Minor Trauma in Ankylosing Spondylitis Causing Combined Cervical Spine Fracture and Esophageal Injury

Accepted Manuscript Minor trauma in ankylosing spondylitis causing combined cervical spine fracture and oesophageal injury C.R. Vonhoff, MBBS, PhD, K. Scandrett, MBBS, D. Al-Khawaja, FRACS PII:

S1878-8750(18)31658-9

DOI:

10.1016/j.wneu.2018.07.180

Reference:

WNEU 8745

To appear in:

World Neurosurgery

Received Date: 27 March 2018 Revised Date:

19 July 2018

Accepted Date: 21 July 2018

Please cite this article as: Vonhoff CR, Scandrett K, Al-Khawaja D, Minor trauma in ankylosing spondylitis causing combined cervical spine fracture and oesophageal injury, World Neurosurgery (2018), doi: 10.1016/j.wneu.2018.07.180. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.

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Minor trauma in ankylosing spondylitis causing combined cervical spine fracture and oesophageal injury.

Authors:

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Scandrett K.A,: MBBS Al-Khawaja D.A: FRACS

Neurosurgery Department Nepean Hospital, Derby St, Penrith NSW Australia 2750

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Vonhoff C. R.A,: MBBS, PhD

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Corresponding Author: Vonhoff C. R.

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Mailing Address: Neurosurgery Department Wollongong Hospital 252 Loftus St & Locked Bag 8808, South Coast Mail Centre NSW Australia 2521 Email Address: [email protected]

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Phone: +61 414 977 811

Conflict of interest: None declared

Sources of financial and material support: None declared

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No portion of the contents of this paper have been presented or published previously

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ABSTRACT Background: We illustrate the case of an unstable fracture of the cervical spine in ankylosing spondylitis with associated oesophageal injury after minor trauma.

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Case Description: A 66 year old man fell backwards from the first rung of a ladder

sustaining a transverse fracture of C6 vertebral body and a new diagnosis of ankylosing

spondylitis. He was taken for surgical fixation, however his oesophagus was discovered

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entrapped within the fracture at the time of surgery. Despite the severity of the injury, with surgical reduction, fixation and oesophageal exclusion this patient made a full recovery.

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Conclusion: This case demonstrates the severity of injury after minor trauma in the context of ankylosing spondylitis, the capacity for full recovery in oesophageal perforations in spinal trauma, and that clinical suspicion of such injuries allows early diagnosis, treatment and

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reduced complications.

KEYWORDS: cervical spine injury, oesophageal perforation, ankylosing spondylitis,

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cervical fracture

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CASE SUMMARY

History: A 66 year old builder fell flat on his back from standing height. He was immobilised at the scene and transferred to hospital where he described no radicular pain, paraesthesia or weakness, and had no symptoms of dysphagia, odynophagia or dysphonia. The patient was nursed in full spinal precautions with the cervical spine immobilised. CT of his cervical spine demonstrated extensive ossification of the anterior longitudinal ligament and auto-fusion of C4/5, C5/6 and C6/7. A transverse fracture of the C6 vertebral body was evident extending bilaterally through lateral masses and transverse processes, and the C6

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spinous process, with 20 degrees of dorsal rotation and associated anterior paravertebral soft tissue swelling, but no free gas (Figure 1). Subsequent CT of thoracolumbar spine demonstrated features consistent with ankylosing spondylitis including extensive ossification of the anterior longitudinal ligament, posterior longitudinal ligament and supraspinous

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ligaments from T2 to L1 along with ossification of the annulus fibrosis at these levels and partial fusion of bilateral sacro-iliac joints. MRI of the cervical spine was consistent with CT appearances showing canal stenosis at C6 vertebral level, but no cord compression or

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associated cord signal change. A moderate sized haematoma can be seen in the prevertebral space from the clivus at the base of skull down to T2 vertebral level. However at the level of

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the fracture, this space is obliterated by the posterior deviation of the prevertebral fascia, and the phalangeal-oesophagus junction which appears to abut the fracture. Due to delays with imaging and consent, surgery was undertaken 7 days post-injury. During this time the patient remained immobilised, and did not develop neurological deficits, however in the 2days prior

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to surgery suffered some dysphasia with difficulty clearing oral secretions.

Operation: Surgery was planned for an anterior and posterior fusion with an anterior plate

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followed by posterior lateral mass/pedicle screw fixation. The prevertebral space was

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approached through a curved incision from left side and the fractured level was immediately evident. The rostral fracture fragment was anterolisthesed on the caudal fragment, and the posterior wall of the oesophagus was entrapped within the fracture line in the anterior aspect of the C6 vertebral body. The prevertebral tissues were oedematous and showed signs of acute inflammation, however there was no evidence of pus, collection or otherwise of localised infection.

The oesphagus was dissected free with the aid of an upper GI surgeon, and a 2cm horizontal

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laceration in the muscular layer was closed primarily. It was unclear intraoperatively if the defect had transgressed the inner mucosal layer of the oesophagus, however an intraoperative gram stain before oesophageal dissection was positive for gram positive cocci and gram positive rods with subsequent growth of group B Streptococcus. This suggests full thickness

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oesophageal tear either preoperatively or during the initial soft tissue dissection in the approach to the prevertebral space. A nasogastric tube was not placed as the surgeon

believed the perforation was so small that primary closure was sufficient. Following the

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repair of the oesophagus, the fracture was debrided and an iliac crest graft was placed in the fracture cavity and reduction achieved under xray guidance. An anterior plate was not placed

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due to risk of infection, and a wound drain was left in situ. Posterior C4-T1 posterior lateral mass/pedicle screw fixation was performed as planned without complication (Figure 2).

Postoperative Course: The patient was kept intubated for 24 hours post operatively, and

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when extubated was well with no neurological deficits. He was nursed in a collar for the first 3 weeks post operatively. Oral feeding was avoided and the patient was initially fed by total parenteral nutrition until on the seventh post-operative day a barium swallow showed a

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persistent contrast leak at the posterior aspect of the cervical in keeping with oesophageal

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perforation (Figure 3). A percutaneous endoscopic gastrostomy was placed to allow nutrition supplementation and oesophageal exclusion. Serial barium swallow imaging showed resolution of oesophageal leak at 1 month post surgery. The surgical drain was removed and an oral diet was gradually reintroduced. He was treated with intravenous benzyl penicillin and oral metronidazole until resolution of the oesophageal leak. 3 months post operatively he was well, neurologically intact with no signs of infection.

DISCUSSION

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ACCEPTED MANUSCRIPT Oesophageal injury is occasionally seen in penetrating trauma, however it is rarely a

manifestation of blunt trauma. The occurrence of combined oesophageal and cervical spine injuries has been reported in few case studies, almost exclusively in the context severe trauma 1 ,2 ,3 ,4

. Beer-Furlan and colleagues reported a single case of oesophageal perforation due to

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a C5 anterior osteophyte fracture in an 80 year old lady after a fall from standing height in 2006 3 . The injury was initially missed on presentation to the emergency department, and was picked up subsequently due to representation with severe sepsis and subcutaneous

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emphysema. Oesophageal injury most commonly occurs at the normal anatomical

constrictions, particularly at the level of cricopharyngeus muscle (c5/6) where the oesophagus

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is relatively immobile. Most cases are diagnosed late, required ICU support, and demonstrate a high rate of mortality due to complications such as pneumomediastinum, polymicrobial sepsis, inflammatory mediastinitis, and posterior mediastinal abscess. Delayed diagnosis can result in need for oesophagectomy as opposed to primary repair, and can significantly

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increase mortality.

The presented case demonstrates an occult oesophageal injury identified at time of surgery

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for an unstable cervical spine fracture after minor trauma in a patient with ankylosing

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spondylitis. Ankylosing spondylitis is an inflammatory arthropathy primarily affecting the axial skeleton. It manifests as autofusion of the spinal column and sacroiliac joints caused by chronic inflammation and new bone formation. It is also associated with osteoporosis combining to make a long, rigid spine brittle and prone to fractures with minimal trauma 6 . This is particularly evident in the cervical spine due to the weight and mobility of the head against the lever arm segments 7 . Spinal fractures in ankylosing spondylitis are frequently unstable injuries, commonly present with neurological injury, and can have very high reported mortality rate (18-32%) 4 . 2 previous cases of combined oesophageal and cervical

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spine injury in the setting of ankylosing spondylitis have been published. Both were in circumstances of a significant mechanism of trauma – a motor vehicle accident and a fall from height. The oesophageal injury in both cases was initially overlooked, discovered

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subsequently due to development of local infection and severe sepsis 8 .

In the current case, the oesophagus was discovered trapped within the vertebral body fracture at the time of operative fixation rather than simply perforated by a bony spur or fracture

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fragment. The patient had been afebrile and hemodynamically stable despite 1week delay between injury and surgical fixation. Intraoperatively there was no evidence of abscess or

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significant infection, however an intraoperative gram stain did show evidence of bacteria in the surgical wound. This suggests that a full thickness oesophageal injury occurred at the time of surgery, or shortly preceding surgery. Laceration of the muscular layer and entrapment in the bony injury likely occurred earlier as evidenced by the oedematous

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oesophagus. Fortunately, we were able to reduce the fracture from an anterior approach and fixate posteriorly without significant ongoing morbidity associated with oesophageal rupture.

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An intraoperative nasogastric tube was not placed intraoperatively because the defect in the

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oesophageal wall was small, and we were confident in the complete repair of the injury. Post-operative barium swallow investigation demonstrated inadequate surgical closure, and therefore a protracted coarse of oesophageal exclusion followed. Reoperation to repair the persistent oesophageal leak was considered, however in the experience of the surgeon the small leak was expected to heal within a relatively short timeframe. In retrospect we suggest the oesophageal repair should be undertaken after fracture fixation. This would ensure no further traction or pressure on the oesophagus after the repair which could minimise the chance of postoperative leak.

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We demonstrate a case of combined oesophageal and cervical spine injury after minor trauma in a patient with ankylosing spondylitis. This rare case highlights the required index of

early diagnosis in the management of these injuries.

DISCLOSURE

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suspicion for such injuries which can easily escape early identification, and the importance of

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This research did not receive any specific grant from funding agencies in the public,

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commercial, or not-for-profit sectors.

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ACCEPTED MANUSCRIPT REFERENCES:

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1. Beer-Furlan A, Brock RS, Mendes LS, Mutarelli EG: Minor blunt cervical spine trauma associated with esophageal perforation and epidural empyema. Acta Neurol Belg 116:691–693, 2016 2. Caron T, Bransford R, Nguyen Q, Agel J, Chapman J, Bellabarba C: Spine Fractures in Patients With Ankylosing Spinal Disorders. Spine 35:E458, 2010 3. Fahr M, Thomas B, Barker D: Esophageal injury from cervical spine fracture in blunt trauma. Am Surg 76:915–6, 2010 4. Nérot, Jeanneret, Lardenois, Lépousé: Esophageal perforation after fracture of the cervical spine: case report and review of the literature. Journal of spinal disorders & techniques 15:513–8, 2002 5. Prieto-Alhambra, Muñoz-Ortego, Vries D, Vosse, Arden, Bowness, et al.: Ankylosing spondylitis confers substantially increased risk of clinical spine fractures: a nationwide casecontrol study. Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA 26:85–91, 2015 6. Qian S-J, Ye X-S, Chen W-S, Li W-L: Missed diagnosis of oesophageal perforation in ankylosing spondylitis cervical fracture: Two case reports and literature review. J Int Med Res 44:170–175, 2016 7. REDDIN A, MIRVIS S, DIACONIS J: Rupture of the Cervical Esophagus and Trachea Associated with Cervical Spine Fracture. J Trauma Acute Care Surg 27:564, 1987 8. Tomaszek D, Rosner M: Occult Esophageal Perforation Associated with Cervical Spine Fracture. Eur Man Med 14:492, 1984 9. Westerveld, Verlaan, Oner: Spinal fractures in patients with ankylosing spinal disorders: a systematic review of the literature on treatment, neurological status and complications. European spine journal : official publication of the European Spine Society, the European Spinal Deformity Society, and the European Section of the Cervical Spine Research Society 18:145–56, 2009

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FIGURE LEGENDS

Figure 1: sagittal CT (A right, B mid, C left) imaging showing ossification of spinal ligaments and annulus fibrosis and C5 vertebral body fracture. D Sagital MRI T2 sequence showing anterior longitudinal ligament rupture, prevertebral haematoma and C5 vertebral body fracture Figure 2: Sagittal CT imaging of the cervical spine showing post-operative fixation (A midsagittal, B right, C left) Figure 3: Barium swallow showing persistent leak of contrast into the prevertebral space

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ACCEPTED MANUSCRIPT HIGHLIGHTS -Patients with ankylosing spondylitis are at risk of unstable vertebral fractures from minor trauma -Combined cervical spine and oesophageal injury is a rare occurrence in blunt trauma and is

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almost universally fatal. It has not been reported previously in minor trauma.

-We report a case of minor trauma causing a combined cervical spine and oesophageal

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injury in a patient with ankylosing spondylitis. In this case the patient recovered to

premorbid status after internal fixation of the cervical spine injury and repair of the

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oesophageal injury