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Abstract

We describe of a case, a 50 year old male who was operated for carcinoma of the descending colon and diverting loop ileostomy, developed a fungating lesion in mucocutaneous junction of ileostomy after one year which on histology revealed to be a Verrucous carcinoma. Resumo: Descrevemos um caso, homem, 50 anos, que foi operado para carcinoma de cólon descendente e ileostomia em alça para desvio. Transcorrido um ano, o paciente desenvolveu uma lesão vegetante na junção mucocutânea da ileostomia; a histologia revelou ser um carcinoma verrucoso. Keywords: Ileostomy, Verrucous carcinoma, Mucocutaneous junction, Palavras-chave: Ileostomia, Carcinoma verrucoso, Junção mucocutânea
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228
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Please
cite
this
article
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Dhakre
V,
Nagral
S.
Verrucous
carcinoma
at
ileostomy
site.
J
Coloproctol
(Rio
J).
2017.
http://dx.doi.org/10.1016/j.jcol.2017.05.002
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Journal
of
Coloproctology
Case
Report
Verrucous
carcinoma
at
ileostomy
site
Vijay
Dhakre,
Sanjay
Nagral
Q1
Jaslok
Hospital
and
Research
Centre,
Mumbai,
India
a
r
t
i
c
l
e
i
n
f
o
Article
history:
Received
12
January
2017
Accepted
1
May
2017
Available
online
xxx
Keywords:
Q3
Ileostomy
Verrucous
carcinoma
a
b
s
t
r
a
c
t
We
describe
of
a
case,
a
50
year
old
male
who
was
operated
for
carcinoma
of
the
descending
colon
and
diverting
loop
ileostomy,
developed
a
fungating
lesion
in
mucocutaneous
junction
of
ileostomy
after
one
year
which
on
histology
revealed
to
be
a
Verrucous
carcinoma
(VC).
Q2
©
2017
Sociedade
Brasileira
de
Coloproctologia.
Published
by
Elsevier
Editora
Ltda.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://creativecommons.org/
licenses/by-nc-nd/4.0/).
Carcinoma
verrucoso
em
local
de
ileostomia
Palavras-chave:
Ileostomia
Carcinoma
verrucoso
r
e
s
u
m
o
Descrevemos
um
caso,
homem,
50
anos,
que
foi
operado
para
carcinoma
de
cólon
descen-
dente
e
ileostomia
em
alc¸a
para
desvio.
Transcorrido
um
ano,
o
paciente
desenvolveu
uma
lesão
vegetante
na
junc¸ão
mucocutânea
da
ileostomia;
a
histologia
revelou
ser
um
carci-
noma
verrucoso
(CV).
©
2017
Sociedade
Brasileira
de
Coloproctologia.
Publicado
por
Elsevier
Editora
Ltda.
Este
´
e
um
artigo
Open
Access
sob
uma
licenc¸a
CC
BY-NC-ND
(http://creativecommons.org/
licenses/by-nc-nd/4.0/).
Introduction
Complications
of
stomal
site
are
often
encountered
by
clinicians1but
neoplasms
of
stomal
sites
are
very
rare.2
It
can
be
confused
or
get
obscured
by
granulation,
tis-
sue
excoriation
or
ignorance
by
health
care
provider.
We
share
our
experience
of
a
rare
case
of
verrucous
carcinoma.
Corresponding
author.
E-mail:
vddrvijayd@gmail.com
(V.
Dhakre).
Case
report
A
50
year
old
male
seropositive
for
human
immunodeficiency
virus
(HIV)
was
planned
for
a
loop
ileostomy
closure
after
one
year
of
anterior
resection
for
adenocarcinoma
of
descending
colon.
Patient
had
received
adjuvant
chemotherapy.
Patient
was
also
on
HAART
(highly
active
anti-retroviral
therapy)
for
HIV
infection.
http://dx.doi.org/10.1016/j.jcol.2017.05.002
2237-9363/©
2017
Sociedade
Brasileira
de
Coloproctologia.
Published
by
Elsevier
Editora
Ltda.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
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JCOL
228
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Please
cite
this
article
in
press
as:
Dhakre
V,
Nagral
S.
Verrucous
carcinoma
at
ileostomy
site.
J
Coloproctol
(Rio
J).
2017.
http://dx.doi.org/10.1016/j.jcol.2017.05.002
ARTICLE IN PRESS
JCOL
228
1–3
2
j
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(rio
j).
2
0
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7;x
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x(x
x):xxx–xxx
Fig.
1
Intraoperative
ileostomy
site
with
the
lesion
(black
arrow)
in
the
superomedial
mucocutaneous
junction
which
is
dissected
and
mobilized.
Fig.
2
Marked
acanthosis
with
broad
bulbous
process
(black
arrow
head).
The
bulbous
rete
processes
“push”
deep
into
the
lamina
propria
so
that
the
base
of
the
lesion
is
below
the
adjacent
basement
membrane.
Before
stoma
closure
CT
scan
had
shown
normal
distal
bowel
and
no
evidence
of
recurrence.
Three
months
prior
to
the
closure;
patient
had
reported
oozing
of
blood
from
the
stomal
site.
A
local
examination
revealed
an
area
of
hyper-
granulation
which
was
attributed
to
chronic
irritation
and
was
left
alone.
During
the
surgery
we
noticed
a
small
fungating
mass
on
the
right
lateral
edge
of
the
mucocutaneous
site
(Fig.
1).
In
view
of
suspicion
of
malignancy
a
wide
local
resection
of
the
loop
ileostomy
was
performed,
keeping
a
margin
of
2
cm
over
the
skin
and
abdominal
wall
layers
(Fig.
2).
Patient
is
well
after
six
months
after
surgery
without
evi-
dence
of
recurrence.
Discussion
Complications
of
stoma
are
well
described
and
include
stomal
herniation,
prolapse
of
stoma,
retraction,
skin
excoriation,
intestinal
obstruction,
stenosis,
abscess,
fistula,
diarrhoea,
urinary
calculus,
ileitis,
and
inflammatory
polyps.1Develop-
ment
of
malignancy
at
ileostomy
in
is
rare2Suarez
et
al.
estimated
the
incidence
of
ileostomy
carcinomas
in
patients
with
ileostomy
for
various
indications
in
the
UK
to
be
2
to
4
per
1000
ileostomies.
Squamous
cell
carcinoma
(SSC)
have
been
described
commonly
in
HIV
positive
patients
at
muco-
cutaneous
junctions
specially
anorectal
junction.3However
to
our
knowledge
VC
has
not
being
described
at
the
ileostomy
site,
although
there
are
reports
of
SCC.
Till
date
only
a
few
cases
of
SCC
at
ileostomy
have
been
described.4Ulcerative
colitis
was
the
most
common
under-
lying
condition;
Farshid
in
their
review
described
timing
of
lesion
from
ileostomy
fashioning
was
in
the
range
of
26
years
to
54
years.
In
our
case
this
time
was
very
short
(12
months).
Maw
et
al.5had
mentioned
in
their
case
series
of
44
stomal
neoplasms;
described
40
adenocarcinomas
and
four
squa-
mous
cell
carcinoma,
but
no
VC
variant
was
described.
They
described
of
ileostomy
neoplasm
associated
with
Crohn’s
dis-
ease,
familial
adenomatous
polyposis
and
ulcerative
colitis.
They
postulated
that
chronic
irritation
predisposed
the
stoma
to
malignant
changes.
They
discussed
a
strong
association
in
patient
with
chronic
diseases
like
ulcerative
colitis
or
primary
sclerosing
cholangitis
making
it
a
high
risk
group
for
ileostomy
neoplasms.
The
chronic
irritation
theory
is
supported
by
the
fact
that
majority
of
stomal
carcinoma
have
been
described
only
in
long
standing
permanent
stomas.6In
the
setting
of
HIV,
human
papilloma
virus
(HPV)
infection
is
well
known
to
predispose
to
carcinomas.
Viral
interactions
in
HPV
can
pre-
dispose
to
VC.6In
our
case
though
the
immunohitochemical
analysis
for
HPV
was
negative.
The
E6/E7
viral
oncoproteins
of
HPV
are
proved
to
inactivate
the
tumour
suppressor
gene
like
p53
and
pRb
which
in
turn
lead
to
the
cell
proliferation
and
eventually
turning
them
in
to
malignant
cells.
Highly
active
anti-retroviral
therapy
(HAART)
modulating
the
immune
is
also
taken
into
account
while
considering
interactions
of
these
viruses.7
Overall
risk
of
carcinomas
in
HIV
patients
is
more
than
in
general
population.7Moreover
associations
of
HIV
non-
Hodgkin’s
lymphoma
(NHL)
at
ileostomy
site
specifically
because
of
microtraumatisms
and
locally
present
antigen
stimulation
and
activation
has
been
discussed
in
their
work
by
Levecq
et
al.3
Also
it
is
pertinent
to
mention
activation
of
signalling
path-
ways
like
Akt/mTOR
which
can
get
activated
and
is
also
known
to
cause
VC
in
oral
malignancy.8
Conclusion
Peristomal
carcinoma
is
a
rare
entity.
VC
(a
rare
form
of
SCC)
may
arise
as
a
lesion
from
an
ileostomy.
A
clinician
must
sus-
pect
malignancy
when
there
is
a
hypergranulation
or
mass
like
lesion
at
ileostomy
site.
Immunodeficiency
conditions
like
HIV
31
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94
JCOL
228
1–3
Please
cite
this
article
in
press
as:
Dhakre
V,
Nagral
S.
Verrucous
carcinoma
at
ileostomy
site.
J
Coloproctol
(Rio
J).
2017.
http://dx.doi.org/10.1016/j.jcol.2017.05.002
ARTICLE IN PRESS
JCOL
228
1–3
j
coloproctol
(rio
j).
2
0
1
7;x
x
x(x
x):xxx–xxx
3
should
be
considered
a
high
risk
group
for
such
rare
forms
of
malignancy.
Conflicts
of
interest
The
authors
declare
no
conflicts
of
interest.
Acknowledgements
Dr
Vijay
Dhakre
designed
and
wrote
up
the
article
with
review
of
literature
and
procuring
the
data,
Dr
Sanjay
Nagral
finalized
the
article
and
literature
review.
r
e
f
e
r
e
n
c
e
s
1.
Attanoos
R,
Billings
P,
Hughes
L,
Williams
G.
Ileostomy
polyps,
adenomas,
and
adenocarcinomas.
Gut.
1995;37:840–4.
2.
Suarez
V,
Alexander-Williams
J,
O’Connor
J,
Campos
A,
Fuggle
J,
Thompson
H,
et
al.
Carcinoma
developing
in
ileostomies
after
25
or
more
years.
Gastroenterology.
1988;95:205–8.
3.
Levecq
H,
Hautefeuille
M,
Hoang
C,
Galian
A,
Hautefeuille
P,
Rambaud
JC.
Primary
stomal
lymphoma.
An
unusual
complication
of
ileostomy
in
a
patient
with
transfusion-related
acquired
immune
deficiency
syndrome.
Cancer.
2006;65:1028–32.
4.
Farshid
E,
Metin
N,
Rangasamy
S.
Squamous
cell
carcinoma
at
an
ileostomy
site
Fiftyfour
years
following
colectomy
for
ulcerative
colitis:
a
case
report
and
literature
review.
Int
J
Surg
Case
Rep.
2013;4:678–80.
5.
Quah
HM,
Samad
A,
Maw
A.
Ileostomy
carcinomas
a
review:
the
latent
risk
after
colectomy
for
ulcerative
colitis
and
familial
adenomatous
polyposis.
Colorectal
Dis.
2005;7:
538–44.
6.
Tonna
J,
Palefsky
JM,
Rabban
J,
Campos
GM,
Theodore
P,
Ladabaum
U.
Esophageal
verrucous
carcinoma
arising
from
hyperkeratotic
plaques
associated
with
human
papilloma
virus
type
51.
Dis
Esophagus.
2010;23:E17–20.
7.
Righetti
E,
Ballon
G,
Ometto
L,
Cattelan
AM,
Menin
C,
Zanchetta
M,
et
al.
Dynamics
of
Epstein–Barr
virus
in
HIV-1-infected
subjects
on
highly
active
antiretroviral
therapy.
AIDS.
2002;16:63–73.
8.
Chaisuparat
R,
Limpiwatana
S,
Kongpanitkul
S,
Yodsanga
S,
Jham
BC.
The
Akt/mTOR
pathway
is
activated
in
verrucous
carcinoma
of
the
oral
cavity.
J
Oral
Pathol
Med.
2016;45:581–5.
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Article
Full-text available
Carcinoma arising at an ileostomy site is an extremely rare occurrence. The rate of malignancy arising at an ileostomy site is reported as being 2-4 of every 1000 cases. The development of squamous cell carcinoma at the mucocutaneous junction of an ileostomy is extremely rare. We present a case of a 76-year-old male who developed squamous cell carcinoma at an ileostomy site fifty-four years after total colectomy as management for ulcerative colitis. Our literature review has identified only four similar cases previously published in English literature. All cases of squamous cell carcinoma developing in ileostomy have occurred after a minimum of twenty-six years following ileostomy. This suggests that the etiology may be due to chronic factors. Patients with chronic stomal inflammation, bleeding or persistent induration and/or mass formation should be followed up closely and investigated for recurrence or development of a new malignancy. There should be a low threshold to obtain an early definitive tissue diagnosis by taking biopsies to prevent local or systemic invasion.
Article
Full-text available
Ileostomy polyps are uncommon and poorly described. The aim of this study was to undertake a retrospective clinicopathological review of ileostomy polyps. Seven patients with 60 polyps arising on ileostomies performed for ulcerative colitis were studied. The histopathological evaluation of archival ileostomy biopsy specimens, polypectomy or excision specimens, and clinical review of patient records was undertaken. Fifty of 60 polyps were inflammatory cap polyps and six further polyps were composed of granulation tissue only. They occurred anywhere on the stoma at any time after ileostomy construction and were strongly associated with overt stomal prolapse. Four neoplastic polyps were identified in two patients 27-36 years after ileostomy construction; all occurred at the mucocutaneous junction. One patient presented with a 2 cm polypoid invasive adenocarcinoma while in the second a 1.7 cm polypoid mucinous adenocarcinoma and a 0.7 cm ileal tubular adenoma with high grade dysplasia occurred at the site of excision of a cap polyp showing focal low grade adenomatous dysplasia six years previously. Neoplastic and non-neoplastic polyps could not be differentiated clinically. It was found that most ileostomy polyps are inflammatory cap polyps associated with stomal prolapse. Less common are polypoid adenomas or adenocarcinomas arising at the mucocutaneous anastomosis > 20 years after ileostomy construction. To prevent ileostomy carcinoma it is recommended that a biopsy of all polyps at the mucocutaneous anastomosis and of any non-prolapse associated polyps elsewhere on the stoma occurring > 15 years after ileostomy construction is done.
Article
Background: The Akt/mTOR pathway is activated in many malignancies, including oral squamous cell carcinoma (OSCC). However, the role of the Akt/mTOR pathway in oral verrucous carcinoma (OVC), a low-grade variant of OSCC, remains unknown. Thus, the objective of this study was to investigate the activation level of important markers of the Akt/mTOR pathway in OVC and to compare the results with OSCC samples. Methods: The expression of p-Akt (Thr308), p-Akt (Ser473), and p-RPS6 was evaluated by immunohistochemistry in 30 OSCC cases, 18 OVC cases, and 30 control cases (normal epithelium overlying fibromas). Statistical analysis was performed to determine the differences in protein expression between samples. Results: All OVC cases were positive for p-Akt (Thr308), p-Akt (Ser473), and p-RPS6. There were significant differences in expression level of all studied proteins between OVC and control, as well as between OVC and OSCC. However, OVC showed significant lower staining scores than OSCC. Conclusions: Our findings demonstrate that the Akt/mTOR pathway is upregulated in OVC, indicating a role for this pathway in the development and progression of this malignancy.
Article
Esophageal verrucous carcinoma is a rare variant of esophageal squamous cell carcinoma. We report a case of esophageal verrucous carcinoma associated with human papilloma virus (HPV) type 51. The patient had long-standing dysphagia and odynophagia, and white esophageal plaques showing hyperkeratosis on biopsy. At repeat endoscopy, the esophagus was covered with verrucous white plaques and areas of nodular mucosa with white fronds, with a distal 10-cm smooth mass protruding into the lumen. Biopsies demonstrated an atypical squamoproliferative lesion but no frank malignancy. HPV type 51 DNA was detected in endoscopic biopsy specimens by polymerase chain reaction. Because the size of the lesion favored an underlying verrucous carcinoma, our patient underwent minimally invasive esophagectomy with gastric pull-up and cervical anastomosis. The pathologic diagnosis was a well-differentiated esophageal verrucous carcinoma. One year after esophagectomy, the patient feels well and is free of disease. Although HPV DNA was not detected in the cancer tissue obtained at surgery, our case suggests an association between HPV type 51 and esophageal verrucous carcinoma. The clinical evolution in this case highlights the importance of endoscopic surveillance in patients with exuberant esophageal hyperkeratosis, and of definitive surgical resection when malignancy is suspected even if frank malignancy is not demonstrated on superficial biopsies.
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A 73-year-old heterosexual man developed a high-grade non-Hodgkin's lymphoma at the site of an ileostomy only 2 years after proctectomy for undetermined colitis not cured by previous colectomy. In fact, the early occurrence of this usually very late and rare complication of ileostomy was probably favored by the simultaneous presence of acquired immune deficiency syndrome (AIDS) due to repeated blood transfusions for refractory anemia with excess blasts. The intestinal location of the tumor, its high-grade malignancy and B-cell origin are all features of AIDS-related non-Hodgkin's lymphoma. This case report seems to be one of the rarely identified examples of the cooperation between general predisposing factors and local irritating agents at the origin of a malignant tumor.
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Primary carcinoma of the ileal mucosa in patients who have had an ileostomy has been documented rarely. This report describes 4 patients seen within the last 4 yr with ileal carcinoma adjacent to the mucocutaneous junction of an ileostomy 25, 29, 30, and 34 yr after the stoma was formed. The incidence of this serious complication may be increasing.
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Patients infected with HIV-1 are at high risk of developing Epstein-Barr virus (EBV)-associated lymphoproliferative disorders. This study evaluated the impact of highly active antiretroviral therapy (HAART) on EBV infection. To measure EBV content in peripheral blood lymphocytes (PBL) and in plasma, we set up a quantitative analysis using the real-time PCR. EBV latent membrane protein 1 (LMP1) expression was determined by reverse transcriptase-PCR. EBV levels were determined in 33 HIV-1- and EBV-coinfected patients at the start of HAART, and during therapy. At baseline, EBV content in PBL samples ranged from 8 to 14 532 copies/microg DNA. EBV levels transiently increased in nine out of 17 patients in whom HIV-1 plasmaviraemia declined to undetectable levels (virological response) and CD4 cell counts increased (immunological response), while they remained fairly stable or decreased in the other eight virological and immunological responders, and in seven patients who showed a virological response only. Of interest, a significant increase in EBV load was observed in five out of nine patients who showed an increase in CD4 cell counts but lack of HIV-1 suppression during HAART. This EBV increase was accompanied by the detection of both LMP1 transcripts in PBL and EBV DNA in plasma, and was paralleled by an increase in immunoglobulin levels, a marker of B-cell stimulation. These findings suggest that peripheral immune reconstitution during HAART without a reduction in HIV-1 replication may increase B-cell stimulation and the number of EBV-infected B cells.
Article
Ileostomy carcinoma after colectomy for ulcerative colitis and familial adenomatous polyposis is rare. Forty-three case reports from the literature and a case of ours are reviewed. The risk of malignancy following ileostomy formation appears to be increased compared to the very low incidence of primary small bowel carcinoma. Chronic physical or chemical irritation of the stoma may predispose the ileal mucosa to colonic metaplasia with subsequent adenoma formation, dysplasia and invasive malignant change. This is particularly so where ileostomies are fashioned for familial adenomatous polyposis and ulcerative colitis. Routine surveillance of patients who have had an ileostomy for 15 years or longer may lead to earlier detection of this complication.