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g:	Pitfalls of Management of Atlanto-Occipital Dislocation: A Case Report

Masahiro Aoyama, Muneyoshi Yasuda, Masahioro Joko, Mikinobu Takeuchi, Aichi Niwa, and Masakazu Takayasu

Department of Neurosurgery, Aichi Medical University, Nagakute, Aichi, Japan

Corresponding author:
Masahiro Aoyama
Department of Neurosurgery, Aichi Medical University
1-1 Yazakokrimata, Nagakute, Aichi 480-1195, Japan
Tel: +81 561623311
Fax: +81 561632879
E-mail: HYPERLINK "mailto:maoyama@aichi-med-u.ac.jp"maoyama@aichi-med-u.ac.jp


Abstract
Atlanto-occipital dislocation (AOD) is a traumatic condition that is rarely seen clinically because it is characteristically immediately fatal. With recent progress in pre-hospital care, the number of reports of AOD survivors is increasing. However, because the pathophysiology of AOD is not clearly understood, the appropriate strategy for initial management remains unclear. We herein report successful treatment of AOD and summarize the most appropriate management procedures. A 33-year-old man was transferred to our clinic after falling from a height of 10 m. Computed tomography revealed AOD. A hard collar was fitted on the first day and was replaced by a halo orthosis on the second day. However, the dissociation between the occipital condyles and atlas increased. Because of stretching of the cervical arteries, the patient developed decreased regional saturation of oxygen in the cerebrum and became stuporous. Abducens nerve palsy also developed. Therefore, longitudinal shortening of the halo orthosis was repeated with compression force and occipito-axial posterior fixation was performed on the 16th day. Postoperatively, he regained consciousness, the abducens nerve palsy resolved, and he experienced complete restoration of his daily living activities. Based on these findings, the immediate initial step in management of AOD should be to fit a halo vest to protect neural and vascular competence. Abducens nerve palsy is a warning sign of AOD, which results in life-threatening distortion of the arteries and brain stem. Horn�s grade is useful for assessing indications for surgery. When performing posterior fixation, C2 should be included because of the ligamentous architecture. 

Keywords: 
Abducens nerve palsy; atlanto-occipital dislocation; craniocervical junction; halo orthosis; occipito-axial posterior fixation 

Abbreviations:
AOD, atlanto-occipital dislocation; CT, computed tomography; DSA, digital subtraction angiography; GCS, Glasgow Coma Scale; MCA, middle cerebral artery; MRI, magnetic resonance imaging; rSO2, regional saturation of oxygen

Introduction
The atlanto-occipital junction, which comprises the lateral masses of the atlas and the occipital condyles, is one of the most important structures in the human body. It serves as a supporting base for the skull and contains vital organs such as the vertebral arteries and the spinal cord. Although this junction bears the heavy load of the head, it is also a pivot point for head motion. The joint is therefore flexible in any direction and has loose bone connections. Many ligaments surround it, maintaining its stability [1]. As suggested by this anatomical background, the atlanto-occipital junction is often vulnerable to dislocation in high impact injuries such as falling and motor vehicle accidents. Atlanto-occipital dislocation (AOD) has long been thought to be a rare clinical entity because of its rapid mortality [2, 3]. With recent developments in emergency medical care, however, the number of reports of AOD survivors is increasing [1, 2, 4-6]. We herein report the successful treatment of a patient with AOD and discuss an appropriate surgical strategy for managing this type of trauma.

Case Report
A 33-year-old male engineer fell 10 m while working on the roof of a factory. He was transferred to our hospital by helicopter. Upon arrival, his vital signs were stable with steady respiration. His Glasgow Coma Scale (GCS) score was E2V(T)M6 with normal pupillary findings. Although he did not respond to verbal commands, he had retained rectal tone.
Radiological examinations showed multiple injuries including contusions of the lung and liver and fractures of the left forearm, scapulae, and pelvis. Remarkable retropharyngeal swelling was evident on cervical X-ray analyses (Figure 1A). Computed tomography (CT) of the cervical spine showed translocation of both occipital condyles anterior to the lateral masses of the atlas (Figure 1B). The numeric measurements were consistent with published criteria for AOD [7-10]. In addition, magnetic resonance imaging (MRI) showed compression of the spinal cord by a hematoma caused by root avulsion injury (Figure 1C and D). Digital subtraction angiography (DSA) showed that the internal carotid and vertebral arteries were stretched bilaterally because of distraction between the atlas and occiput (Figure 1E). However, they were patent with no evidence of circulatory disruption. No evidence of vascular dissection or occlusion was found. 
On the day of admission, interventional embolization was performed to achieve hemostasis in the contused organs (Figure 2, left panel). The patient was fitted with a hard collar and placed supine on a bed. On the second day, a halo orthosis was fitted to augment cervical immobilization. He gradually regained consciousness (GCS E3V[T]M6). On the third day (Figure 2, middle panel), C3�4 laminectomy, and hematoma removal was performed. The right C4 and 5 nerve roots were severely avulsed. Because the dura mater was partially destroyed, a piece of fascia was used to cover it. After the first surgery, however, the patient became drowsy with progressive abducens nerve palsy. X-ray results showed worsening of the atlanto-occipital distraction in spite of the halo. Cerebral regional saturation of oxygen (rSO2) monitoring suggested decreased blood flow to the brain, with values being 78% on the first day, and 67% on the third day. Therefore, longitudinal shortening of the halo orthosis was repeated with compression force. This succeeded in reducing the AOD and his consciousness improved (Figure 2). On the 16th day, occipito-axial posterior fixation was performed. 
The patient was placed in the prone position with a halo in place. A connector was used to fix the halo ring to a Mayfield arm from the bed. Both vertebral arteries, which had been entrapped by the bony arch on C1 (arcuate foramen), were completely freed. There was a wide atlanto-occipital gap. Adequate curettage and bone grafting were performed. C2 pedicle screws and an occipital plate were inserted. During final fixation of a rod on each side, the Mayfield device was unlocked for manual compression of the halo, restoring occipital-C2 stability (Figure 3).
After fixation surgery, a hard collar was substituted for the halo. The patient regained the ability to perform daily living activities and was able to walk without support within 2 months. The abducens nerve palsy resolved completely.

Discussion
AOD is caused by a combination of considerable forces, namely hyperextension, lateral flexion, and/or hyperflexion. Because this type of injury generally results from a high impact trauma, patients often present with systemic traumatic damage. Thus, AOD has a significant mortality rate [3]. On postmortem examination, AOD is found in 20% to 30% of patients who die of cervical spine injury. Thus, AOD was once regarded as rare in the clinical setting [11, 12]. However, cases of survivors have recently been increasingly reported. This trend is associated with improvements in pre-hospital care. Clinicians should therefore be informed on how to promptly identify and manage AOD.
CT and MRI are essential for the diagnosis of AOD. It is difficult to exclude AOD on plain cervical X-rays because the relevant landmarks are not clearly visible on these images. By comparison, the specific landmarks and indices for AOD are clearly demonstrated by CT [1, 3, 6, 13]. These indices include the Powers ratio, x-line method, basion-dens interval, basion-axial interval, and condyle-C1 interval (Figure 4) [3, 7, 9, 10]. Moreover, CT may show edematous changes secondary to hemorrhage at the craniocervical junction. MRI is another useful tool for the diagnosis of AOD. It is appropriate for evaluation of breakage of the supporting ligaments and connective tissues of the junction [1, 3, 6, 13, 14]. It also identifies spinal cord injury. In addition, CT angiography, magnetic resonance angiography, and DSA are essential for investigating damage to the cervical vessels. In our case, for example, the internal carotid and vertebral arteries were stretched bilaterally by the AOD, which explained the patient�s deterioration in consciousness. 
Once AOD is suspected, a halo vest is more appropriate than a rigid cervical collar and axial traction because of the high risk of distraction at the craniocervical junction [1, 3, 15, 16]. However, a halo orthosis is not always safe. Our patient�s distraction certainly deteriorated during use of a halo. Van de Pol et al. also reported re-dislocation in the presence of a halo vest in a patient with AOD [13]. Because of the instability of a displaced atlanto-occipital junction, a halo orthosis was unable to provide enough compression force to immobilize the lesion [13]. Thus, frequent cervical spine radiographs should be performed to check for dissociation between the condyle(s) and the lateral mass.
In the current case, cerebral rSO2 values correlated with the degree of distraction between the condyles and the lateral mass of C1. The patient lost consciousness in parallel with worsening of the distraction. Distraction stretches the arteries, reducing blood flow and inducing drowsiness. Furthermore, the patient developed abducens nerve paralysis as the distraction increased. Abducens paralysis under the same conditions has been previously reported [1, 15, 17, 18]. The abducens nerve originates from the caudal border of the pons near the atlanto-occipital junction and runs longitudinally along the clivus. Its distal end is fixed by the dural ring of Dorello�s canal. This anatomical characteristic makes the nerve vulnerable to longitudinal stretching forces at the atlanto-occipital junction. Indeed, in this case the patient�s nerve paralysis resolved after AOD reduction. Thus, abducens nerve paralysis in patients with AOD can be a warning sign of insufficient cerebral perfusion. 
Principles of surgical indications for patients with AOD have been proposed by Horn et al. [6] (Table 1). Their grading of AOD was based on both CT and MRI findings. Grade I was defined as ligamentous damage without bony dissociation that could be managed by conservative observation with an external orthosis. Grade II, on the contrary, involved bony dissociation with ligamentous damage and required internal fixation [3, 6].
Occipito-cervical short fusion is the gold standard surgical technique for AOD. However, fusion should be extended to at least C2. The major ligaments of the atlanto-occipital connection are also responsible for competency between C1 and C2. In patients with AOD, therefore, atlanto-axial dislocation may become chronic [3]. If C2 is involved, an anchor screw does not have to be placed in C1.

Conclusions
Rigid cervical collars are contraindicated in the management of AOD because they can lead to increasing distraction at the craniocervical junction. The most appropriate management uses a halo vest. However, the distraction can deteriorate in spite of a halo vest. The level of consciousness and abducens nerve function are useful as early indications of distraction. After fitting halo fixation, internal surgical fixation is necessary if a Horn�s grade II injury is present. In such cases, C2 fixation should be considered.

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11. Alker GJ, Jr, Oh YS, Leslie EV (1978) High cervical spine and craniocervical junction injuries in fatal traffic accidents: a radiological study. Orthop Clin North Am 9: 1003-1010.
12. Bohlman HH (1979) Acute fractures and dislocations of the cervical spine. An analysis of three hundred hospitalized patients and review of the literature. J Bone Joint Surg Am 61:1119-1142.
13. van de Pol GJ, Hanlo PW, Oner FC, Castelein RM (2005) Re-dislocation in a halo vest of an atlanto-occipital dislocation in a child: recommendations for treatment. Spine 30: E424-428.
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16. Farley FA, Graziano GP, Hensinger RN (1992) Traumatic atlanto-occipital dislocation in a child. Spine 17: 1539-1541.
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18. Woodring JH, Selke AC, Jr, Duff DE (1981) Traumatic atlantooccipital dislocation with survival. AJR Am J Roentgenol 137: 21-24.









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