CLINICAL STUDY
Dysphagia and Airway Obstruction Due to Large Cervical Osteophyte in a Patient With Ankylosing Spondylitis Yasin Kürşad Varsak, MD, Mehmet Akif Eryilmaz, MD, and Hamdi Arbağ, MD Abstract: Anterior cervical osteophytes are excessive bony formation of cervical vertebra bodies. They are common but rarely symptomatic lesions mostly seen in geriatric population. Large anterior cervical osteophytes may cause symptoms such as dysphagia, dyspnea, dysphonia, and odynophagia. They have been attributed to multiple etiologies including diffuse idiopathic skeletal hyperostosis, following trauma, cervical spondylitis, and infectious spondylitis. However, symptomatic large anterior cervical osteophyte with ankylosing spondylitis is extremely rare. Surgical excision is the main treatment for symptomatic cases. We report a case of a 53-year-old man with airway obstruction and dysphagia due to large cervical osteophyte who has a history of ankylosing spondylitis, and we also addressed the etiological factors and management of large symptomatic cervical osteophytes. Key Words: Ankylosing spondylitis, anterior cervical osteophytes, dysphagia (J Craniofac Surg 2014;25: 1402–1403)
O
steophytes are bony outgrowths found at the articular margins in many musculoskeletal disorders. Cervical vertebras are one of the most commonly effected bones. Anterior cervical hypertrophic spurs of the anterior cervical spine may occur in 20% to 30% of the population.1 Although cervical osteophytes are asymptomatic, they may lead to symptoms such as dysphagia, cough, dyspnea, and dysphonia. If symptomatic, dysphagia appears to be the most common presentation, caused by mechanical obstruction of the pharyngoesophageal segment by anterior cervical hyperostosis. Mosher,2 in 1926, first described 2 patients with dysphagia caused by large anterior cervical osteophytes. Large anterior cervical osteophytes are associated with senile degenerative skeletal disease, posttraumatic osteophytogenesis, idiopathic skeletal hyperostosis (DISH), cervical spondylitis, and infectious spondylitis, but ankylosing spondylitis (AS) has been very rarely mentioned as an etiological factor in the literature.3,4
PATIENT A 53-year-old man was referred to our clinic for airway obstruction, which caused failed intubation for appointed hip surgery. The patient was complaining of a slowly progressive dysphagia and, recently, the onset of dyspnea. The dysphagia has been From the Department of Otorhinolaryngology, Head and Neck Surgery, Meram Medical Faculty, Necmettin Erbakan University, Meram, Konya, Turkey. Received January 25, 2014. Accepted for publication February 16, 2014. Address correspondence and reprint requests to Yasin Kursad Varsak, MD, Department of Otorhinolaryngology, Head and Neck Surgery, Meram Medical Faculty, Necmettin Erbakan University, Meram, 42080 Konya, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000933
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present for 6 years, and the dyspnea has been present for 3 weeks. The patient has a medical history of AS for 17 years. There was a noticeable kyphosis on physical examination. Oropharyngeal examination revealed a 2- to 3-cm submucosal lesion with a smooth surface and firm margin, which narrows the oropharyngeal inlet. The lesion was hard on palpation. Indirect laryngoscopy revealed a smooth bulge in the posterior wall of the pharynx without mucosal lesions. The bulge was rigid and nonpulsatile. The patient was examined through spiral computed tomography (CT) of the cervical spine. It demonstrated a large hypertrophic anterior cervical osteophyte of C2-C3 vertebra (Fig. 1). Three-dimensional reconstruction of CT image was obtained as well (Fig. 2). First, the airway was secured under local anesthesia via tracheotomy, then the patient underwent transoral cervical osteophytectomy surgery under general anesthesia (Fig. 3). Pathologic findings macroscopically revealed a 4.5 4 1.5-cm gray bone colored solid bony tissue (Fig. 4) and microscopically revealed a trabecular mature bone tissue with marginal cartilaginous tissue. The patient was decannulated at postoperative day 8. Postoperative control lateral cervical x-ray film has shown no osteophytes at the posterior pharyngeal wall (Fig. 5). There was no complaint of dysphagia and dyspnea 12 months after the surgery.
DISCUSSION Large osteophyte of the cervical spine presenting with dysphagia is a rare condition that can be seen more in elderly patients. In this case, we report a patient with anterior cervical osteophyte that is secondary to AS. Ankylosing spondylitis is a chronic inflammatory seronegative rheumatic arthritis.5 Typically, this process first affects the sacroiliac joints, later progressing to involve the hips and spine.6 Anterior cervical osteophytes are associated with senile degenerative skeletal disease, posttraumatic osteophytogenesis, DISH, cervical spondylitis, and infectious spondylitis. Most patients with dysphagia based on cervical hyperostosis are attributed to DISH.7 The etiology of this condition is unknown, but researchers have found strong correlations with metabolic disorders, especially high body mass index and insulin-independent diabetes mellitus.8 Baraliakos et al9 has shown that the rates of new bone formation in AS and DISH are largely similar. Whereas syndesmophytes are more frequent in AS, patients with DISH have more degenerative bone spurs.9 However, there are only a few cases of symptomatic large anterior cervical osteophytes with underlying AS in the literature.10–12 Osteophytes of the anterior cervical spine are common in elderly patients and are usually asymptomatic. They may impinge on the pharynx or esophagus and can cause dysphagia, dyspnea, or stridor. When a patient known to have a cervical vertebral bone spur complains of dysphagia, it must be determined whether the protuberance is the singular cause of the dysphagia or a contributing factor, or whether it plays any role at all. Alternative causes of dysphagia must be considered, including neurologic disease (stroke, Parkinson disease, and amyotrophic lateral sclerosis) and/or mechanical obstruction due to head/neck cancer, mediastinal masses, the Zenker diverticulum, esophagus webs, and stricture or cancer of the esophagus.13 The mechanisms involved in the origin of dysphagia include mechanical compression causing esophageal obstruction, an inflammatory
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
FIGURE 1. Preoperative axial CT image of the cervical spine shows the large ostephyte.
FIGURE 2. Preoperative three-dimensional CT image of the cervical spine.
FIGURE 3. Intraoperative image of the oropharynx and the cervical osteophyte.
FIGURE 4. Macroscopic image of the tumor.
FIGURE 5. Postoperative cervical x-ray film shows no bony spur at the anterior cervical spine.
reaction that causes pharyngitis, fibrosis and adhesions with fixation of the esophagus at the level of the cricoid cartilage, cricopharyngeal spasm triggered by pressure on the esophagus, as well as impaired motility of epiglottis.14,15 Dysphonia may arise from compression, glottis inflammation and edema, as well as vocal fold immobility as a result of an ulceration of the cricoid produced by laryngeal movement over a large
Large Cervical Osteophyte
osteophyte.16 It has also been reported that dysphonia may occasionally be caused by a large extraluminar osteophyte, which directly compresses, distorts, and, finally, obstructs the airway.16,17 In this case, our patient has both dysphagia and dyspnea. Diagnostic investigation should include laryngoscopic examination because the lesion may not be large enough to be seen with oropharyngeal examination. A lateral plain radiograph can be helpful in the evaluation of the cervical spine. Computed tomography or magnetic resonance imaging with sagittal reconstruction is advised to enable the location of anterior bony lesions in relation to the surrounding soft tissues, large vessels, and nerve sheets. Barium swallow test can also be used to exclude neoplasia as well as reveal compression and obstruction of the esophagus.17,18 We used CT and lateral cervical xray in this case. Treatment is conservative or surgical. Conservative treatment is antibiotics, anti-inflammatory agents, steroids, and muscle relaxants. There are many surgical techniques, including anterolateral, posterolateral, and transoral approaches. The patient was treated surgically through removal of the hyperostosis via the transoral approach. Surgical removal of the osteophyte was performed, and the patient was relieved from symptoms. He was examined 2 months later, and no recurrence was noted.
REFERENCES 1. Papadopoulos SM, Chen JC, Feldenzer JA, et al. Anterior cervical osteophytes as a cause of progressive dysphagia. Acta Neurochir (Wien) 1989;101:63–65 2. Mosher HP. Exostosis of the cervical vertebrae as a cause of difficulty in swallowing. Laryngoscope 1926;36:181–182 3. Hirano H, Suzuki H, Sakakibara T, et al. Dysphagia due to hypertrophic cervical osteophytes. Clin Orthop Relat Res 1982;167:168–172 4. Carlson MJ, Stauffer RN, Payne WS. Anklyosing vertebral hyperostosis causing dysphagia. Arch Surg 1974;109:567–570 5. Braun J, Sieper J. Ankylosing spondylitis. Lancet 2007;369:1379–1390 6. Luken MG, Patel DV, Ellman MH. Symptomatic spinal stenosis associated with ankylosing spondylitis. Neurosurgery 1982;11:703–705 7. Gamache FW, Voorhies RM. Hypertrophic cervical osteophytes causing dysphagia. J Neurosurg 1980;53:338–344 8. Kiss C, Szilagyi M, Paksy A, et al. Risk factors for diffuse idiopathic skeletal hyperostosis: a case control study. Rheumatology 2002;41:27–30 9. Baraliakos X, Listing J, Buschmann J, et al. A comparison of new bone formation in patients with ankylosing spondylitis and patients with diffuse idiopathic skeletal hyperostosis: a retrospective cohort study over six years. Arthritis Rheum 2012;64:1127–1133 10. Cesur M, Alıcı HA, Erdem AF. An unusual cause of difficult intubation in a patient with a large cervical anterior osteophyte: a case report. Acta Anaesthesiol Scand 2005;49:264–266 11. Ranasınghe DN, Calder I. Large cervical osteophyte—another cause of difficult flexible fibreoptic intubation. Anaesthesia 1994;49:512–514 12. Lin HW, Quesnel AM, Holman AS, et al. Hypertrophic anterior cervical osteophytes causing dysphagia and airway obstruction. Ann Otol Rhinol Laryngol 2009;118:703–707 13. McCafferty RR, Harrison MJ, Tamas LB, et al. Ossification of the anterior longitudinal ligament and Forestier’s disease: an analysis of seven cases. J Neurosurg 1995;83:13–17 14. Faruqi S, Thirumanan M, Blaxill P. An osseous cause of dysphagia. Med J Aust 2008;188:671 15. Uzunca K, Birtane M, Tezel A. Dysphagia induced by a cervical osteophyte: a case report of cervical spondylosis. Chin Med J (Engl) 2004;117:478–480 16. Giger R, Dulguerov P, Payer M. Anterior osteophytes causing dysphagia and dyspnea: an uncommon entity revisited. Dysphagia 2006;21:259–263 17. Miauri F, Stella L, Buonamassa S. Dysphagia and dyspnea due to an anterior cervical osteophyte. Arch Orthop Trauma Surg 2002;122:245–247 18. Bruna E, Alessandrini M, De Angelis E. Giant cervical hyperostosis of the prevertebral space: presentation of two cases and review of the literature. Acta Otorhinolaryngol Ital 1996;16532–536
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
1403
Dysphagia and Airway Obstruction Due to Large Cervical Osteophyte in a Patient With Ankylosing Spondylitis Yasin Kürşad Varsak, MD, Mehmet Akif Eryilmaz, MD, and Hamdi Arbağ, MD Abstract: Anterior cervical osteophytes are excessive bony formation of cervical vertebra bodies. They are common but rarely symptomatic lesions mostly seen in geriatric population. Large anterior cervical osteophytes may cause symptoms such as dysphagia, dyspnea, dysphonia, and odynophagia. They have been attributed to multiple etiologies including diffuse idiopathic skeletal hyperostosis, following trauma, cervical spondylitis, and infectious spondylitis. However, symptomatic large anterior cervical osteophyte with ankylosing spondylitis is extremely rare. Surgical excision is the main treatment for symptomatic cases. We report a case of a 53-year-old man with airway obstruction and dysphagia due to large cervical osteophyte who has a history of ankylosing spondylitis, and we also addressed the etiological factors and management of large symptomatic cervical osteophytes. Key Words: Ankylosing spondylitis, anterior cervical osteophytes, dysphagia (J Craniofac Surg 2014;25: 1402–1403)
O
steophytes are bony outgrowths found at the articular margins in many musculoskeletal disorders. Cervical vertebras are one of the most commonly effected bones. Anterior cervical hypertrophic spurs of the anterior cervical spine may occur in 20% to 30% of the population.1 Although cervical osteophytes are asymptomatic, they may lead to symptoms such as dysphagia, cough, dyspnea, and dysphonia. If symptomatic, dysphagia appears to be the most common presentation, caused by mechanical obstruction of the pharyngoesophageal segment by anterior cervical hyperostosis. Mosher,2 in 1926, first described 2 patients with dysphagia caused by large anterior cervical osteophytes. Large anterior cervical osteophytes are associated with senile degenerative skeletal disease, posttraumatic osteophytogenesis, idiopathic skeletal hyperostosis (DISH), cervical spondylitis, and infectious spondylitis, but ankylosing spondylitis (AS) has been very rarely mentioned as an etiological factor in the literature.3,4
PATIENT A 53-year-old man was referred to our clinic for airway obstruction, which caused failed intubation for appointed hip surgery. The patient was complaining of a slowly progressive dysphagia and, recently, the onset of dyspnea. The dysphagia has been From the Department of Otorhinolaryngology, Head and Neck Surgery, Meram Medical Faculty, Necmettin Erbakan University, Meram, Konya, Turkey. Received January 25, 2014. Accepted for publication February 16, 2014. Address correspondence and reprint requests to Yasin Kursad Varsak, MD, Department of Otorhinolaryngology, Head and Neck Surgery, Meram Medical Faculty, Necmettin Erbakan University, Meram, 42080 Konya, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000933
1402
present for 6 years, and the dyspnea has been present for 3 weeks. The patient has a medical history of AS for 17 years. There was a noticeable kyphosis on physical examination. Oropharyngeal examination revealed a 2- to 3-cm submucosal lesion with a smooth surface and firm margin, which narrows the oropharyngeal inlet. The lesion was hard on palpation. Indirect laryngoscopy revealed a smooth bulge in the posterior wall of the pharynx without mucosal lesions. The bulge was rigid and nonpulsatile. The patient was examined through spiral computed tomography (CT) of the cervical spine. It demonstrated a large hypertrophic anterior cervical osteophyte of C2-C3 vertebra (Fig. 1). Three-dimensional reconstruction of CT image was obtained as well (Fig. 2). First, the airway was secured under local anesthesia via tracheotomy, then the patient underwent transoral cervical osteophytectomy surgery under general anesthesia (Fig. 3). Pathologic findings macroscopically revealed a 4.5 4 1.5-cm gray bone colored solid bony tissue (Fig. 4) and microscopically revealed a trabecular mature bone tissue with marginal cartilaginous tissue. The patient was decannulated at postoperative day 8. Postoperative control lateral cervical x-ray film has shown no osteophytes at the posterior pharyngeal wall (Fig. 5). There was no complaint of dysphagia and dyspnea 12 months after the surgery.
DISCUSSION Large osteophyte of the cervical spine presenting with dysphagia is a rare condition that can be seen more in elderly patients. In this case, we report a patient with anterior cervical osteophyte that is secondary to AS. Ankylosing spondylitis is a chronic inflammatory seronegative rheumatic arthritis.5 Typically, this process first affects the sacroiliac joints, later progressing to involve the hips and spine.6 Anterior cervical osteophytes are associated with senile degenerative skeletal disease, posttraumatic osteophytogenesis, DISH, cervical spondylitis, and infectious spondylitis. Most patients with dysphagia based on cervical hyperostosis are attributed to DISH.7 The etiology of this condition is unknown, but researchers have found strong correlations with metabolic disorders, especially high body mass index and insulin-independent diabetes mellitus.8 Baraliakos et al9 has shown that the rates of new bone formation in AS and DISH are largely similar. Whereas syndesmophytes are more frequent in AS, patients with DISH have more degenerative bone spurs.9 However, there are only a few cases of symptomatic large anterior cervical osteophytes with underlying AS in the literature.10–12 Osteophytes of the anterior cervical spine are common in elderly patients and are usually asymptomatic. They may impinge on the pharynx or esophagus and can cause dysphagia, dyspnea, or stridor. When a patient known to have a cervical vertebral bone spur complains of dysphagia, it must be determined whether the protuberance is the singular cause of the dysphagia or a contributing factor, or whether it plays any role at all. Alternative causes of dysphagia must be considered, including neurologic disease (stroke, Parkinson disease, and amyotrophic lateral sclerosis) and/or mechanical obstruction due to head/neck cancer, mediastinal masses, the Zenker diverticulum, esophagus webs, and stricture or cancer of the esophagus.13 The mechanisms involved in the origin of dysphagia include mechanical compression causing esophageal obstruction, an inflammatory
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 4, July 2014
FIGURE 1. Preoperative axial CT image of the cervical spine shows the large ostephyte.
FIGURE 2. Preoperative three-dimensional CT image of the cervical spine.
FIGURE 3. Intraoperative image of the oropharynx and the cervical osteophyte.
FIGURE 4. Macroscopic image of the tumor.
FIGURE 5. Postoperative cervical x-ray film shows no bony spur at the anterior cervical spine.
reaction that causes pharyngitis, fibrosis and adhesions with fixation of the esophagus at the level of the cricoid cartilage, cricopharyngeal spasm triggered by pressure on the esophagus, as well as impaired motility of epiglottis.14,15 Dysphonia may arise from compression, glottis inflammation and edema, as well as vocal fold immobility as a result of an ulceration of the cricoid produced by laryngeal movement over a large
Large Cervical Osteophyte
osteophyte.16 It has also been reported that dysphonia may occasionally be caused by a large extraluminar osteophyte, which directly compresses, distorts, and, finally, obstructs the airway.16,17 In this case, our patient has both dysphagia and dyspnea. Diagnostic investigation should include laryngoscopic examination because the lesion may not be large enough to be seen with oropharyngeal examination. A lateral plain radiograph can be helpful in the evaluation of the cervical spine. Computed tomography or magnetic resonance imaging with sagittal reconstruction is advised to enable the location of anterior bony lesions in relation to the surrounding soft tissues, large vessels, and nerve sheets. Barium swallow test can also be used to exclude neoplasia as well as reveal compression and obstruction of the esophagus.17,18 We used CT and lateral cervical xray in this case. Treatment is conservative or surgical. Conservative treatment is antibiotics, anti-inflammatory agents, steroids, and muscle relaxants. There are many surgical techniques, including anterolateral, posterolateral, and transoral approaches. The patient was treated surgically through removal of the hyperostosis via the transoral approach. Surgical removal of the osteophyte was performed, and the patient was relieved from symptoms. He was examined 2 months later, and no recurrence was noted.
REFERENCES 1. Papadopoulos SM, Chen JC, Feldenzer JA, et al. Anterior cervical osteophytes as a cause of progressive dysphagia. Acta Neurochir (Wien) 1989;101:63–65 2. Mosher HP. Exostosis of the cervical vertebrae as a cause of difficulty in swallowing. Laryngoscope 1926;36:181–182 3. Hirano H, Suzuki H, Sakakibara T, et al. Dysphagia due to hypertrophic cervical osteophytes. Clin Orthop Relat Res 1982;167:168–172 4. Carlson MJ, Stauffer RN, Payne WS. Anklyosing vertebral hyperostosis causing dysphagia. Arch Surg 1974;109:567–570 5. Braun J, Sieper J. Ankylosing spondylitis. Lancet 2007;369:1379–1390 6. Luken MG, Patel DV, Ellman MH. Symptomatic spinal stenosis associated with ankylosing spondylitis. Neurosurgery 1982;11:703–705 7. Gamache FW, Voorhies RM. Hypertrophic cervical osteophytes causing dysphagia. J Neurosurg 1980;53:338–344 8. Kiss C, Szilagyi M, Paksy A, et al. Risk factors for diffuse idiopathic skeletal hyperostosis: a case control study. Rheumatology 2002;41:27–30 9. Baraliakos X, Listing J, Buschmann J, et al. A comparison of new bone formation in patients with ankylosing spondylitis and patients with diffuse idiopathic skeletal hyperostosis: a retrospective cohort study over six years. Arthritis Rheum 2012;64:1127–1133 10. Cesur M, Alıcı HA, Erdem AF. An unusual cause of difficult intubation in a patient with a large cervical anterior osteophyte: a case report. Acta Anaesthesiol Scand 2005;49:264–266 11. Ranasınghe DN, Calder I. Large cervical osteophyte—another cause of difficult flexible fibreoptic intubation. Anaesthesia 1994;49:512–514 12. Lin HW, Quesnel AM, Holman AS, et al. Hypertrophic anterior cervical osteophytes causing dysphagia and airway obstruction. Ann Otol Rhinol Laryngol 2009;118:703–707 13. McCafferty RR, Harrison MJ, Tamas LB, et al. Ossification of the anterior longitudinal ligament and Forestier’s disease: an analysis of seven cases. J Neurosurg 1995;83:13–17 14. Faruqi S, Thirumanan M, Blaxill P. An osseous cause of dysphagia. Med J Aust 2008;188:671 15. Uzunca K, Birtane M, Tezel A. Dysphagia induced by a cervical osteophyte: a case report of cervical spondylosis. Chin Med J (Engl) 2004;117:478–480 16. Giger R, Dulguerov P, Payer M. Anterior osteophytes causing dysphagia and dyspnea: an uncommon entity revisited. Dysphagia 2006;21:259–263 17. Miauri F, Stella L, Buonamassa S. Dysphagia and dyspnea due to an anterior cervical osteophyte. Arch Orthop Trauma Surg 2002;122:245–247 18. Bruna E, Alessandrini M, De Angelis E. Giant cervical hyperostosis of the prevertebral space: presentation of two cases and review of the literature. Acta Otorhinolaryngol Ital 1996;16532–536
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
1403
