1 3
Int Urol Nephrol
DOI 10.1007/s11255-015-0966-4
UROLOGY – LETTER TO THE EDITOR
Keratinising cystitis with intestinal metaplasia following a
Crohn’s vesico-intestinal fistula
Deepak Batura
1
· Grigorios Mitsopoulos
2
Received: 25 March 2015 / Accepted: 27 March 2015
© Springer Science+Business Media Dordrecht 2015
melanocytic population. There was no evidence of dyspla-
sia or malignancy. She is currently being followed-up by
six-monthly cystoscopic biopsies.
Keratinising cystitis is a sub-type of squamous meta-
plasia. It is more common in males and is usually asso-
ciated with chronic irritation. It usually presents with
haematuria, dysuria and urgency. Most keratinised areas
are whitish deposits, though pigmented areas have been
described [1]. This patient presented with black mem-
brane-like deposits. Endoscopy is not diagnostic, and
microscope examination is needed to exclude non-kerati-
nizing squamous metaplasia, fungal cystitis, malakoplakia
or amyloidosis [2]. Unlike the non-keratinising variety,
it is a premalignant condition associated with squamous
cell cancer [3]. Extensive keratinization may also result in
bladder contracture [4]. Chronic irritation due to UTI and
entero-vesical fistula, such as our patient had, predisposes
to its development [4, 5].
There is no effective treatment for this enigmatic and
challenging condition that can cause significant morbid-
ity. Strategies such as antibiotics and vitamin A have not
been uniformly helpful. Nor have intravesical instillations
of silver nitrate or acetic acid [4]. Transurethral resection of
affected areas has been described [2]. This patient did not
respond to antibiotics. The keratinising membrane was too
extensive and thin to be resected, and therefore, endoscopic
surveillance was decided upon.
This case highlights the importance of repeat cystos-
copy in recurrent UTI, lest evolving underlying conditions
be missed. Surveillance assumes greater importance in
this patient because of the coexistence of intestinal meta-
plasia, as both these types of metaplasia are premalignant,
in a rare combination of pathologies. Surveillance is also
required for early detection of complications like bladder
contracture.
Editor,
A 45-year-old woman developed an entero-vesical fistula
secondary to Crohn’s disease in 2006. She was treated
with a right hemicolectomy, anterior resection and bladder
repair with omental interposition. An anastomotic leak led
to pelvic and presacral sepsis, which was treated by per-
cutaneous drainage without defunctioning; however, pre-
sacral sepsis persists till date. She is on systemic azathio-
prine. She continued to have recurrent UTI after surgery.
A cystoscopy in 2009 was normal. UTI recurred despite
several courses of appropriate antimicrobials leading to
another cystoscopy in 2013 which showed a patchy black
membrane over the entire bladder (Fig. 1a, b). The mem-
brane peeled away readily with biopsy forceps with under-
lying raw areas. Histology of the membrane and mucosa
showed keratinising squamous epithelium with prominent
keratohyaline granules and foci of glandular epithelium
with intestinal metaplasia (Fig. 1c, d). Detached fragments
of keratinous debris were seen along with bacteria. Tissue
culture of the membrane isolated Enterococcus faecalis and
Morganella morganii. PAS and Grocott’s stains for fungi
were negative. Because of the colour of the membrane,
melanin deposits were sought. Masson-Fontana stain did
not detect melanin deposits within epithelial cells. S100,
HMB 45 and Melan A immunostains failed to identify any
* Deepak Batura
deepakbatura@gmail.com
1
Department of Urology, London North West Healthcare
NHS Trust, Ealing Hospital, Uxbridge Road, Southall,
London UB1 3HW, UK
2
Department of Pathology, London North West Healthcare
NHS Trust, Ealing Hospital, Uxbridge Road, Southall,
London UB1 3HW, UK