S-123
1
Department of Otolaryngology,
Head & Neck Surgery, University Hospital
Limerick, Dooradoyle, Limerick, Ireland;
2
Department of Rheumatology, University
Hospital Limerick, Dooradoyle, Limerick,
Ireland;
3
Department of Otolaryngology,
Head & Neck Surgery, Royal Victoria
Eye and Ear Hospital, Dublin, Ireland;
4
University of Limerick, Graduate Entry
Medical School, Limerick, Ireland.
Conall W.R. Fitzgerald, MD, FRCSI
Fahd Adeeb, MRCPI
Conrad V. Timon, MD, FRCSI
Neville P. Shine, FRCSI
Alexander D. Fraser, MD, FRCSI
Joseph P. Hughes, FRCSI
Please address correspondence to:
Alexander Duncan Fraser,
Consultant Rheumatology,
University Hospital Limerick,
Dooradoyle,
Limerick, Ireland.
E-mail: alexander.fraser@hse.ie
Received on June 26, 2015; accepted in
revised form on August 31, 2015.
Clin Exp Rheumatol 2015; 33 (Suppl. 94):
S123-S128.
© Copyright CliniCal and
ExpErimEntal rhEumatology 2015.
Key words: Behçet’s disease,
otolaryngology, laryngeal changes,
ear, nose, throat
Competing interests: none declared.
ABSTRACT
Objective. Behçet’s disease (BD) is
a multisystem autoimmune disease of
unknown origin typically affecting the
triad of oral and genital mucosa and the
eye. Limited data are available in the
literature regarding the otolaryngology-
related manifestations of BD, particu-
larly in northern Europeans. This is a
novel study detailing surprising and sig-
nifcant laryngeal structural changes in
a northern European cohort of BD.
Methods. Patients meeting the Interna-
tional Study Group for Behçet’s Disease
(ISGBD) and the International Criteria
for Behçet’s Disease (ICBD) criteria
for diagnosis were identifed from an
institutional database. Patients under-
went examination with an otolaryngol-
ogist, including fexible laryngoscopy.
Intra-oral, pharyngeal and laryngeal
manifestations of BD were documented
and characterised. Patients underwent
hearing assessment with pure-tone au-
diometry.
Results. Fifteen patients with BD were
identifed (4 male, 11 female; median
age 36 years). 60% (n=9) showed
evidence of disease on examination
and fexible laryngoscopy. 33% (n=5)
showed laryngeal changes related to
BD. 13% (n=2) demonstrated bilateral
sensorineural hearing loss. The 5 cases
demonstrating laryngeal manifesta-
tions of disease are described in detail
with photographic records.
Conclusion. Limited data has been pub-
lished regarding the laryngeal manifes-
tations of BD, particularly in a north-
ern European population. Our cohort
of BD patients demonstrate signifcant
laryngeal structural changes. It would
appear that these clinically relevant
changes may be more common than
was previously thought. Raised aware-
ness of the risk of laryngeal pathology
in BD patients, often in the absence of
overt clinical symptomatology, may re-
sult in earlier diagnosis and treatment.
Rheumatologists and otolaryngologists
should consider closer multi-discipli-
nary co-operation in the management
and follow up of patients with BD.
Introduction
Behçet’s disease (BD) is a complex,
infammatory, systemic disease of un-
known aetiology. Although Hippocrates
frst described a patient with a triad of
mouth ulcers and symptoms affect-
ing the genitals and eyes 2,500 years
ago, Professor Hulusi Behçet, a Turk-
ish dermatologist, in 1937 provided the
frst characterisation of the trisymptom
‘occulo-buccal-genital’ complex (1, 2).
The clinical manifestations of BD are
believed to be attributable to vasculi-
tis, which could involve blood vessels
of any size from arterial or venous cir-
culation. The prevalence of BD shows
signifcant geographical variation with
a reported prevalence ranging from
80–400/100,000 in Turkey and Middle-
Eastern countries, to less than 1/100,000
in western Europe (3, 4). Pathogenesis
of the disease is not well understood,
however, environmental and genetic
predisposition may play a role in the de-
velopment of the disease (3, 4).
No pathognomonic laboratory test ex-
ists to diagnosis BD. As such, the di-
agnosis of BD is guided by validated
criteria, most commonly used are the
International Study Group for Behçet’s
Disease (ISGBD) or the International
Criteria for Behçet’s Disease (ICBD).
In the ISGBD criteria, which were pub-
lished in 1990, recurrent oral aphthosis
three or more times in a year is man-
datory, with the presence of any two of
the following: genital ulceration, ocu-
lar or cutaneous manifestation, or skin
pathergy.
While having excellent specifcity, the
ISGBD criteria lacked sensitivity, and
for this reason the ICBD criteria were
developed and frst presented in 2006
(5, 6). For this classifcation, which is
Signifcant laryngeal destruction in a northern European cohort
of Behçet’s disease patients
C.W.R. Fitzgerald
1
, F. Adeeb
2,4
, C.V. Timon
3
, N.P. Shine
1
, A.D. Fraser
2,4
, J.P. Hughes
1