Two different causes of acute respiratory failure in a patient with diffuse idiopathic skeletal hyperostosis and ankylosed cervical spine

Abstract

We report a case of 73-year-old man with massive hyperostosis of the cervical spine associated with diffuse idiopathic skeletal hyperostosis (DISH), resulting in dysphagia, hoarseness and acute respiratory insufficiency. An emergency operation was performed, which involved excision of osteophytes at the level of C6-C7, compressing the trachea against enlarged sternoclavicular joints, also affected by DISH. Approximately 3 years later, the patient sustained a whiplash injury in a low impact car accident, resulting in a C3-C4 fracture dislocation, which was not immediately diagnosed because he did not seek medical attention after the accident. For the next 6 months, he had constant cervical pain, which was growing worse and eventually became associated with dysphagia and dyspnoea, ending once again in acute respiratory failure due to bilateral palsy of the vocal cords. The patient underwent a second operation, which comprised partial reduction and combined anteroposterior fixation of the fractured vertebrae. Twenty months after the second operation, mild hoarseness was still present, but all other symptoms had disappeared. The clinical manifestations, diagnosis and treatment of the two unusual complications of DISH are discussed.

Fig. 1

Radiograph of the cervical and upper thoracic spine, lateral view, revealed massive ossification of the anterior longitudinal ligament at C2–C6 (a) and large ventral osteophytes (arrowhead) at C6–C7 (b)

Fig. 2

CT of the thorax confirmed pronounced narrowing of the trachea (arrowhead) caused by large ventral osteophytes at C6–C7 (arrow)

Fig. 3

Radiograph of the cervical spine, lateral view, revealed a fracture dislocation at C3–C4, with anterior dislocation of C4 (arrow)

Fig. 4

Postoperative radiograph of the cervical spine: lateral (a) and antero-posterior views (b) show anterior stabilization of C3–C4 with a plate and screws (thick arrow) and lateral fixation with mass screws (thin arrow). Resected bone of the body of C4 was replaced by a cage filled with autologous bone (arrowhead). At C6–C7, anterior stabilization with a plate and screws (black arrowhead) was performed during the first operation

Fig. 5

CT of the sacroiliac joints revealed normal cartilaginous parts with no signs of erosion or sclerosis (arrow). Cranially, ossifications of the interosseous ligaments were present (arrowhead)