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International Journal of Surgery Case Reports 114 (2024) 109133

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International Journal of Surgery Case Reports


journal homepage: www.elsevier.com/locate/ijscr

Case report

Traumatic posterior dislocation of atlanto-axial joint with anterior arch


fracture in the patient of atlanto-occipital assimilation: A case report
Sungan Hong, Gang-Un Kim *
Department of Orthopaedic Surgery, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Incheon, Republic of Korea

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction and importance: Traumatic atlanto-axial dislocation (AAD) is relatively uncommon and can pose life-
Case report threatening risks. In this case, we describe a patient with a combination of AAD, an anterior arch fracture of the
Cervical spine dislocation atlas, and a rare congenital anomaly known as atlanto-occipital assimilation (AOA).
Atlanto-axial dislocation
Case presentation: A 70-year-old man presented with posterior neck pain and right-sided torticollis following an
Atlanto-occipital assimilation
accident that collision with a car while riding an electric scooter. Radiographic findings confirmed posterior AAD
Closed manual reduction
Neurologic deficit with anterior arch fracture of C1 in the inherent setting of AOA. The patient showed no neurologic deficit, so a
closed reduction technique using Gardner-Wells tongs was attempted in an awakened state, and successful
reduction could achieve without a neurologic deficit. After about three months of rigid brace application, head
and neck motion was allowed, and no recurrence of dislocation or cervical pain occurred during the follow-up
period of about one year.
Clinical discussion: Because the posterior AAD is usually accompanied by anterior arch fracture of atlas, the
transverse atlantal ligament remained intact. So nonoperative management after manual reduction was possible.
The presence of a C1 anterior arch fracture observed in our case can be regarded as an indicator predicting the
success of closed reduction of AAD.
Conclusion: Our case highlighted the successful nonoperative management of traumatic posterior AAD with an
accompanying anterior arch fracture of the atlas in a peculiar inherent combination of AOA through the closed
reduction technique and rigid cervical brace application.

1. Introduction traumatic condition based on congenital anomaly was successfully


treated through non-surgical techniques using manual reduction and
Atlantoaxial dislocation (AAD) is a condition that causes a loss of rigid brace application.
stability between the atlas and the axis [1,2]. Depending on the etiology In the case of traumatic AAD, treatment strategies have not been
of AAD, it can be broadly categorized into traumatic, congenital, and established because reported treatment cases are sporadic. Moreover,
inflammatory causes [2–5]. Among them, traumatic AAD is a relatively when combined with a very rare congenital anomaly such as AOA, there
uncommon condition, often observed in association with life- is no choice but to establish a treatment strategy tailored to each pa­
threatening circumstances [6–8]. Posterior AAD typically arises from a tient's condition. Through this case report, we attempt to provide a
traumatic mechanism involving hyperextension of the head and neck comprehensive description of the clinical presentation, radiological
and may be accompanied by an anterior arch fracture of the atlas [7]. findings, treatment approach, and long-term results of posterior AAD
In this case, we present a unique scenario where AAD, coupled with with anterior arch in the setting of AOA and to suggest conditions for
an anterior arch fracture of the atlas, occurred in conjunction with a rare successful non-surgical treatment of AAD. The work has been reported in
congenital abnormality known as atlanto-occipital assimilation (AOA) line with the SCARE 2020 criteria [10].
[9]. AOA can be defined as a state in which any part of the atlas is
completely or partially fused with the occiput of the skull [1]. A rare

Abbreviations: AAD, atlanto-axial dislocation; AOA, atlanto-occipital assimilation; CT, computed tomography; TAL, transverse atlantal ligament.
* Corresponding author at: Department of Orthopaedic Surgery, Incheon St. Mary's Hospital, College of Medicine, The Catholic University of Korea, 56 Dongsu-ro,
Bupyeong-gu, Incheon 21431, Republic of Korea.
E-mail address: yume14os@gmail.com (G.-U. Kim).

https://doi.org/10.1016/j.ijscr.2023.109133
Received 16 October 2023; Received in revised form 24 November 2023; Accepted 2 December 2023
Available online 13 December 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

2. Case description (Fig. 2c), along with right-sided posterolateral displacement of the
lateral mass of C1 (Fig. 2d). CT Angiography revealed the preserved
A 70-year-old man visited our clinic with posterior neck pain, right- continuity of the vertebral artery without evidence of vascular injury
sided torticollis, and an inability to rotate his head. About 8 h before (Fig. 2e).
visiting our clinic, the patient was involved in a collision with a car while The patient presented the incarcerated dislocation of the atlanto-
riding an electric scooter, during which he struck his forehead on the axial joint in the environment of the atlanto-occipital assimilation
ground, resulting in hyperextension of his neck. Initial neurological without any neurological deficits or vascular damage. Given the clinical
examination just after arrival at the emergency department revealed no presentation and radiographic findings, our treatment approach aimed
motor deficits or sensory abnormalities in either the upper or lower to achieve reduction and realignment of the atlantoaxial joint while
extremities. The patient's medical history included a stereotactic brain minimizing the potential risks associated with iatrogenic spinal cord
surgery that was deep brain stimulation approximately three years ago damage or cerebral ischemia resulting from arterial insufficiency during
for the treatment of his essential tremor. the reduction process. We opted for a nonoperative approach to mini­
The radiographic imaging of the cervical spine taken at the emer­ mize the potential risks associated with surgery, especially in a patient
gency department it revealed several notable findings, including right- with a history of deep brain stimulation. In the operating room with the
sided torticollis in the anteroposterior image and posterior displace­ awakened patient, we initiated the reduction process using the Gardner-
ment of the C1 facet of the atlantoaxial joint in the lateral image, with its Wells tongs under fluoroscopic guidance. Under continuous fluoroscopic
position located superiorly to the C2 pars (Fig. 1a, b). The open-mouth imaging guidance, gentle traction was attempted by pulling the handle
view showed a right-sided translation of the C1 lateral mass, along of Gardner-Wells tongs toward the cranial direction and a slight poste­
with an indistinct visualization of the odontoid process of C2 (Fig. 1c). rior vector. The procedure was conducted with utmost care to prevent
Subsequent computed tomography (CT) imaging revealed incarceration any further injury to the spinal cord or vertebral arteries. Subsequently,
of the posterior margin of the C1 lateral mass with impingement of C2 a clicking feeling was transmitted to the hand holding the tongs. At the
pars (Fig. 2a, b). CT imaging also indicated complete bone union at the same time, fluoroscopy confirmed that the atlanto-axial joint had been
atlanto-occipital joint and confirmed underlying atlanto-occipital reduced to its normal position. Cervical spine radiograph taken imme­
assimilation, as the C1 posterior arch was fused to the skull base diately after reduction showed that the surfaces of the C1–2 facet joint
(Fig. 2a, b). Furthermore, it demonstrated an anterior arch fracture of C1 remained in good contact, but the left-side lateral mass of C1 was a

Fig. 1. Initial cervical spine radiograph showed right-sided torticollis (a) and posterior displacement of the C1 facet of the atlantoaxial joint (b, white arrowhead).
The open-mouth view showed a right-sided translation of the C1 lateral mass, and indistinct visualization of the odontoid process (c, white arrow).

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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

Fig. 2. Computed tomography revealed incarceration of the posterior margin of the C1 lateral mass with impingement of C2 pars (white arrowhead) with atlanto-
occipital assimilation (white asterisk) (a–b). Also, anterior arch fracture of C1 (black asterisk, c) and right-sided posterolateral displacement of both C1 lateral masses
were noted (white arrow, d). CT Angiography confirmed no vascular injury around the injured cervical spine.

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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

slightly right-side translated feature (Fig. 3). No changes were observed 3. Discussion
in the patient's neurologic status before and after reduction. Neverthe­
less, in magnetic resonance imaging taken after reduction, the spinal Atlanto-axial dislocation combined with fracture of the anterior arch
cord at the C2 level showed focal high signal intensity C2 level (Fig. 4a), of the atlas is a rare and complex condition, mainly when it occurs in the
with no evidence of transverse atlantal ligament (TAL) injury (Fig. 4b). concomitant atlanto-occipital assimilation. This case report presents a
Following the successful reduction, we employed a 3-month appli­ unique instance of traumatic posterior AAD with an accompanying
cation of a rigid Miami cervical brace for immobilization and preventing anterior arch fracture of the atlas in the environment of AOA that was
neck extension and accompanying posterior translation of the head. The successfully treated by the closed reduction technique and rigid brace
posterior part of the Miami brace, which has a broad and wide shape, application without surgical intervention.
had the effect of pushing the occiput of the skull forward, which served Because approximately 70 % of AADs are anterior dislocations,
as an effective external device to prevent the reduced atlas from being atlantodental distance (ADI) measurements are often used in the diag­
translated and dislocated back. Also, the brace allowed minimal neck nosis and treatment planning of AAD [11]. The ADI is a small slitlike
motion, ensuring the reduction remained stable during the initial heal­ space between the posterior aspect of the anterior atlas ring and the
ing period. In addition, we provided additional patient education while anterior aspect of the odontoid process, and the measurement of ADI is
wearing the brace, such as not extending the neck beyond looking from a line projected superiorly along the anterior border to the axis
straight ahead and moving the torso instead of turning the head from body to the anterior arch of the atlas [2]. However, in posterior AAD,
side to side. After discharge, regular follow-ups were conducted at such as our patient's case, the lateral mass of the atlas is displaced
postreduction 1, 3, 6 months and 1 year. Radiographic assessments used posteriorly, and the anterior arch remains alone in front of the dens due
cervical plain radiograph to monitor the reduction and alignment sta­ to fracture. Therefore, the measurement of ADI either shows a negative
bility, and serial radiography consistently demonstrated stable reduc­ value or does not accurately reflect the status of the patient's dislocation.
tion of atlanto-axial joint. Cervical spine CT taken 3 months after Ghailane et al. mentioned the meaning of negative ADI as an indicator
reduction showed that the C1–2 facet joint was maintained as a stable that the dens of C2 migrate anteriorly and not posteriorly against the
construct, but the fracture gap at the C1 anterior arch remained (Fig. 5a, spinal cord [6].
b). The Miami brace was no longer worn three months after dislocation A previous report on AAD accompanied by AOA included a case
reduction, and all head and neck motion was allowed to the extent reported by Gnanadev et al. [12]. However, unlike this case, it was an
possible. Even without wearing a brace, symptoms such as recurrence of anterior AAD, and the patient arrived at the hospital without con­
dislocation or cervical pain did not occur during the follow-up period. sciousness or spontaneous breathing. Therefore, there was no evaluation
Also, a flexion-extension lateral radiograph of the cervical spine taken at or treatment other than the initial lateral cervical spine radiograph. This
the post-reduction one year showed no evidence of atlanto-axial insta­ case was a case of posterior AAD accompanying C1 anterior arch frac­
bility such as an increase of atlanto-dental interval (Fig. 5c, d). ture in the peculiar inherent setting of AOA. The patient's vital signs
were normal, and there was no neurologic deficit or vascular compro­
mise. Therefore, a more careful reduction strategy and thoughtful

Fig. 3. Cervical spine radiograph taken immediately after reduction.

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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

Fig. 4. Post-reduction magnetic resonance imaging showed that the spinal cord at the C2 level showed focal high signal intensity C2 level (a), with no evidence of
transverse atlantal ligament (TAL) injury (b).

postreduction management plan were required. 4. Conclusion


Ghailane et al. [6] reported successful C1 anterior arch fracture
treatment accompanying posterior AAD using closed reduction and rigid In conclusion, this case highlights the successful nonoperative
brace application. Unlike our case, in the report by Ghailane et al., management of traumatic atlanto-axial dislocation in a patient with
congenital anomalies such as AOA were not accompanied, and the de­ atlanto-occipital assimilation and an accompanying anterior arch frac­
gree of posterior translation of C1 observed on CT was limited to the ture of the atlas through the closed reduction technique and rigid cer­
posterior margin of the C2 facet. In our case, the lateral mass of C1 vical brace application. Throughout the entire decision-making process,
completely deviated from the C2 facet joint. It showed an incarcerated including initial evaluation, reduction maneuver, postreduction man­
feature at the superior margin of the pars, so we had no choice but to agement that resulted in successful outcome, we would use this report as
think more deeply about concerns such as reduction strategy, post­ an appropriate example of handling unusual emergencies accompanied
reduction management, and the development of recurrent dislocation or by scarce patient environments.
instability. Nevertheless, it was agreed that the hypothesis proposed in
the report by Ghailane et al. that posterior AAD was accompanied by Consent
anterior arch fracture of atlas indicates that the TAL is intact [6].
Therefore, a maintenance plan was set with rigid cervical brace appli­ Written informed consent was obtained from the patient for publi­
cation after successful manual reduction. In our case, although the cation and any accompanying images. A copy of the written consent is
radiographic union of the C1 anterior arch was not confirmed after available for review by the Editor-in-Chief of this journal on request.
wearing a rigid brace for three months, no recurrence of AAD or
occurrence of atlanto-axial instability was observed during the 1-year Ethical approval
follow-up period.
C1 anterior arch fracture accompanying posterior AAD can be This paper is a case report using already prepared medical records
considered an indirect radiographic sign that tentatively indicates an and images, and therefore ethics approval is not required.
intact TAL. Also, we confirmed through MRI taken after reduction that
the TAL was intact without disruption. For that reason, even though the Funding
displaced anterior arch of the atlas still exists after reduction, the patient
in our case can be classified as type II according to Wang's AAD classi­ None.
fication, in which successful reduction is possible through skeletal
traction [13]. Therefore, if the anatomical construct can be restored CRediT authorship contribution statement
through manual reduction, the external immobilization procedure using
a brace will likely be successful. Jiang et al. [14] reported pure posterior Sungan Hong: Writing the paper, preparing figures.
AAD without anterior arch fracture. In this case, reduction through the Gang-Un Kim: Study concept and design, data collection, data
closed reduction technique failed, and open reduction through partial interpretation, writing the paper.
odontoidectomy was required. Conversely, the presence of a C1 anterior
arch fracture observed in our case can be regarded as an indicator pre­ Guarantor
dicting the success of closed reduction of AAD.
In our case, it is assumed that AOA acted further to concentrate Gang-Un Kim: Incheon St. Mary's Hospital, Republic of Korea.
mechanical posterior force on the atlanto-axial joint, resulting in a more
severe form of AAD compared to previously reported cases. On the other Research registration number
hand, after reduction, the AOA may have provided an environment in
which the external fixation effect could be concentrated on the atlanto- Not applicable.
axial joint due to the inherent stability of the atlanto-occipital joint.

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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

Fig. 5. Cervical spine CT taken three months after reduction showed that the C1–2 facet joint was maintained as a stable construct (a), although the fracture gap at
the C1 anterior arch remained (b). Flexion-extension lateral radiograph of the cervical spine taken at the post-reduction one year showed no evidence of atlanto-axial
instability (c, d).

Declaration of competing interest Acknowledgements

No funds were received in support of this work. No benefits in any None.


form have been or will be received from a commercial party related
directly or indirectly to the subject of this manuscript.

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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133

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