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DISH of the cervical spine causing epiglottis impingement.
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MedLine Citation:
PMID:  19881069     Owner:  NLM     Status:  In-Data-Review    
Abstract/OtherAbstract:
Diffuse idiopathic skeletal hyperostosis (DISH) is a condition characterized by calcification and ossification of ligaments and entheses; it mainly affects the vertebral column. We report the case of a patient with pharyngeal dysphagia and episodic aspiration secondary to DISH involvement of the cervical spine, which had caused alteration in the epiglottic tilt mechanism during deglutition.
Authors:
Tommaso Bartalena; Francesco Buia; Alberto Borgonovi; Maria Francesca Rinaldi; Cecilia Modolon; Francesco Bassi
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  The Indian journal of radiology & imaging     Volume:  19     ISSN:  1998-3808     ISO Abbreviation:  Indian J Radiol Imaging     Publication Date:    2009 Apr-Jun
Date Detail:
Created Date:  2009-11-02     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8503873     Medline TA:  Indian J Radiol Imaging     Country:  India    
Other Details:
Languages:  eng     Pagination:  132-4     Citation Subset:  -    
Affiliation:
Department of Radiology, S. Orsola University Hospital - via Massarenti 9, 40138, Bologna, Italy.
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Full Text
Journal Information
Journal ID (nlm-ta): Indian J Radiol Imaging
Journal ID (publisher-id): IJRI
ISSN: 0971-3026
ISSN: 1998-3808
Publisher: Medknow Publications, India
Article Information
© Indian Journal of Radiology and Imaging
open-access:
Print publication date: Month: 5 Year: 2009
Volume: 19 Issue: 2
First Page: 132 Last Page: 134
ID: 2765173
Publisher Id: IJRI-19-132
DOI: 10.4103/0971-3026.50831
PubMed Id: 19881069

DISH of the cervical spine causing epiglottis impingement
Tommaso BartalenaAF0001
Francesco BuiaAF0001
Alberto BorgonoviAF0001
Maria Francesca RinaldiAF0001
Cecilia ModolonAF0001
Francesco BassiAF0001
Department of Radiology, S. Orsola University Hospital - via Massarenti 9, 40138, Bologna, Italy
Correspondence: Correspondence: Dr. Tommaso Bartalena, Department of Radiology. S. Orsola University Hospital - via Massarenti 9, 40138, Bologna, Italy. E-mail: t.bartalena@email.it

Epiglottic tilt is a protective mechanism of the airways, which acts in the pharyngeal phase of deglutition. Prevertebral ossification in patients with DISH may cause mechanical impingement on the epiglottis with resultant incomplete closure of the laryngeal inlet during swallowing, putting the patient at risk for aspiration.


Case Report

An 83-year-old man was referred to our hospital with a long-standing history of dysphagia to liquids and solid food and episodic aspirations, along with the complaint of a sensation of having a lump in the back of the throat. His symptoms had worsened over the last 3 months. The patient also reported long-standing stiffness of his neck, with limitation of the range of motion.

Precontrast images [Figure 1A] showed diffuse, thick prevertebral ossification extending from C3 downward, which was responsible for a smooth bulge on the posterior wall of the pharynx. A congenital C2-C3 block vertebra was also present. The heights of the intervertebral spaces were preserved. Because of the history of aspiration, videofluoroscopy was performed with the administration of a water-soluble iodinated contrast medium (Gastrografin, 370 mg I/ml; Bayer Schering Pharma, Berlin, Germany). The oral phase of deglutition showed premature leakage of the bolus from the mouth into the valleculae because of age-related deficiency in the tongue volume. In the pharyngeal phase, the epiglottis failed to completely invert because of mechanical impingement due to the prevertebral ossification at the level of C3-C4, and it remained in a semiupright position during bolus passage [Figure 1, B and C]. Premature leakage of oral content into the pharynx and epiglottic tilt failure acted together to bring about laryngeal penetration. The esophageal phase was within normal limits. The radiologic findings were consistent with diffuse idiopathic skeletal hyperostosis (DISH) of the cervical spine causing dysphagia.

Because of his advanced age and cardiac comorbidity, surgery was not considered. The patient was given swallowing training and nonsteroidal anti-inflammatory drugs (NSAIDs), partially relieving his symptoms.


Discussion

DISH, also known as Forestier disease, is disorder of unknown origin. It was first described by Forestier and Rotes-Querol in 1950.[1] It is characterized by diffuse ossification and calcification of tendons, ligaments, and fasciae in both the axial and the appendicular skeleton. This disease has a predilection for men (65%) and is common in patients over the age of 50, with a prevalence of approximately 15–20% in the elderly population.[2, 3] The spinal column is most often affected, with the thoracic spine being most commonly involved (95% of the patients), followed by the lumbar and cervical spines.[4, 5]

According to Resnick,[6] the radiographic diagnostic criteria in the spine include: 1) osseous bridging along the anterolateral aspect of at least four vertebral bodies; 2) relative sparing of intervertebral disc heights, with minimal or absent disc degeneration; and 3) absence of apophyseal joint ankylosis and sacroiliac sclerosis.

The clinical manifestations are variable. Some subjects are completely asymptomatic, while others complain of back pain and stiffness and symptoms secondary to encroachment of the bony excrescences on neighboring structures. Pharyngoesophageal and tracheal compression may result in dysphagia, dyspnea, and stridor.[7] The reported incidence of dysphagia in patients with DISH ranges from 0.2–28%; dyspnea is less common.[8] Myelopathy associated with ossification of the posterior longitudinal ligament (OPLL) is another possible complication since OPLL has been described in association with DISH in up to 50% of cases.[9] DISH also predisposes to complications such as iatrogenic mucosal and laryngeal damage during endoscopic procedures and intubation.[10]

Conventional radiography is usually sufficient to confirm the diagnosis of DISH and dynamic videofluoroscopy should be reserved for demonstrating the exact relationship of the cervical spine alterations with the swallow function in those patients who are experiencing dysphagia.[11] CT and MRI may be used to better detect associated findings (e.g., OPLL) and complications (e.g., spinal canal stenosis and compressive myelomalacia).[5]

Dysphagia in patients with DISH involvement of the cervical spine develops mainly because of mechanical compression causing varying degrees of esophageal obstruction, impaired epiglottic motility, and distortion of the laryngeal cartilages. Moreover, chronic compression may result in an inflammatory process in the esophageal wall that can lead to fibrosis and adhesions, with fixation of the esophagus and disruption of the neural plexus.[8] Alterations of the upper cervical spine, especially at the C3-C4 level, may interfere with laryngeal function,[10] while more distal involvement may induce spasm of the upper esophageal sphincter (which is usually located at C5-C6) or esophageal compression.[8, 12] In our case, dynamic videofluoroscopy showed mechanical impingement between the prevertebral ossification at C3-C4 and the epiglottis, altering the pharyngeal phase of swallowing and leading to laryngeal penetration. Epiglottic tilt is a laryngeal protective mechanism that acts in the early part of the pharyngeal phase of deglutition to prevent penetration and aspiration of the bolus into the airways during swallowing. Epiglottic tilt occurs as a two-step procedure in the majority of patients. With the first movement, which is passive and induced by hyoid elevation, the epiglottis assumes a horizontal position. With the second movement, which is probably brought about by contraction of the thyroepiglottic muscle, the epiglottis finally reaches the inverted position, closing the laryngeal inlet.[12] Thus, inadequate tilt of the epiglottis may predispose to passage of bolus into the airways and the risk of aspiration pneumonia.

Patients with dysphagia due to DISH may be initially treated conservatively with dietary modifications and swallowing therapy. However, when the dysphagia is severe or when conservative treatment fails, surgical decompression with osteophytectomy via the perioral-transpharyngeal route for the C1 and C2 vertebrae or an anterior cervical approach for the C3 to C7 vertebrae should be considered.[13] Some authors recommend early surgery even when the patient has only mild dysphagia,[14] while others think that surgical intervention should be considered only after trials of medical therapy (NSAIDs, steroids, skeletal relaxants, anti-reflux drugs, etc.) have failed to provide relief.[11]


Conclusion

Videofluoroscopy is the main tool for investigating deglutition and is indicated in patients with DISH who experience dysphagia since it can provide exact correlation between the anatomical alterations of the cervical spine and functional impairment of the swallowing chain.


Notes

Source of Support: Nil

Conflict of Interest: None declared.

References
1. Forestier J,Rotes-Querol J. Senile ankylosing hyperostosis of the spineAnn Rheum DisYear: 195093213014800245
2. Resnick D. Degenerative Diseases of the Vertebral ColumnRadiologyYear: 19851563143923556
3. Weinfeld RM,Olson PN,Maki DD,Griffiths HJ. The prevalence of diffuse idiopathic skeletal hyperostosis (DISH) in two large American Midwest metropolitan hospital populationsSkeletal RadiolYear: 19972622259151370
4. Utsinger PD. Diffuse idiopathic skeletal hyperostosisClin Rheum DisYear: 198511325513899489
5. Cammisa M,De Serio A,Guglielmi G. Diffuse idiopathic skeletal hyperostosisEur J RadiolYear: 199827S7119652495
6. Resnick D,Niwayama G. Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis (DISH)RadiologyYear: 197611955968935390
7. Nelson RS,Urquhart AC,Faciszewski T. Diffuse Idiopathic Skeletal Hyperostosis: A Rare Cause of Dysphagia, Airway Obstruction, and DysphoniaJ Am Coll SurgYear: 20062029384216735209
8. Giger R,Dulguerov P,Payer M. Anterior Cervical Osteophytes Causing Dysphagia and Dyspnea: An Uncommon Entity RevisitedDysphagiaYear: 2006212596317216390
9. McAfee PC,Regan JJ,Bohlman HH. Cervical cord compression from ossification of the posterior longitudinal ligament in non orientalsJ Bone Joint Surg BrYear: 198769569753112161
10. Mader R. Clinical Manifestations of Diffuse Idiopathic Skeletal Hyperostosis of the Cervical SpineSemin Arthritis RheumYear: 200232130512430101
11. Aydin E,Akdogan V,Akkuzu B,Kirbaş. I, Ozgirgin ON. Six cases of Forestier syndrome, a rare cause of dysphagiaActa OtolaryngolYear: 2006126775816803720
12. Jones B,Donner MW. Normal and Abnormal SwallowingImaging in Diagnosis and TherapyYear: 1991New-YorkNY: Springer Verlag
13. Uppal S,Wheatley AH. Transpharyngeal approach for the treatment of dysphagia due to Forestier's diseaseJ Laryngol OtolYear: 1999113366810474677
14. McCafferty RR,Harrison MJ,Tamas LB,Larkins MV. Ossification of the anterior longitudinal ligament and Forestier's disease: An analysis of seven casesJ NeurosurgYear: 1995831377782829

Figures

[Figure ID: F0001]
Figure 1 (A–C) 

Spot films from a videofluoroscopy swallowing study. Precontrast image (A) shows diffuse thick prevertebral ossification (arrows) of the cervical spine. Intervertebral discs heights are preserved. A congenital C2-C3 block vertebra is seen. The tip of the epiglottis (at rest) is located at the C3-C4 level. Pharyngeal phase of deglutition (B, C). Hyoid bone and laryngeal elevation are seen, but the epiglottis fails to invert because of impingement by the C3-C4 ossification (arrowhead in B). Note pooling of contrast in the valleculae (asterisk in B) and laryngeal penetration (arrow in C)



Article Categories:
  • Head & Neck Radiology

Keywords: Diffuse idiopathic skeletal hyperostosis, dysphagia, videofluoroscopy.

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