Background: Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic non-inflammatory disorder characterized by enthesopathy and osteophyte formation. DISH can also cause several other symptoms. Limited range of motion (ROM) is the most common symptom; however, dysphagia and respiratory distress are clinically important symptoms. Dysphagia caused by cervical DISH is initially treated conservatively, but surgical treatment is performed when conservative treatment is ineffective. Although there are many reports on the surgical excision of osteophytes for refractory dysphagia, only a few reports on surgery for dysphagia caused by DISH associated with ossification of the posterior longitudinal ligament (OPLL) exist. Here, we report a rare case of cervical spinal cord injury following osteophyte excision for a respiratory distress and dysphagia caused by DISH associated with OPLL.
Case presentation: A 76-year-old male with hypertension and diabetes presented with dysphagia, respiratory insufficiency, and palpitations. Four months later, he experienced severe dyspnea and was hospitalized. His vital signs indicated respiratory distress, which led to intubation and tracheotomy due to his worsening condition. Imaging revealed massive anterior cervical osteophytes and multisegmental OPLL that caused spinal canal stenosis and tracheal compression. Surgical excision of the osteophytes was performed, but the patient later developed tetraplegia attributed to C5/C6 instability. Posterior fusion and laminoplasty were performed, resulting in neurological improvement but persistent dysphagia and motor deficits. He was transferred to another hospital for rehabilitation but died of aspiration pneumonia.
Conclusions: Patients with cervical OPLL and spinal cord compression may experience spinal cord injury when intervertebral mobility is slightly increased due to osteophyte excision. If dysphagia or respiratory distress occur in patients with DISH and OPLL, decompression and fusion surgery at the mobile segment is required, in addition to osteophyte excision surgery. Posterior decompression and fusion surgery should be performed before anterior osteophyte excision surgery to avoid implant infection, particularly in patients with respiratory distress who have undergone tracheostomy. Patients receiving long-term mechanical ventilation are less likely to recover their swallowing function and should undergo a total laryngectomy.
Keywords: Diffuse idiopathic skeletal hyperostosis; Forestier’s disease; Ossification of posterior longitudinal ligament; Osteophyte excision.
© 2024. The Author(s).