroma and smooth muscle (Fig. 1a). Exogenous material surrounded by numerous foreign-body type giant cells was noted near the right ureteric orifice (Fig. 1b). She had gradual relief of symptoms and improvement in appetite. BMI had increased to 22. She does not have an infusion pump and is being weaned off opiates. Discussion Eosinophilic cystitis is rare, being described in elderly men with recent instrumentation and young adults. Symptoms include suprapubic pain, frequency, dysuria and hematuria.1 Radiological findings include bladder mass lesions and upper tract dilatation.2 Symptomatic control is advocated, as EC is considered self-limiting.3 Surgical intervention is unusual.4 Cystourethrectomy for debilitating EC has been reported in four cases previously, none associated with anorexia nervosa or with macroplastique injection. We report here an association between EC and anorexia nervosa in an adult woman requiring radical surgery for progressive relentless disease. Women with anorexia nervosa have been reported to have a higher incidence of irritative urinary symptoms. Anorexic women tend to be estrogen deficient which results in increased loss of intermediate and superficial cells in the urinary bladder and urethra.4 Estrogen deficiency associated with a possible allergic etiology could explain this association.
Eosinophilic cystitis and anorexia nervosa a)
References 1 2 3 4
b)
Fig. 1 (a) The inflammatory cell infiltrate comprises large numbers of eosinophils diffusely infiltrating within the stroma. These have multilobated nuclei and bright orange/ pink cytoplasm. Several small blood vessels are also present within this field. (hematoxylin and eosin (H&E), ×20 magnification). (b) Numerous foreign-body type multinucleated giant cells are present (arrows), which are intimately associated with slightly polarisable oleus material (arrow heads). (H&E ×10 magnification).
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