Airway management for patients with ossification of the anterior longitudinal ligament of the cervical spine
© The Author(s) 2015
Received: 12 May 2015
Accepted: 22 June 2015
Published: 31 December 2015
Ossification of the anterior longitudinal ligament (OALL), also called Forestier’s disease or diffuse idiopathic skeletal hyperostosis, is characterized by anterior bridging osteophytes of unknown etiology. OALL may cause dysphagia, dyspnea, dysphonia, and acute airway obstruction. We report difficulty in tracheal intubation during anesthesia induction in two OALL patients. In an 82-year-old man, anterior bridging osteophytes (of the cervical region) were observed on preoperative lateral radiograph after several attempts of tracheal intubation for the operation of the anterior fusion of cervical spine. During the same procedure in another 69-year-old man, fiberoptic-assisted awake intubation was extremely difficult because of posterior hypopharyngeal wall protuberance by osteophytes of cervical spine; although tracheal intubation for anesthesia was uneventful on two previous occasions over the months. OALL is usually asymptomatic, but it has been found in 12 % of autopsies and may exaggerate with age. Dysphagia, difficulties with tracheal and/or gastric intubation, acute respiratory compromise, and sleep apnea result from the presence of cervical osteophytes. Anesthesiologists should be aware that tracheal intubation for such patients may be difficult, and thus the preoperative evaluation and airway management need careful consideration.
KeywordsOssification of the anterior longitudinal ligament Forestier’s disease Diffuse idiopathic skeletal hyperostosis Difficult intubation
Ossification of the anterior longitudinal ligament (OALL), also called Forestier’s disease or diffuse idiopathic skeletal hyperostosis (DISH), is a non-inflammatory disease characterized by the presence of anterior bridging osteophytes of unknown etiology. Although it is asymptomatic in some cases, this disease may produce dysphagia, dyspnea, dysphonia, and (exceptionally) acute airway obstruction . Difficulties in tracheal intubation, as well as aspiration pneumonia and spinal cord injury, are potential problems in anesthetic management. Herein, we report two patients with OALL in which airway problems occurred during the induction of anesthesia and tracheal intubation.
A 69-year-old man was scheduled for an off-pump coronary artery bypass graft (CABG) by way of a left anterolateral thoracotomy. His chief complaint was dysesthesia of the left arm caused by ossification of the posterior longitudinal ligament (OPLL). However, his preoperative ECG suggested an old antero–septal myocardial infarction, and subsequent coronary angiography revealed occlusion of both the left anterior descending branch and left circumflex artery, so CABG was planned first. He had a history of hypertension and renal insufficiency (creatinine clearance 49.9 mL/min). Upon physical examination, his neck movement was slightly restricted, but his dysesthesia was not made worse by neck movement. He could open his mouth widely, without limitation, and his Mallampati score was Class II. The chest radiograph was unremarkable. In the operating room, after the establishment of noninvasive monitors (five-lead ECG and pulse oximeter), an arterial catheter was inserted at the left radial artery for arterial blood pressure monitoring, and general anesthesia was induced with intravenous midazolam and fentanyl. After muscle relaxation had been induced using vecuronium, direct laryngoscopy was performed, and we assigned a Cormack Lehane score of Class II. We succeeded with left bronchial intubation at the first attempt. Insertion of transesophageal echocardiography was easy, too. The procedure was completed uneventfully. His vital signs were stable, and he was transported to the ICU under sedation. His postoperative course was uneventful.
Two months after the CABG, he was scheduled for posterior fusion of the cervical spine. After general anesthesia had been induced using thiopental and fentanyl, and muscle relaxation using vecuronium, direct laryngoscopy was performed. However, only the epiglottis could be seen. We therefore decided to use an airway scope (AWS; Pentax, Tokyo, Japan), and by this means, visualization of his vocal cords was achieved easily. After the surgical procedure had been completed, his endotracheal tube was extubated in the operation room without airway problems.
Ossification most notably affects the anterior longitudinal ligament of the spine; a condition called OALL. It mostly affects men, and the mean age of the patients is 60 years . The thoracic region is most commonly affected, followed by the lumbar and cervical regions . Ossification does not always occur solely in the vertebral column, it can occur in other parts of the musculoskeletal system too . There are three radiologic criteria for the diagnosis of OALL: osseous bridging of at least four contiguous vertebral bodies, preservation of intervertebral disc height, and absence of sacroiliac or apophyseal joint ankylosis . A number of possible causal factors of metabolic, environmental, or endocrine origin have been investigated, but no conclusive results have been obtained [2, 3].
OALL has been reportedly found in 12 % of autopsies, but it is generally asymptomatic [2, 3]. Indeed, it is often discovered coincidentally as a remarkable radiologic finding when a radiograph is solicited for the other disease . Dysphagia, difficulties with tracheal and/or gastric intubation, acute respiratory compromise, and sleep apnea has been reported to result from the presence of cervical osteophytes [2–4]. The cervical hyperostosis associated with OALL may lead to symptoms resulting from compression and/or distortion of the airway and/or the alimentary tract . Complications from these cervical osteophytes are rare, although they can occasionally be severe, even life-threatening . The level of the cervical spine at which osteophytes form has been postulated to correlate with the presenting symptoms. The most commonly involved vertebrae are C4–C5, followed by C5–C6 and C3–C4 . Lower cervical vertebrae, around the levels of C4, C5, and C6, may impinge on the esophagus and distal trachea, where it is tethered at the level of the cricoids, whereas the upper cervical spine may impinge more on the oropharynx, causing globus, stridor, or respiratory compromise . Although cervical radiographs may show cervical osteophytes, enhanced CT with multiplanar reformatted has proved to be the best diagnostic method for the localization of laryngeal compression and can be of help in the planning of anesthesia . If laryngeal compression is suspected, a preoperative sagittal CT is also helpful for the evaluation of airway obstruction and for the planning of tracheal intubation. Our two cases suggest that although the difficulties in tracheal intubation resulting from OALL are rare, anesthesiologist should suspect the presence of OALL, whenever they encounter unexplained difficulty in airway management in a patient who has no airway signs and/or no remarkable previous anesthesia history.
For patients with OALL, fiberoptic-assisted awake intubation is perhaps the safest choice when difficult intubation is suspected [5, 6]. Since great difficulty in tracheal intubation may occur secondary to anterior bulging of the posterior pharyngeal wall, anterior laryngeal displacement, angulation of the trachea distally and neck stiffness , and hyperostosis on the ligaments around the larynx , careful evaluation of the epiglottis during laryngoscopic examination is mandatory if such difficulties are to be avoided. In fact, tracheotomy may be indicated in such cases to avoid a catastrophic event.
Furthermore, osteophytes are likely to be missed in routine airway examination by frontal radiography. Therefore, it is necessary that preoperative radiography is evaluated including the lateral view, if the patient has diagnosis of OALL. And airway management of OALL patients needs careful consideration and modifications made if necessary, dependent upon progress, in each case.
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- Nelson RS, Urquhart AC, Faciszewski T. Diffuse idiopathic skeletal hyperostosis: a rare cause of dysphagia, airway obstruction, and dysphonia. J Am Coll Surg. 2006;202:938–42.View ArticlePubMedGoogle Scholar
- Caminos CB, Cenoz IZ, Louis CJ, Otano TB, Esain BF, Perez de Ciriza MT. Forestier disease: an unusual cause of upper airway obstruction. Am J Emerg Med. 2008;26:e1–3.View ArticlePubMedGoogle Scholar
- Karkas AA, Schmerber SA, Gay EP, Chahine KN, Righini CA. Respiratory distress and vocal cord immobilization caused by Forestier’s disease. Otolaryngol Head Neck Surg. 2008;139:327–8.View ArticlePubMedGoogle Scholar
- Gokce A, Beyzadeoglu T, Hanci L, Erdogan F. Diffuse idiopathic skeletal hyperostosis as a cause of acute respiratory distress in early postoperative period of total knee arthroplasty. Arch Orthop Trauma Surg. 2007;127:553–5.View ArticlePubMedGoogle Scholar
- Ozkalkanli MY, Katircioglu K, Ozkalkanli DT, Savaci S. Airway management of a patient with Forestier’s disease. J Anesth. 2006;20:304–6.View ArticlePubMedGoogle Scholar
- Thompson C, Moga R, Crosby ET. Failed videolaryngoscope intubation in a patient with diffuse idiopathic skeletal hyperostosis and spinal cord injury. Can J Anesth. 2010;57:679–82.View ArticlePubMedGoogle Scholar
- Bougaki M, Sawamura S, Matsushita F, Hanaoka K. Difficult intubation due to ossification of the anterior longitudinal ligament. Anaesthesia. 2004;59:303–4.View ArticlePubMedGoogle Scholar