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Case report
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.
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
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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).
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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).
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S. Hong and G.-U. Kim International Journal of Surgery Case Reports 114 (2024) 109133
References [8] B. Ni, F. Zhou, N. Xie, et al., Transarticular screw and C1 hook fixation for os
odontoideum with atlantoaxial dislocation, World Neurosurg. 75 (3–4) (2011)
540–546.
[1] A.J. Electricwala, A. Harsule, V. Chavan, J.T. Electricwala, Complete
[9] R.P. Mudaliar, S. Shetty, K. Nanjundaiah, J.P. Kumar, J. Kc, An osteological study
atlantooccipital assimilation with basilar invagination and atlantoaxial subluxation
of occipitocervical synostosis: its embryological and clinical significance, J. Clin.
treated non-surgically: a case report, Cureus 9 (6) (2017), e1327.
Diagn. Res. 7 (9) (2013) 1835–1837.
[2] S.Y. Yang, A.J. Boniello, C.E. Poorman, A.L. Chang, S. Wang, P.G. Passias, A review
[10] R.A. Agha, T. Franchi, C. Sohrabi, G. Mathew, A. Kerwan, The SCARE 2020
of the diagnosis and treatment of atlantoaxial dislocations, Global Spine J. 4 (3)
guideline: updating consensus Surgical CAse REport (SCARE) guidelines, Int. J.
(2014) 197–210.
Surg. 84 (2020) 226–230.
[3] M. Venkatesan, R. Bhatt, M.L. Newey, Traumatic atlantoaxial rotatory subluxation
[11] J.S. Yeom, J.M. Buchowski, H.J. Kim, B.S. Chang, C.K. Lee, K.D. Riew, Risk of
(TAARS) in adults: a report of two cases and literature review, Injury 43 (7) (2012)
vertebral artery injury: comparison between C1-C2 transarticular and C2 pedicle
1212–1215.
screws, Spine J. 13 (7) (2013) 775–785.
[4] D. Hedequist, K. Bekelis, J. Emans, M.R. Proctor, Single stage reduction and
[12] R. Gnanadev, B. Ishak, J. Iwanaga, M. Loukas, R.S. Tubbs, A traumatic dislocation
stabilization of basilar invagination after failed prior fusion surgery in children
of the atlas from the axis in a patient with atlantooccipital assimilation, Cureus 11
with Down’s syndrome, Spine (Phila Pa 1976) 35 (4) (2010) E128–E133.
(4) (2019), e4402.
[5] W. Suphamungmee, L. Yurasakpong, K. Poonudom, et al., Radiological study of
[13] S. Wang, C. Wang, M. Yan, H. Zhou, G. Dang, Novel surgical classification and
atlas arch defects with meta-analysis and a proposed new classification, Asian
treatment strategy for atlantoaxial dislocations, Spine (Phila Pa 1976) 38 (21)
Spine J. 17 (5) (2023) 975–984.
(2013) E1348–E1356.
[6] S. Ghailane, M.A. Alsofyani, V. Pointillart, H. Bouloussa, O. Gille, Traumatic
[14] L.S. Jiang, L. Shen, W. Wang, H. Wu, L.Y. Dai, Posterior atlantoaxial dislocation
posterior Atlanto-axial dislocation: case report of an atypical C1-C2 dislocation
without fracture and neurologic deficit: a case report and the review of literature,
with an anterior arch fracture of C1, BMC Musculoskelet. Disord. 20 (1) (2019)
Eur. Spine J. 19 Suppl 2 (Suppl. 2) (2010) S118–S123.
612.
[7] A. Amirjamshidi, K. Abbassioun, M. Khazenifar, A. Esmailijah, Traumatic rotary
posterior dislocation of the atlas on the axis without fracture. Report of a case and
review of literature, Surg. Neurol. 71 (1) (2009) 92–97 (discussion 8.).