DISHphagia—a difficult problem to swallow

E. Clark, P. Preston, A. Wates and P. Merry

Rheumatology, NNUH, Norwich, UK

Correspondence to: E. Clark, Specialist Registrar in Rheumatology, Royal National Hospital for Rheumatic Diseases, Upper Borough Walls, Bath BA1 1RL.

SIR, We present a case report of a male patient seen in the rheumatology clinic with dysphagia secondary to diffuse idiopathic skeletal hyperostosis (DISH), and a review of the literature. Informed consent was obtained from the patient to publish this case report.

A 56-yr-old male was diagnosed as having DISH in 1998 after investigation into spinal pain. In February 2000 he presented with difficulty swallowing solids. He was not diabetic and, apart from hypertension, there were no medical problems.

On examination, his cervical movements were only 10% of normal in all directions. There was no abnormal neurology in the arms. Lateral radiographs of his cervical spine showed florid ossification anteriorly, resulting in fusion from C4 downwards. There was also a large osteophyte anteriorly at C3–4 (Fig. 1).



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FIG. 1. Preoperative lateral cervical spine radiograph showing florid anterior ossification, fusion from C4 downwards and a large osteophyte anteriorly at C3–4.

 
Video fluoroscopy demonstrated the large anterior osteophyte indenting the posterior aspect of the hypopharynx. Liquid barium flowed easily down either side of the osteophyte. With a solid bolus (marshmallow) there was laryngeal penetration and aspiration. As the tip of the epiglottis moved back, it opposed the osteophyte at C3–4 and did not go below this.

Cervical osteophytectomy via the anterolateral approach was undertaken. The large posterior osteophyte was excised and C3–4 were fused. Postoperatively, the dysphagia had completely resolved.

DISH is generally a benign disease and is found in 3.8% of males and 2.6% of females over the age of 40 [1]. Thoracic involvement occurs in 99% of cases. Cervical involvement, particularly of C4–7, is less common (75%). Dysphagia due to DISH involving the cervical spine is relatively unusual (17% of patients with DISH) [2].

DISH is characterized by calcification and ossification along the anterolateral aspects of contiguous vertebral bodies, with preservation of intervertebral disc height and absence of degenerative disc disease. It was originally described by Forestier and Rotes-Querol in 1950 as calcification of anterolateral perivertebral ligaments. In 1975 Resnick named the condition ‘diffuse idiopathic skeletal hyperostosis’, defining it as spinal and peripheral enthesopathy.

The aetiology of DISH is unknown. Positive associations include diabetes, obesity, hypertension, gout and Dupuytrens contractures [2].

Dysphagia due to osteophytic masses was first reported in 1905 by Zahn [3], and the first surgical removal of the osteophyte was carried out in 1926 by Mosher [4]. In 1971, Maran and Jacobson reported the 10th surgical case [5].

There are five possible ways that DISH causes dysphagia: (i) giant osteophytes may cause dysphagia by simple mechanical obstruction; (ii) smaller osteophytes may impinge at sites of relative immobility of the oesophagus (anatomically the oesophagus is anchored at the level of the cricoid cartilage and the diaphragm); (iii) inflammation in the immediate vicinity of the osteophyte, provoking oesophageal obstruction; (iv) pain and spasm [6]; and (v) secondary neurological lesions, e.g. recurrent nerve palsies provoked by the hyperostosis [7].

Lateral plain X-rays of the cervical spine will show the presence of anterior osteophytes. Video fluoroscopy will show the filling defect, pooling or aspiration. It may also show other unexpected causes of dysphagia, such as oesophageal tumours. Careful endoscopy has been advocated as there is a concern that irritation by the osteophyte may be associated with oesophageal carcinoma [8].

Various conservative therapies have been described, including the use of non-steroidal anti-inflammatory drugs, steroid pulses, skeletal relaxants, anti-reflux regimes and bisphosphonates [9]. Cervical surgery has proven to be useful in the management of severely affected patients [10] and many surgeons advocate excision combined with fusion of the affected vertebrae to reduce the incidence of relapse [11].

Rheumatologists should be aware of dysphagia in DISH, and in particular that it may be caused by osteophytes producing mechanical obstruction.

The authors have declared no conflicts of interest.

References

  1. Julkunen H. The epidemiology of hyperostosis of the spine together with its symptoms and related mortality in a general population. Scand J Rheumatol 1973;40:581–91
  2. Utsinger D. Diffuse idiopathic skeletal hyperostosis. Clin Rheum Dis 1985;11:325–51[ISI][Medline]
  3. Zahn H. Ein Fall von Abknicking der Speiserohre durch vertebrale Ekchondrosen. Munch Med Wochenschr 1905;52:1680–2
  4. Mosher HP. Exostoses of the cervical vertebrae as a cause for difficulty swallowing. Laryngoscope 1926;36:181–2
  5. Maran A, Jacobson I. Cervical osteophytes presenting with pharyngeal symptoms. Laryngoscope 1971;81:412–7[ISI][Medline]
  6. Masayoshi O. Dysphagia complications in ankylosing spinal hyperostosis and ossification of the posterior longitudinal ligament. Spine 1993;18:391–14[ISI][Medline]
  7. Coelto C. DISH with dysphagia: a neurological mechanism? Clin Exp Rheumatol 1995;13:268[ISI][Medline]
  8. Meeks LW. Vertebral osteophytosis and dysphagia. J Bone Joint Surg 1973;55:197–201[Medline]
  9. Fulton JD. Analgesic use of etidronate in Forestier's disease. Lancet 1992;340:1287
  10. Krause P. Cervical hyperostosis: a rare cause of dysphagia. Eur Spine J 1994;13:56–8
  11. Akhtar S. The management of dysphagia in skeletal hyperostosis. J Laryngol Otol 2000;114:154–7[CrossRef][ISI][Medline]
Accepted 5 March 2003





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