A decade of unexplained dysphagia.

A 72-year-old man was referred to the Department of Neurosurgery at the John Radcliffe Hospital for surgical management of dysphagia, the underlying cause of which had eluded diagnosis for almost 10 years. At his initial presentation nearly a decade earlier, he had reported symptoms of high dysphagia for solid foods and episodes of coughing and choking during meals. Findings on the physical examination at that time were normal, and a barium swallow test detected no obvious abnormality. A provisional diagnosis of mild neuromuscular incoordination of the pharynx was made.

[FIGURE 1 OMITTED]

Over a number of years, the patient's symptoms progressed. A videofluoroscopic swallow examination demonstrated a delayed and inconsistent cough reflex, evidence of silent aspiration, and pooling of secretions in the valleculae and piriform sinuses. In the cervical spine, an x-ray showed large anterior osteophytes at C2 and C3 (figure 1, A). These osteophytes were found to interrupt bolus flow and impede the action of pharyngeal constrictors. Magnetic resonance imaging (MRI) (figure 1, B) and computed tomography of the cervical spine confirmed an underlying diagnosis of diffuse idiopathic skeletal hyperostosis (DISH).

Surgical excision of the osteophytes was achieved via an anterior cervical approach. At the 3-month follow-up, the patient reported a significant reduction of his dysphagia. Postoperative cervical x-rays confirmed that a satisfactory decompression of the pharynx had been accomplished (figure 2).

Also known as Forestier disease, DISH is estimated to affect about 10% of the population. (1) It is more common in men, and its prevalence increases with age (35% in men older than 70 years); it is also more severe in men. (2) In some cases DISH is asymptomatic, and the diagnosis is made solely on the basis of radiographic criteria established by Resnick and Niwayama. (3) In rare cases, DISH affects the cervical spine and can cause compression of structures within the neck, leading to dysphagia or even airway obstruction. (4) More common conditions such as ankylosing spondylitis and cervical spondylosis may also cause pronounced anterior osteophyte formation of the cervical vertebrae and consequent dysphagia. (5)

[FIGURE 2 OMITTED]

References

(1.) Julkunen H, Heinonen OP, Knekt P, Maatela J. The epidemiology of hyperostosis of the spine together with its symptoms and related mortality in a general population. Scand J Rheumatol 1975; 4(1):23-7.

(2.) Weinfeld RM, Olson PN, Maki DD, Griffiths HI. The prevalence of diffuse idiopathic skeletal hyperostosis (DISH) in two large American Midwest metropolitan hospital populations. Skeletal Radiol 1997;26(4):222-5.

(3.) Resnick D, Niwayama G. Radiographic and pathologic features of spinal involvement in diffuse idiopathic skeletal hyperostosis (D1SH). Radiology 1976;119(3):559-68.

(4.) Nelson RS, Urquhart AC, Faciszewski T. Diffuse idiopathic skeletal hyperostosis: A rare cause of dysphagia, airway obstruction, and dysphonia. J Am Coll Surg 2006;202(6):938-42.

(5.) Ladenheim SE, Marlowe FI. Dysphagia secondary to cervical osteophytes. Am J Otolaryngol 1999;20(3):184-9.

Christopher Burgess, BM, BCh; Richard Hughes, MRCS; Stewart Griffiths, FRCS (SN); Thomas Cadoux-Hudson, FRCS (SN)

From the Department ENT Surgery, Wexham Park Hospital, Slough, Berkshire, U.K. (Mr. Burgess), and the Department of Neurosurgery, John Radcliffe Hospital, Oxford, U.K. (Mr. Hughes, Mr. Griffiths, and Mr. Cadoux-Hudson).