Diffuse Idiopathic Skeletal Hyperostosis (DISH)—A Rare Etiology of Dysphagia

Article (PDF Available)inClinical Medicine Insights: Arthritis and Musculoskeletal Disorders 4(4):71-5 · September 2011with38 Reads
DOI: 10.4137/CMAMD.S6949 · Source: PubMed
Abstract
A 72-year-old gentleman presented to the hospital with progressively worsening dysphagia to soft foods and liquids. He was diagnosed with severe pharyngeal dysphagia by modified barium swallow. A CT scan of the neck with IV contrast showed anterior flowing of bridging osteophytes from C3-C6, indicative of DISH, resulting in esophageal impingement. He underwent resection of the DISH segments. Following the surgery, a PEG tube for nutrition supplementation was placed. However, the PEG tube was removed after five months when the speech and swallow evaluation showed no residual dysphagia. DISH is a rare non-inflammatory condition that results in pathological ossification and calcification of the anterolateral spinal ligaments.
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Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4 71–75
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Clinical Medicine Insights: Arthritis and
Musculoskeletal Disorders
CASE REPORT
Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4 71
Diffuse Idiopathic Skeletal Hyperostosis (DISH)—A Rare
Etiology of Dysphagia
Balakumar Krishnarasa, Abhirami Vivekanandarajah, Lucinda Ripoll, Edwin Chang and Robert Wetz
Staten Island University Hospital, Staten Island, NY, USA. Corresponding author email: doc_bala@yahoo.com
Abstract: A 72-year-old gentleman presented to the hospital with progressively worsening dysphagia to soft foods and liquids. He was
diagnosed with severe pharyngeal dysphagia by modied barium swallow. A CT scan of the neck with IV contrast showed anterior
owing of bridging osteophytes from C3-C6, indicative of DISH, resulting in esophageal impingement. He underwent resection of the
DISH segments. Following the surgery, a PEG tube for nutrition supplementation was placed. However, the PEG tube was removed
after ve months when the speech and swallow evaluation showed no residual dysphagia. DISH is a rare non-inammatory condition
that results in pathological ossication and calcication of the anterolateral spinal ligaments.
Keywords: DISH, dysphagia, diffuse idiopathic skeletal hyperostosis
Krishnarasa et al
72 Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4
Introduction
Diffuse idiopathic skeletal hyperostosis (DISH) with
cervical involvement causing dysphagia is a rare
entity. It was described as an abnormal ossication
along the anterior and lateral aspects of the vertebral
column in 1950 by Forestier and Rotes-Querol.
1
We
report a case of a patient with progressive dysphagia
secondary to DISH.
Case Presentation
A 72-year-old gentleman with coronary artery disease,
atrial brillation, hypertension, gout, and obstructive
sleep apnea presented to the hospital with progres-
sively worsening dysphagia to soft foods and liquids
for a few days. He had begun to experience dysphagia
ve years ago and had restricted his diet to mainly
soft foods and liquids. He denied fatigue, neck pain,
drooping of eye lids, difculty in breathing, hoarse-
ness, dysarthria, numbness or limb weakness. Sur-
gical history was signicant for bilateral cataract
surgeries and childhood exploratory laporotomy after
a traumatic incident. He denied a family history of
cancer. His medications included warfarin, digoxin,
atenolol, lovastatin, colchicine, hydrocholorothiaz-
ide, omeprazole, albuterol and ipratropium inhalers.
He used to smoke 1 pack per day for 40 years, but
quit 20 years ago. Esophagogastroduodenoscopy
and colonoscopy were done three years ago and
were unremarkable. Vital signs were stable and the
physical exam was unremarkable. Laboratory tests,
including an autoimmune panel were normal. CT
scan of the head was normal. A speech and swal-
low evaluation revealed dysphagia to thin liquids.
A modied barium swallow (MBS) revealed severe
pharyngeal dysphagia. CT of the neck with IV con-
trast showed anterior owing of bridging osteo-
phytes from C3-C6, indicative of DISH, causing
esophageal impingement (Fig. 1). He also had a MRI
of the neck with gadolinium that showed anterior
owing of bridging osteophytes from C3-C6 con-
sistent with diffuse idiopathic skeletal hyperostosis,
C3-C6 spinal stenosis and degenerative changes at
C3-C4 (Fig. 2). He underwent corpectomy of C4
with C3-C5 spinal fusion and resection of the C3-C6
DISH segments. Although post-operative radiologi-
cal studies showed an anatomical improvement, an
esophagram revealed residual dysphagia (Fig. 3).
He was discharged with a percutaneous endoscopic
gastrostomy (PEG) tube. Five months later, he
underwent speech and swallow evaluation, which
showed marked functional improvement and the
PEG tube was removed.
Discussion
Diffuse idiopathic skeletal hyperostosis, also known as
Forestiers disease,
2
is a non-inammatory condition
Figure 1. CT of neck with IV contrast (bone window) showing anterior ow-
ing of bridging osteophytes (arrows) from C3-C6, indicative of DISH.
Figure 2. MRI of neck with gadolinium showing anterior owing of bridg-
ing osteophytes (arrows) from C3-C6 consistent with DISH, C3-C6 spinal
stenosis and degenerative changes at C3-C4.
A rare etiology of dysphagia
Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4 73
with pathological ossication and calcication of the
anterolateral spinal ligaments and attachment sites
of the tendons and ligaments. Ossication along
the posterior vertebral border has also been noted.
3
While etiology of DISH remains unknown, various
hypotheses were proposed relating to mechanical
factors, diet, metabolic conditions and environmental
exposures.
4
Specically, mechanical factors leading
to ligament stretching, high uoride levels in water,
excess of vitamin A, long term isotretinoin therapy,
and metabolic conditions that produce increased
insulin-like growth factor-1 to stimulate osteoblasts
have been documented as potential causative factors.
5
While some conrmation came from clinical stud-
ies, there were conicting results reported as well.
The pathogenesis of DISH has been explained as an
enthesopathy due to pathological osteoblast differen-
tiation and ossication.
6
Prevalence is higher in men
(typically in their 60s) than women.
1
The incidence
increases with age and varies by population.
Symptoms of DISH depend on the localization
and involvement of adjacent structures. Patients
may experience neck, thoracic spine, lumbar spine
and/or extremity pain and disability. Spinal morning
stiffness is experienced by the majority of patients.
Dysphagia and symptoms pertaining to the knee,
shoulder and elbow are less common. Dysphagia,
odynophagia, foreign body sensation, stridor,
obstructive sleep apnea, and aspiration of solids and
liquids may occur when large anterior osteophytes
of the cervical spine impinge or displace the adja-
cent structures of the gastrointestinal and respiratory
tracts.
7
Dysphagia is usually progressive and greater
for solids than liquids. The osteophytes arising from
the C5-C6 level frequently cause dysphagia, followed
by C4-C5.
8
Stridor may be a rare manifestation of the
large C2-C3 anterior osteophyes, while point pres-
sure between osteophytes and the posterior cricoid
cartilage results in hypopharyngeal ulceration caus-
ing odynophagia.
9
Spinal cord compression can hap-
pen in patients with posterior ligament ossication
and this may lead to myelopathy and radiculopathy.
10
All these symptoms can manifest due to mechani-
cal obstruction, inammation in the immediate sur-
rounding of the osteophytes or spasm due to pain.
Presumably, a summation of all these factors plays
a role in producing the symptoms. Physical ndings
include decrease range of spinal motion, particularly
when the thoracic segment is involved, and palpable
nodules when the appendicular skeleton is involved.
Neurological ndings may be elicited when poste-
rior ligament calcication involves the spinal cord.
Differential diagnoses for DISH include Ankylosing
spondylitis (AS) and Spondylosis deformans (SD).
There are spurs seen in the region of the cervical
and lumbar spine similar to DISH but involvement
of the anterior longitudinal ligament in the thoracic
region is not seen in SD. AS is more common in
males than females and there is an association with
ligamentous ossication and syndesmophytes simi-
lar to DISH but the anterior longitudinal ligament is
not involved and the bony bridges involve the outer
margin of the annulus brosis. In addition, erosions
and bony ankylosis of the sacroiliac and apophy-
seal joints are seen in AS. Radiological imaging of
the spine will show squaring of the vertebrae also
known as “bamboo spine.” Other conditions that
may be associated with DISH include gout,
11
rheu-
matoid arthritis,
12
Paget’s disease
13
and chondro-
calcinosis.
14
Evaluation of progressive dysphagia
should include speech and swallow evaluation,
Figure 3. Post-operative esophagram showing residual dysphagia (arrow)
[uid entering esophagus and trachea] and anatomical correction of
osteophytes.
Krishnarasa et al
74 Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4
barium swallow studies, motility studies, otolaryn-
gologic examination, and other radiological studies,
which include CT and MRI of the head and neck.
Endoscopy should be utilized with caution as this
may cause perforation of esophagus.
15
An emphasis
is placed on diagnostic imaging to diagnose DISH.
The radiological ndings of DISH may occur in the
absence of clinical symptoms. Resnick and Utsinger
proposed radiographic criteria for the diagnosis of
DISH.
5
The Resnick radiographic criteria for the
diagnosis of DISH include: (a) the presence of ow-
ing ossication and calcication along the antero-
lateral aspect of at least four contiguous vertebral
bodies, (b) the preservation of intervertebral disk
heights in the involved vertebrae, (c) the absence of
apophyseal joint bony ankylosis and sacroiliac joint
erosion.
16
Extra-spinal ossication in DISH may
occur at ligamentous attachments and para-articular
soft tissues.
17
There are no specic laboratory abnor-
malities that have been associated with DISH.
Treatment of DISH depends on the presence of
symptoms. Physical activity and physical therapy
with heat can give symptomatic relief in patients with
DISH. The pain associated with DISH is treated simi-
lar to patients with osteoarthritis. Dysphagia, radicu-
lopathy, myelopathy, recurrent laryngeal nerve palsy,
and stridor need surgical intervention.
11
Conclusion
DISH is an uncommon etiology of dysphagia, which
is a common symptom. There have been other case
reports that have described dysphagia as a manifesta-
tion of DISH.
18,19
Although a rare cause of dysphagia,
DISH can cause considerable morbidity in the geri-
atric population. Keeping this clinical entity in the
differential diagnosis is important in arriving at the
diagnosis.
Disclosures
Author(s) have provided signed conrmations to the
publisher of their compliance with all applicable legal
and ethical obligations in respect to declaration of
conicts of interest, funding, authorship and contribu-
torship, and compliance with ethical requirements in
respect to treatment of human and animal test subjects.
If this article contains identiable human subject(s)
author(s) were required to supply signed patient con-
sent prior to publication. Author(s) have conrmed
that the published article is unique and not under con-
sideration nor published by any other publication and
that they have consent to reproduce any copyrighted
material. The peer reviewers declared no conicts of
interest.
References
1. Forestier J, Rotes-Querol J. Senile ankylosing hyperostosis of the spine.
Ann Rheum Dis. 1950;9:321–30.
2. Resnick D, Shapiro RF, Wiesner KB, et al. Diffuse idiopathic skeletal
hyperostosis (DISH). Semin Arthritis Rheum. 1978;7:153.
3. Resnick D, Guerra J Jr, Robinson CA, Vint VC. Association of diffuse idio-
pathic skeletal hyperostosis (DISH) and calcication and ossication of the
posterior longitudinal ligament. AJR. 1978;131:1049–53.
4. Ohishi H, Furukawa K, Iwasaki K, et al. Role of prostaglandin I2 in the gene
expression induced by mechanical stress in spinal ligament cells derived
from patients with ossication of the posterior longitudinal ligament.
J Pharmacol Exp Ther. 2003;305:818.
5. Utsinger PD, Resnick D, Shapiro R. Diffuse skeletal abnormalities in Forestier
disease. Arch Intern Med. 1976;136:763.
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cervical spine. N Engl J Med. 1960;263:11–4.
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14. Bruges-Armas J, Couto AR, Timms A, et al. Ectopic calcication among
families in the Azores: Clinical and radiologic manifestations in families
with diffuse idiopathic skeletal hyperostosis and chondrocalcinosis. Arthritis
Rheum. 2006;54:1340.
15. Smith EEK, Tanner NC. The complications of gastroscopy and o esophogoscopy.
Br J Surg. 1956;43:396–403.
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18. Aydin E, Akdogan V, Akkuzu B, et al. Six cases of Forestier syndrome,
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2010 Jul 17.
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A rare etiology of dysphagia
Clinical Medicine Insights: Arthritis and Musculoskeletal Disorders 2011:4 75
    • "Interestingly, the presence of radiographical features of DISH does not correlate with back pain [4] . In severe cases, dysphagia may occur and surgical treatment is required [5]. Conservative treatment includes anti-inflammatory drugs and corticosteroids [6]. "
    [Show abstract] [Hide abstract] ABSTRACT: In diffuse idiopathic skeletal hyperostosis (DISH), recurrence of hyperostosis after surgical removal is common. While both non-steroidal anti-inflammatory drugs (NSAIDs) and radiotherapy have been used in the prevention of heterotopic ossification, only NSAIDs have been applied for prophylaxis in DISH. As a previously undescribed prophylaxis of recurrence in DISH, we report a case treated with local radiotherapy.
    Full-text · Article · Jan 2014
    • "DISH and ankylosing spondylitis are the most common causes of bony outgrowth of the cervical spine, causing dysphagia and aspiration in the elderly5,9). However, with depending their size and location, symptoms such as dysphagia or dyspnea, may occur. "
    [Show abstract] [Hide abstract] ABSTRACT: Symptomatic diffuse idiopathic skeletal hyperostosis (DISH) is not common. Gelfoam is one of the most commonly used topical hemostatic agents. But, in the partially moistened state, air retained in its pores may result in excessive expansion on contact with liquid. The onset of swallowing difficulty after anterior cervical spine surgery due to appling gelfoam is a rare complication. A 77-year-old man with swallowing difficulty was admitted to our hospital and we diagnosed him as DISH confirmed by radiological study. After removing the DISH, patient's symptom was relieved gradually. However, on postoperative day (POD) 7, the symptom recurred but lesser than the preoperative state. We confirmed no hematoma and esophageal perforation on the operation site. We observed him closely and controlled the diet. Three months later, he had no symptom of swallowing difficulty, and was able to be back on a regular diet, including solid foods. We present a complication case of swallowing difficulty occurring by gelfoam application.
    Full-text · Article · Jun 2013
  • Full-text · Article · Oct 2015
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