DOI: 10.3171/2014.6.SPINE13908 ©AANS, 2014
Cervical osteophyte resulting in compression of the jugular foramen Case report Andrew Q. Le, B.S., Brian P. Walcott, M.D., Navid Redjal, M.D., and Jean-Valery Coumans, M.D. Department of Neurosurgery, Massachusetts General Hospital, and Harvard Medical School, Boston, Massachusetts Jugular foramen syndrome is a condition characterized by unilateral paresis of cranial nerves IX, X, and XI in the setting of extrinsic compression. Here, the authors describe the case of a giant cervical osteophyte resulting in compression of the jugular foramen. A 74-year-old man who presented with progressive dysphagia and dysarthria was found to have right-sided tongue deviation, left palatal droop, and hypophonia. His dysphagia had progressed to the point that he had lost 25 kg over a 4-month period, necessitating a gastrostomy to maintain adequate nutrition. He underwent extensive workup for his dysphagia with several normal radiographic studies. Ultimately, CT scanning and postcontrast MRI revealed a posterior osteophyte arising from the C1–2 joint space and projecting into the right jugular foramen. This resulted in a jugular foramen syndrome in addition to delayed filling of the patient’s right internal jugular vein distal to the osteophyte. Although rare, a posterior cervical osteophyte should be considered in cases of jugular foramen syndrome. (http://thejns.org/doi/abs/10.3171/2014.6.SPINE13908)
Key Words • spondylosis • cervical spine • jugular foramen • osteophyte
O
steophytes are defined as abnormal bony growths or bone spurs that project from joints. They commonly occur in the vertebral bodies of the cervical spine and are radiographically found in approximately 20%–30% of the elderly population.4 While osteophyte formation in other degenerating joints of the body typically cause only local pain and discomfort, cervical osteophytes can result in a wide spectrum of symptoms because of their unique location.6,7,14,22 Here, we describe a unique sequela of a cervical osteophyte: an osteophyte narrowing the right internal jugular vein and jugular foramen, causing progressive dysphagia and dysarthria, characteristic of jugular foramen syndrome.
Case Report
A 74-year-old-man with a history of chronic obstructive pulmonary disease and hyperlipidemia presented with slowly progressive dysphagia that had begun with solid food difficulties and left him unable to swallow. Over a 4-month course, he had lost 25 kg. He also suffered from dysarthria. His dysphagia had progressed to the point that he could no longer eat, necessitating enteral feeding via gastrostomy to support his nutritional needs. Abbreviation used in this paper: CN = cranial nerve.
J Neurosurg: Spine / July 11, 2014
On initial examination in the clinic, he exhibited right-sided tongue deviation, left palatal droop, and profound hypophonia and dysarthria. Further workup included a CT scan of the neck and an MR image of the skull base. Initial interpretation of these images was unremarkable for a causative pathological process. A bronchoscopy was performed and revealed no evidence of endobronchial masses. Direct laryngoscopy demonstrated paralysis and edema of the right vocal cord. Biopsies of the epiglottis and vallecula failed to show a pathological process. An esophagogram was significant for reflux, although pharmacological treatment of this disorder did not improve his symptoms. A follow-up CT scan of the neck revealed no laryngeal mass but findings consistent with a paralyzed right vocal cord. There was narrowing of the right internal jugular vein just below the level of the skull base, with the more distal portion of the internal jugular appearing widely patent. A reread of the previous brain MRI study revealed an enhancement in the region of the pars nervosa of the jugular foramen and effacement of the right internal carotid artery at this level, suggesting a mass lesion. A second MRI study was obtained and showed a posterior osteophyte caused by a degenerated atlantooccipital joint at C1–2 adjacent to a small area of enhancement anteromedial to the right internal jugular vein, with no features 1
A. Q. Le et al. surgical intervention at this time, he continues to follow up at regular intervals to review treatment options.
Discussion
Fig. 1. Axial contrast-enhanced T1-weighted MR image showing a small area of enhancement anteromedial to the right internal jugular vein, consistent with degenerative inflammation (white arrow).
suggesting a tumor (Fig. 1). An indium-111–labeled pentetreotide scan was obtained, as was a SPECT scan, neither of which revealed any area of abnormal signal. Further workup included a CT venogram of the head and neck, demonstrating narrowing of the right internal jugular vein as well as severe stenosis of the pars nervosa and pars vascularis (Figs. 2 and 3), which were congruent with the patient’s cranial nerve (CN) deficits. Currently, the patient is being managed expectantly. While resection of the mass is technically feasible via a standard far lateral approach,16 the patient’s medical comorbidities place him at significant risk for complications. While he defers
The sequela of cervical osteophytes is well documented and primarily related to their anatomical location. Osteophytes of the atlas and axis affect structures of the basiocciput, those of C2–3 involve the posterior pharynx, and bony growths of C4–7 diminish the retropharyngeal space and compress the larynx and esophagus.15 With the esophagus resting on the anterior border of C4–7, anterior cervical osteophytes have repeatedly been found to mechanically disrupt normal esophageal or laryngeal function, resulting in dysphagia.3,8,10,19 While anterior osteophytes are known to cause dysphagia, osteophytes of the posterior vertebral body causing dysphagia are much more rare. In the featured case, dysphagia, dysarthria, tongue deviation, and palatal droop are believed to be consistent with a jugular foramen syndrome. The jugular foramen is divided into the smaller anteromedial pars nervosa, which contains CN IX and the venous return from the inferior petrosal sinus, and the larger posterolateral pars vascularis, which contains CN X, CN XI, and Arnold’s nerve, a branch of CN X. Jugular foramen syndrome is defined as the unilateral involvement of CNs IX, X, and XI as a result of narrowing of the jugular foramen.18,20 Jugular foramen syndrome is commonly seen in response to skull base lesions, such as meningiomas and schwannomas;12 however, there are case reports of metastatic tumors, trauma, infection, cholesteatomas, aneurysms, and even varicella zoster infection leading to this condition.1,2,9,11,13,17 Development of jugular foramen syndrome in association with a posterior cervical osteophyte has not been reported in the literature. In the present case, localization of the patient’s pathology corresponded to his neurological findings. His
Fig. 2. Computed tomography venogram with 3D reconstruction of the neck demonstrating the proximity of the right jugular vein (blue arrow) and the cervical osteophyte (black arrow). The cervical osteophyte encroaches on the right jugular foramen.
2
J Neurosurg: Spine / July 11, 2014
Giant osteophyte compressing the jugular foramen
Fig. 3. Axial CT venogram (A), coronal CT (B), and parasagittal CT (C) demonstrating the relationship of the cervical osteophyte with the pars nervosa (red arrow) and, to a lesser extent, the pars vascularis of the jugular foramen.
progressive dysphagia, palatal droop, tongue deviation, and hypophonia in combination with the venous obstruction on MRI distinctively suggested a process in the jugular foramen. Isolated lesions of CNs IX–XI are rare, and upper motor neuron damage is not typical due to the number of corticobulbar projections to the brainstem.5,21 Furthermore, a lesion in the brainstem would typically manifest with more symptoms given the myriad structures in close proximity. The first line of treatment for cervical osteophytes is typically conservative and involves nonsteroidal antiinflammatory drugs to minimize the inflammatory reaction around the osteophyte. Surgical treatment should be considered if conservative management fails. Improvement following neural decompression is uncertain and very likely depends on the extent and duration of neural compression preoperatively. Disclosure The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper. Author contributions to the study and manuscript preparation include the following. Conception and design: Coumans. Acquisition of data: Coumans. Analysis and interpretation of data: all authors. Drafting the article: all authors. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Walcott.
3. Aronowitz P, Cobarrubias F: Images in clinical medicine. Anterior cervical osteophytes causing airway compromise. N Engl J Med 349:2540, 2003 4. Bone RC, Nahum AM, Harris AS: Evaluation and correction of dysphagia-producing cervical osteophytosis. Laryngoscope 84:2045–2050, 1974 5. Burt AM: Synopsis of the cranial nerves, in Textbook of Neuroanatomy. Philadelphia: WB Saunders, 1993, pp 402–430 6. Cammisa M, De Serio A, Guglielmi G: Diffuse idiopathic skeletal hyperostosis. Eur J Radiol 27 Suppl 1:S7–S11, 1998 7. Chatziavramidis A, Dimitra A: Dysphagia due to anterior cervical osteophytes complicated with hypopharynx abscess. BMJ Case Rep 2011:bcr1120103551, 2011 8. Di Vito J Jr: Cervical osteophytic dysphagia: single and combined mechanisms. Dysphagia 13:58–61, 1998 9. Erol FS, Kaplan M, Kavakli A, Ozveren MF: Jugular foramen syndrome caused by choleastatoma. Clin Neurol Neurosurg 107:342–346, 2005 10. Fuerderer S, Eysel-Gosepath K, Schröder U, Delank KS, Eysel P: Retro-pharyngeal obstruction in association with osteophytes of the cervical spine. J Bone Joint Surg Br 86:837– 840, 2004 11. Hayashi T, Murayama S, Sakurai M, Kanazawa I: Jugular foramen syndrome caused by varicella zoster virus infection in a patient with ipsilateral hypoplasia of the jugular foramen. J Neurol Sci 172:70–72, 2000 12. Jackson CG, Cueva RA, Thedinger BA, Glasscock ME III: Cranial nerve preservation in lesions of the jugular fossa. Otolaryngol Head Neck Surg 105:687–693, 1991 13. Jo Y, Chung CW, Lee JS, Park HJ: Vermet syndrome by varicella-zoster virus. Ann Rehabil Med 37:449–452, 2013 14. Kapetanakis S, Vasileiadis I, Papanas N, Goulimari R, Maltezos E: Can a giant cervical osteophyte cause dysphagia and airway obstruction? A case report. Wien Klin Wochenschr 123:291– 293, 2011 15. Klaassen Z, Tubbs RS, Apaydin N, Hage R, Jordan R, Loukas M: Vertebral spinal osteophytes. Anat Sci Int 86:1–9, 2011 16. Rhoton AL Jr: The far-lateral approach and its transcondylar, supracondylar, and paracondylar extensions. Neurosurgery 47 (3 Suppl):S195–S209, 2000 17. Robbins KT, Fenton RS: Jugular foramen syndrome. J Otolaryngol 9:505–516, 1980 18. Saunders PW: Unilateral paralysis of ninth, tenth, and eleventh cranial nerves. Proc R Soc Med 4 (Neurol Sect):51, 1911 19. Stancampiano FF, Zavaleta EG, Astor F: Anterior cervical osteophytes: a rare cause of dysphagia and upper airway obstruction in older patients. J Am Geriatr Soc 50:1910–1911, 2002 20. Svien HJ, Baker HL, Rivers MH: Jugular foramen syndrome and allied syndromes. Neurology 13:797–809, 1963 21. Wilson-Pauwels L, Akesson EJ, Stewart PA: Glossopharyngeal nerve, in Cranial Nerves: Anatomy and Clinical Comments. Toronto: BC Decker, 1988, pp 113–124 22. Witiw CD, Fallah A, Muller PJ, Ginsberg HJ: Surgical treatment of spontaneous intracranial hypotension secondary to degenerative cervical spine pathology: a case report and literature review. Eur Spine J 21 (Suppl 4):S422–S427, 2012
References 1. Aghaghazvini L, Sedighi N, Karami P, Yeganeh O: Skull base aneurysmal bone cyst presented with foramen jugular syndrome and multi-osseous involvement. Iran J Radiol 9:157–160, 2012 2. Amano M, Ishikawa E, Kujiraoka Y, Watanabe S, Ashizawa K, Oguni E, et al: Vernet’s syndrome caused by large mycotic aneurysm of the extracranial internal carotid artery after acute otitis media—case report. Neurol Med Chir (Tokyo) 50:45– 48, 2010
J Neurosurg: Spine / July 11, 2014
Manuscript submitted October 11, 2013. Accepted June 10, 2014. Please include this information when citing this paper: published online July 11, 2014; DOI: 10.3171/2014.6.SPINE13908. Address correspondence to: Brian P. Walcott, M.D., Department of Neurological Surgery, Massachusetts General Hospital, 55 Fruit St., White Bldg., Rm. 502, Boston, MA 02114. email: walcott. [email protected].
3
Cervical osteophyte resulting in compression of the jugular foramen Case report Andrew Q. Le, B.S., Brian P. Walcott, M.D., Navid Redjal, M.D., and Jean-Valery Coumans, M.D. Department of Neurosurgery, Massachusetts General Hospital, and Harvard Medical School, Boston, Massachusetts Jugular foramen syndrome is a condition characterized by unilateral paresis of cranial nerves IX, X, and XI in the setting of extrinsic compression. Here, the authors describe the case of a giant cervical osteophyte resulting in compression of the jugular foramen. A 74-year-old man who presented with progressive dysphagia and dysarthria was found to have right-sided tongue deviation, left palatal droop, and hypophonia. His dysphagia had progressed to the point that he had lost 25 kg over a 4-month period, necessitating a gastrostomy to maintain adequate nutrition. He underwent extensive workup for his dysphagia with several normal radiographic studies. Ultimately, CT scanning and postcontrast MRI revealed a posterior osteophyte arising from the C1–2 joint space and projecting into the right jugular foramen. This resulted in a jugular foramen syndrome in addition to delayed filling of the patient’s right internal jugular vein distal to the osteophyte. Although rare, a posterior cervical osteophyte should be considered in cases of jugular foramen syndrome. (http://thejns.org/doi/abs/10.3171/2014.6.SPINE13908)
Key Words • spondylosis • cervical spine • jugular foramen • osteophyte
O
steophytes are defined as abnormal bony growths or bone spurs that project from joints. They commonly occur in the vertebral bodies of the cervical spine and are radiographically found in approximately 20%–30% of the elderly population.4 While osteophyte formation in other degenerating joints of the body typically cause only local pain and discomfort, cervical osteophytes can result in a wide spectrum of symptoms because of their unique location.6,7,14,22 Here, we describe a unique sequela of a cervical osteophyte: an osteophyte narrowing the right internal jugular vein and jugular foramen, causing progressive dysphagia and dysarthria, characteristic of jugular foramen syndrome.
Case Report
A 74-year-old-man with a history of chronic obstructive pulmonary disease and hyperlipidemia presented with slowly progressive dysphagia that had begun with solid food difficulties and left him unable to swallow. Over a 4-month course, he had lost 25 kg. He also suffered from dysarthria. His dysphagia had progressed to the point that he could no longer eat, necessitating enteral feeding via gastrostomy to support his nutritional needs. Abbreviation used in this paper: CN = cranial nerve.
J Neurosurg: Spine / July 11, 2014
On initial examination in the clinic, he exhibited right-sided tongue deviation, left palatal droop, and profound hypophonia and dysarthria. Further workup included a CT scan of the neck and an MR image of the skull base. Initial interpretation of these images was unremarkable for a causative pathological process. A bronchoscopy was performed and revealed no evidence of endobronchial masses. Direct laryngoscopy demonstrated paralysis and edema of the right vocal cord. Biopsies of the epiglottis and vallecula failed to show a pathological process. An esophagogram was significant for reflux, although pharmacological treatment of this disorder did not improve his symptoms. A follow-up CT scan of the neck revealed no laryngeal mass but findings consistent with a paralyzed right vocal cord. There was narrowing of the right internal jugular vein just below the level of the skull base, with the more distal portion of the internal jugular appearing widely patent. A reread of the previous brain MRI study revealed an enhancement in the region of the pars nervosa of the jugular foramen and effacement of the right internal carotid artery at this level, suggesting a mass lesion. A second MRI study was obtained and showed a posterior osteophyte caused by a degenerated atlantooccipital joint at C1–2 adjacent to a small area of enhancement anteromedial to the right internal jugular vein, with no features 1
A. Q. Le et al. surgical intervention at this time, he continues to follow up at regular intervals to review treatment options.
Discussion
Fig. 1. Axial contrast-enhanced T1-weighted MR image showing a small area of enhancement anteromedial to the right internal jugular vein, consistent with degenerative inflammation (white arrow).
suggesting a tumor (Fig. 1). An indium-111–labeled pentetreotide scan was obtained, as was a SPECT scan, neither of which revealed any area of abnormal signal. Further workup included a CT venogram of the head and neck, demonstrating narrowing of the right internal jugular vein as well as severe stenosis of the pars nervosa and pars vascularis (Figs. 2 and 3), which were congruent with the patient’s cranial nerve (CN) deficits. Currently, the patient is being managed expectantly. While resection of the mass is technically feasible via a standard far lateral approach,16 the patient’s medical comorbidities place him at significant risk for complications. While he defers
The sequela of cervical osteophytes is well documented and primarily related to their anatomical location. Osteophytes of the atlas and axis affect structures of the basiocciput, those of C2–3 involve the posterior pharynx, and bony growths of C4–7 diminish the retropharyngeal space and compress the larynx and esophagus.15 With the esophagus resting on the anterior border of C4–7, anterior cervical osteophytes have repeatedly been found to mechanically disrupt normal esophageal or laryngeal function, resulting in dysphagia.3,8,10,19 While anterior osteophytes are known to cause dysphagia, osteophytes of the posterior vertebral body causing dysphagia are much more rare. In the featured case, dysphagia, dysarthria, tongue deviation, and palatal droop are believed to be consistent with a jugular foramen syndrome. The jugular foramen is divided into the smaller anteromedial pars nervosa, which contains CN IX and the venous return from the inferior petrosal sinus, and the larger posterolateral pars vascularis, which contains CN X, CN XI, and Arnold’s nerve, a branch of CN X. Jugular foramen syndrome is defined as the unilateral involvement of CNs IX, X, and XI as a result of narrowing of the jugular foramen.18,20 Jugular foramen syndrome is commonly seen in response to skull base lesions, such as meningiomas and schwannomas;12 however, there are case reports of metastatic tumors, trauma, infection, cholesteatomas, aneurysms, and even varicella zoster infection leading to this condition.1,2,9,11,13,17 Development of jugular foramen syndrome in association with a posterior cervical osteophyte has not been reported in the literature. In the present case, localization of the patient’s pathology corresponded to his neurological findings. His
Fig. 2. Computed tomography venogram with 3D reconstruction of the neck demonstrating the proximity of the right jugular vein (blue arrow) and the cervical osteophyte (black arrow). The cervical osteophyte encroaches on the right jugular foramen.
2
J Neurosurg: Spine / July 11, 2014
Giant osteophyte compressing the jugular foramen
Fig. 3. Axial CT venogram (A), coronal CT (B), and parasagittal CT (C) demonstrating the relationship of the cervical osteophyte with the pars nervosa (red arrow) and, to a lesser extent, the pars vascularis of the jugular foramen.
progressive dysphagia, palatal droop, tongue deviation, and hypophonia in combination with the venous obstruction on MRI distinctively suggested a process in the jugular foramen. Isolated lesions of CNs IX–XI are rare, and upper motor neuron damage is not typical due to the number of corticobulbar projections to the brainstem.5,21 Furthermore, a lesion in the brainstem would typically manifest with more symptoms given the myriad structures in close proximity. The first line of treatment for cervical osteophytes is typically conservative and involves nonsteroidal antiinflammatory drugs to minimize the inflammatory reaction around the osteophyte. Surgical treatment should be considered if conservative management fails. Improvement following neural decompression is uncertain and very likely depends on the extent and duration of neural compression preoperatively. Disclosure The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper. Author contributions to the study and manuscript preparation include the following. Conception and design: Coumans. Acquisition of data: Coumans. Analysis and interpretation of data: all authors. Drafting the article: all authors. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Walcott.
3. Aronowitz P, Cobarrubias F: Images in clinical medicine. Anterior cervical osteophytes causing airway compromise. N Engl J Med 349:2540, 2003 4. Bone RC, Nahum AM, Harris AS: Evaluation and correction of dysphagia-producing cervical osteophytosis. Laryngoscope 84:2045–2050, 1974 5. Burt AM: Synopsis of the cranial nerves, in Textbook of Neuroanatomy. Philadelphia: WB Saunders, 1993, pp 402–430 6. Cammisa M, De Serio A, Guglielmi G: Diffuse idiopathic skeletal hyperostosis. Eur J Radiol 27 Suppl 1:S7–S11, 1998 7. Chatziavramidis A, Dimitra A: Dysphagia due to anterior cervical osteophytes complicated with hypopharynx abscess. BMJ Case Rep 2011:bcr1120103551, 2011 8. Di Vito J Jr: Cervical osteophytic dysphagia: single and combined mechanisms. Dysphagia 13:58–61, 1998 9. Erol FS, Kaplan M, Kavakli A, Ozveren MF: Jugular foramen syndrome caused by choleastatoma. Clin Neurol Neurosurg 107:342–346, 2005 10. Fuerderer S, Eysel-Gosepath K, Schröder U, Delank KS, Eysel P: Retro-pharyngeal obstruction in association with osteophytes of the cervical spine. J Bone Joint Surg Br 86:837– 840, 2004 11. Hayashi T, Murayama S, Sakurai M, Kanazawa I: Jugular foramen syndrome caused by varicella zoster virus infection in a patient with ipsilateral hypoplasia of the jugular foramen. J Neurol Sci 172:70–72, 2000 12. Jackson CG, Cueva RA, Thedinger BA, Glasscock ME III: Cranial nerve preservation in lesions of the jugular fossa. Otolaryngol Head Neck Surg 105:687–693, 1991 13. Jo Y, Chung CW, Lee JS, Park HJ: Vermet syndrome by varicella-zoster virus. Ann Rehabil Med 37:449–452, 2013 14. Kapetanakis S, Vasileiadis I, Papanas N, Goulimari R, Maltezos E: Can a giant cervical osteophyte cause dysphagia and airway obstruction? A case report. Wien Klin Wochenschr 123:291– 293, 2011 15. Klaassen Z, Tubbs RS, Apaydin N, Hage R, Jordan R, Loukas M: Vertebral spinal osteophytes. Anat Sci Int 86:1–9, 2011 16. Rhoton AL Jr: The far-lateral approach and its transcondylar, supracondylar, and paracondylar extensions. Neurosurgery 47 (3 Suppl):S195–S209, 2000 17. Robbins KT, Fenton RS: Jugular foramen syndrome. J Otolaryngol 9:505–516, 1980 18. Saunders PW: Unilateral paralysis of ninth, tenth, and eleventh cranial nerves. Proc R Soc Med 4 (Neurol Sect):51, 1911 19. Stancampiano FF, Zavaleta EG, Astor F: Anterior cervical osteophytes: a rare cause of dysphagia and upper airway obstruction in older patients. J Am Geriatr Soc 50:1910–1911, 2002 20. Svien HJ, Baker HL, Rivers MH: Jugular foramen syndrome and allied syndromes. Neurology 13:797–809, 1963 21. Wilson-Pauwels L, Akesson EJ, Stewart PA: Glossopharyngeal nerve, in Cranial Nerves: Anatomy and Clinical Comments. Toronto: BC Decker, 1988, pp 113–124 22. Witiw CD, Fallah A, Muller PJ, Ginsberg HJ: Surgical treatment of spontaneous intracranial hypotension secondary to degenerative cervical spine pathology: a case report and literature review. Eur Spine J 21 (Suppl 4):S422–S427, 2012
References 1. Aghaghazvini L, Sedighi N, Karami P, Yeganeh O: Skull base aneurysmal bone cyst presented with foramen jugular syndrome and multi-osseous involvement. Iran J Radiol 9:157–160, 2012 2. Amano M, Ishikawa E, Kujiraoka Y, Watanabe S, Ashizawa K, Oguni E, et al: Vernet’s syndrome caused by large mycotic aneurysm of the extracranial internal carotid artery after acute otitis media—case report. Neurol Med Chir (Tokyo) 50:45– 48, 2010
J Neurosurg: Spine / July 11, 2014
Manuscript submitted October 11, 2013. Accepted June 10, 2014. Please include this information when citing this paper: published online July 11, 2014; DOI: 10.3171/2014.6.SPINE13908. Address correspondence to: Brian P. Walcott, M.D., Department of Neurological Surgery, Massachusetts General Hospital, 55 Fruit St., White Bldg., Rm. 502, Boston, MA 02114. email: walcott. [email protected].
3
