S-129
1
Division of Gastroenterology, and
2
Division of Rheumatology, Department
of Internal Medicine, Cerrahpasa Medical
School, Istanbul University, Turkey;
3
Division of Rheumatology, Department
of Internal Medicine, Trakya University
Medical School, Edirne, Turkey.
Ibrahim Hatemi, MD , PhD
Gulen Hatemi, MD, PhD
Omer Nuri Pamuk, MD, PhD
Yusuf Erzin, MD, PhD
Aykut Ferhat Celik, MD, PhD
Please address correspondence to:
Ibrahim Hatemi,
Sakayik sokak, Zeynep Cemal ap. 11/19,
Tesvikiye, 34365 Istanbul,
Turkey.
E-mail: ihatemi@yahoo.com
Received on May 1, 2015; accepted in
revised form on August 31, 2015.
Clin Exp Rheumatol 2015; 33 (Suppl. 94):
S129-S137.
© Copyright CliniCal and
ExpErimEntal rhEumatology 2015.
Key words: Behçet’s disease,
Behçet’s syndrome, management,
intestinal involvement, infammatory
bowel disease, infiximab, etanercept,
adalimumab, thalidomide
Competing interests: none declared.
ABSTRACT
Objective. Gastrointestinal involvement
of Behçet’s syndrome is usually treated
with glucocorticoids, 5-aminosalicyl-
ic acid compounds and azathioprine.
However, some patients are refractory
to these conventional therapy modali-
ties. In this paper we report our expe-
rience on 13 patients with gastrointes-
tinal involvement of Behçet’s syndrome
who were refractory to the conventional
therapy and who were treated with TNF-
alpha antagonists and/or thalidomide.
Methods. We reviewed the charts of
our Behçet’s syndrome patients with
gastrointestinal involvement and iden-
tifed those who were treated with TNF-
alpha antagonists and/or thalidomide.
Demographic features, previous and
concomitant drugs, previous surgery,
time to remission and duration of remis-
sion were tabulated. We also performed
a systematic review of publications on
gastrointestinal involvement of Behçet’s
syndrome patients treated with TNF-
alpha antagonists and/or thalidomide.
Results. Among our 64 patients with
gastrointestinal involvement of Behçet’s
syndrome, we identifed 13 (20%) (7
women, 6 men, mean age 27.4 ± 9.4)
who had been treated with TNF-alpha
antagonists and/or thalidomide. Their
previous medications were glucocor-
ticoids (13/13), azathioprine (13/13),
5-aminosalicylic acid derivatives
(3/13) and budesonide (1/13). Clinical
and endoscopic remission was obtained
in 10 patients. One patient died with
sepsis. The systematic literature search
revealed 91 cases who had used TNF-
alpha antagonists and 15 who had used
thalidomide. Among the patients who
had received TNF-alpha antagonists,
clinical remission was obtained in
47/91 patients (51%), while endoscopic
remission was observed in 21/46 (45%)
who had a control colonoscopy.
Conclusion. One ffth of our Behçet’s
syndrome patients with gastrointestinal
involvement were refractory to conven-
tional treatment modalities. Remission
was obtained with TNF-alpha antago-
nists and/or thalidomide in about 75 %
of the cases.
Introduction
Gastrointestinal involvement of Be-
hçet’s syndrome (GIBS) is character-
ised by gastrointestinal ulcers that are
located mainly in the ileocecal region.
These ulcers can also be located in the
other colonic segments, small intes-
tine, oesophagus or duodenum. GIBS
may cause abdominal pain, diarrhoea,
fever or weight loss. The main compli-
cations are perforation, bleeding and
fstula formation (1). Surgical interven-
tions can be necessary in some patients
due to acute complications such as
acute abdomen or bleeding and in oth-
ers due to persistent symptoms despite
medical therapy (2). The prevalence of
GIBS shows differences depending on
the geographic distribution. It is more
frequent in the Far East including Ja-
pan and Korea and the United States,
compared to the Middle East and Eu-
rope (3). This difference may be related
to several factors such as genetic and
environmental factors, as well as the
diagnostic methods and criteria used
(1). The management of GIBS is simi-
lar to infammatory bowel diseases in
terms of the medications used. The
conventional treatment modalities are
glucocorticoids, azathioprine, salazo-
pyrine and other 5-aminosalicylic acid
(5-ASA) derivatives (4). Some patients
with GIBS can be refractory to these
agents, necessitating other medical op-
tions. TNF-alpha antagonists and tha-
lidomide are the main options in such
cases. TNF-alpha antagonists have
been used with increasing frequency
TNF-alpha antagonists and thalidomide for the
management of gastrointestinal Behçet’s syndrome
refractory to the conventional treatment modalities:
a case series and review of the literature
I. Hatemi
1
, G. Hatemi
2
, O.N. Pamuk
3
, Y. Erzin
1
, A.F. Celik
1