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Korean J Intern Med 2013;28:614-618
http://dx.doi.org/10.3904/kjim.2013.28.5.614
Copyright © 2013 The Korean Association of Internal Medicine
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CASE REPORT
A rare cause of dysphagia: compression of the
esophagus by an anterior cervical osteophyte due
to ankylosing spondylitis
İ
lknur Albayrak
1
, Sinan Ba
ğ
cacı
2
, Ali Sallı
2
, Sami Kucuksen
2
, and Hatice U
ğ
urlu
2
1
Department of Physical Therapy
and Rehabilitation, Beysehir Public
Hospital, Konya;
2
Department of
Physical Therapy and Rehabili-
tation, Selcuk University Meram
Faculty of Medicine, Konya, Turkey
Received: February 7, 2012
Revised : April 23, 2012
Accepted: September 17, 2012
Correspondence to
İ
lknur Albayrak, M.D.
Department of Physical
Therapy and Rehabilitation,
Beysehir Public Hospital, Konya
42460, Turkey
Tel: +90-50-5689-9750
Fax: +90-33-2512-6271
E-mail: ilknurftr@gmail.com
Ankylosing spondylitis (AS) is a chronic inflammatory rheumatological disease
affecting the axial skeleton with various extra-articular complications. Dysphagia
due to a giant anterior osteophyte of the cervical spine in AS is extremely rare. We
present a 48-year-old male with AS suffering from progressive dysphagia to soft
foods and liquids. Esophagography showed an anterior osteophyte at C5-C6 re-
sulting in esophageal compression. The patient refused surgical resection of the
osteophyte and received conservative therapy. However, after 6 months there was
no improvement in dysphagia. This case illustrates that a large cervical osteo-
phyte may be the cause of dysphagia in patients with AS and should be included
in the diagnostic workup in early stages of the disease.
Keywords:
Spondylitis, ankylosing; Deglutition disorders; Osteophyte; Cervical
spine; Esophageal compression
INTRODUCTION
Ankylosing spondylitis (AS) is a chronic systemic, in-
flammatory, rheumatic disease affecting primarily the
sacroiliac (SI) joints and the skeleton. Other clinical
manifestations include peripheral arthritis, enthesitis,
and extra-articular organ involvement such as uveitis,
aortitis, pericarditis, pulmonary fibrosis, and amyloi-
dosis [1].
Dysphagia related to the musculoskeletal system is
encountered frequently in cases such as diffuse idio-
pathic skeletal hyperostosis (DISH), acromegaly, hypo-
parathyroidism, fluorosis, ochronosis, and trauma [2].
However, dysphagia is very rare in AS and can develop
due to syndesmophytes. Dysphagia in AS needs to be
defined, as it may lead to complications like weight loss
and nutritional disorders. Here, we describe a case of
dysphagia in AS to raise awareness of this condition.
CASE REPORT
A 48-year-old male, who was being followed up for AS,
visited our hospital complaining of progressive dys-
phagia. Difficulty swallowing solid foods started ~4
years prior, and for the last year, he suffered from dys-
phagia when swallowing both solid and liquid foods.
The patient had no pain while swallowing, but did ex-
perience coughing, and had lost ~16 kg in the last 4
years.
Upon physical examination, cervical motion was
limited in all directions; the occiput-wall distance was
12 cm and chin-manubrium sterni distance was 7 cm.
The patient had dorsal kyphosis with a Schober test
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Albayrak
İ
, et al. A rare cause of dysphagia
score of 1 cm. A SI compression test was bilaterally pos-
itive; the Bath AS disease activity index was 4.7; and
Bath AS functional index was 3.9. Laboratory tests
showed that the whole blood count was within normal
limits. Erythrocyte sedimentation rate was 25 mm/
hour, and C-reactive protein was 19.8 mg/L (normal
range,
≤
5). Blood levels of urea, creatinine, transami-
nases, calcium, and alkaline phosphatase were all with-
in normal limits. While he was negative for antinuclear
antibody and rheumatoid factor, he was positive for
HLA-B27.
Imaging studies revealed SI joint ankylosis and whis-
kering sign in pubic bones with simple pelvic radiogra-
phy (Fig. 1). A bamboo spine appearance was present
with simple thoracolumbar radiography (Fig. 2). Osteo-
phytes were observed at the anterior corners of vertebra
corpuses at C4-5 and C7-T1 levels with lateral cervical
radiography (Fig. 3). Esophagography with barium re-
vealed compression of the esophagus due to osteo-
phytes. Cervical computed tomography, performed to
localize the osteophytes and for differential diagnosis,
revealed fusion between the C5-C6-C7 corpuses with
prominent osteophyte formation anteriorly on the left
at the C7-T1 level. This formation compressed the
esophagus (Fig. 4). Cervical magnetic resonance imag-
ing (MRI) was performed to rule out factors originat-
ing from soft tissues as causes of dysphagia. A large os-
Figure 1.
Sacroiliac joint ankylosis and whiskering sign in
pubic bones in pelvic graphy (shown by the arrow).
Figure 2.
Bamboo spine appearance in the thoracolumbar
graphy (shown by the arrow).
Figure 3.
Osteophyte formation at the anterior corners of
vertebra corpuses at C4-5 and C7-T1 levels in cervical verte-
bra (shown by the arrow).
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The Korean Journal of Internal Medicine Vol. 28, No. 5, September 2013
teophyte formation on anterior vertebra corpuses at the
C7-T1 level, which caused compression on esophagus,
was defined in the cervical MRI (Fig. 5).
The patient was diagnosed with severe dysphagia be-
cause he could not swallow small solid foods and he
complained of coughing when swallowing liquid and
solid foods [3]. After consultation with an orthopedic
surgeon, an operation for osteophyte excision was pro-
posed, but the patient refused. Diet modification, anti-
reflux and swallowing therapy were prescribed. How-
ever, after 6 months, dysphagia had not improved.
DISCUSSION
AS is a systemic anti-inflammatory disease, accompa-
Figure 4.
(A, B) Prominent osteophyte formation and esophageal compression in the left anterior in cervical computed tomog-
raphy (shown by the arrows).
Figure 5.
(A, B) A large osteophyte formation on C4-C5 and C7-T1 vertebra corpuses and esophageal compression of this forma-
tion in the cervical magnetic resonance (shown by the arrows).
A
A
b
b
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İ
, et al. A rare cause of dysphagia
nied frequently by extra-articular involvement [1]. The
presence of HLA-B27 has an important role in AS
pathogenesis and is often concomitantly observed with
environmental factors such as infection [4,5]. AS affects
primarily vertebral joints and intervertebral disc dis-
tance, where lymphotoxic infiltration causes degenera-
tion in the disc interspaces. Ossification and autofu-
sion progressively follow the erosion of vertebral joints.
As a result, characteristic syndesmophyte formation,
expansive ankylosis, and radiological bamboo spine
appearance develop [6]. In our case, there was a large
anterior osteophyte on the cervical vertebra resulting
from ossification. This osteophyte compressed the
esophagus to cause the progressive development of dys-
phagia.
Early diagnosis and treatment of AS is key to prevent-
ing complications, such as compression of adjacent or-
gans by vertebral deformities. Initiation of swallowing
therapy in the early phases and a patient treatment plan
for AS are equally important since most patients with
cervical osteophytes can be managed conservatively [2].
Although dysphagia developed in the case reported
here, it was diagnosed after 4 years of detailed anamne-
sis. As a consequence, the patient did not respond to
conservative therapy that included diet modification,
swallowing and medical therapy, and the condition
progressed to a point that required surgical interven-
tion.
Cervical osteophytes are common but osteophytes
causing dysphagia due to compression of esophagus
are unusual. Abnormal osteoblast growth or activity in
ligamentous regions of bone is the pathogenesis of
DISH. The criteria for spinal involvement in DISH in-
cludes all of the following criteria: 1) presence of flow-
ing calcification and ossification along the anterolater-
al aspect of at least four contiguous vertebral bodies,
with or without associated localized pointed excres-
cences at the intervening vertebral body intervertebral
disc junctions; 2) presence of relative preservation of in-
tervertebral disc height in the involved vertebral seg-
ment and the absence of extensive radiographic chang-
es of degenerative disc disease; and 3) absence of
apophyseal joint bony ankylosis and SI joint erosion,
sclerosis or intra-articular osseous fusion. The case
presented here did not meet the DISH criteria, and in-
volvement of the SI and apophyseal joints was typical
for AS.
DISH usually begins after the age of 50, is more com-
mon in males, and is associated with endocrine and
metabolic syndromes [2]. A similar case reported the
coexistence of DISH and AS in a patient that developed
lung aspiration due to dysphagia secondary to anterior
cervical osteophytes. The patient was elderly at 80 years
of age and had osteophytes in the clavicle and shoul-
ders, as well as spinal stenosis due to DISH and the
posture and bamboo spine appearance suggesting AS
[2]. Our patient had onset of symptoms at age 25, morn-
ing stiffness, inflammatory back pain, and a positive
family history, which are all suggestive findings for AS.
He had no comorbid disease related with DISH.
Large osteophytes in cervical vertebra cause dyspha-
gia through several mechanisms. These include direct
compression of the pharynx or esophagus [7], tilt mech-
anism disorders of normal epiglottis at the laryngeal
inlet due to osteophytes at C3-C4 level [8], inflammato-
ry reactions in paraesophageal tissues [9], and crico-
pharyngeal spasm [10]. In our case, an anterior osteo-
phyte at the C7-T1 vertebrae caused dysphagia by
esophageal compression. DISH occasionally causes
dysphagia. Typically, osteophytes causing dysphagia
are in the C5 cervical interspace [2]. Again, our case had
a large osteophyte compressing the esophagus at the C7
cervical interspace, a localization not typical for DISH.
The patient reported here had a typical presentation
of dysphagia that started mildly and progressed gradu-
ally. Dysphagia is graded as none, mild, or moderate.
Mild dysphagia is defined as an abnormal sensation in
the pharynx while swallowing solid or liquid foods;
moderate dysphagia is defined as difficulty in swallow-
ing of large solid foods, but easy swallowing of small
amounts of liquid foods; severe dysphagia is defined as
inability to swallow small solid foods or development
of cough while swallowing [4]. These classifications can
be used to make either medical or surgical treatment
decisions. Conservative therapies, like diet modifica-
tion and swallowing therapy, can be employed in dys-
phagia of mild and moderate severity; whereas surgical
therapy is employed if there is no response to the con-
servative measures or in cases of severe dysphagia [11].
Indications for surgical therapy in moderate dysphagia
are not well described. Some studies advocate surgical
intervention in the early phases of moderate dysphagia
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The Korean Journal of Internal Medicine Vol. 28, No. 5, September 2013
[12], while others propose surgery to patients who do
not respond to medical therapy (i.e., nonsteroidal an-
ti-inflammatory drugs, steroids, myorelaxants, antire-
flux drugs) [13]. The patient described here had severe
dysphagia, indicating surgery according to this classi-
fication. However, he refused surgical intervention and
diet modification with antireflux and swallowing ther-
apy were prescribed.
In conclusion, anteriorly localized osteophytes in
cervical vertebrae may rarely cause dysphagia in AS.
Early diagnosis is important for conservative therapy
response and should be considered when evaluating
patients with AS.
Conflict of interest
No potential conflict of interest relevant to this article
is reported.
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