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TREATMENT OF POST-TRAUMATIC ATLANTO-AXIAL ROTATIONAL FIXATION IN ADULTS
Rerikh V. V., Sinyavin V. D., Anikin K. A.
Tsivyan Novosibirsk Research Institute of Traumatology
and Orthopaedics, Novosibirsk State
Medical University,
Novosibirsk, Russia
The Atlanto-axial
facet joints are the main ones in the upper cervical complex, providing not
only support, but also more than 90 % of the range of motion in this section of
the spine. Together with the Cruvelier joint and ligamentous apparatus, they
determine stability at this level [1]. Atlanto-axial dislocation can be identified
on the basis of anamnesis and clinical manifestations. But the main thing in
diagnostics is visualization using radiation diagnostic methods, which allow
assessing the state of bone structures and ligamentous apparatus [2, 3]. Stable
deformations that occur during subluxations and dislocations, as a rule, are
due to transcendental rotation at the C1-C2 level. This injury was
called antlanto-axial rotational fixation (AARF) [4].
In adults,
AARF rarely occurs as a result of trauma, but it is a consequence of its high
energy [5-9]. According to Abolfazl Rahimizadeh et al. (2019), studies of this
injury were found in the literature, including the treatment of 57 victims
[10]. All authors emphasize the rarity of this injury in adults [5-9]. The
diagnosis of AARF resulting from trauma is usually a dilemma even for spinal
surgeons [11]. Delayed diagnosis and treatment of AARF can lead to neurological
deficit or impaired respiratory function and death due to spinal cord injury at
the upper cervical level [6, 12, 13]. The attitude to the treatment of these
injuries is rather contradictory both in the early and late periods after the
injury.
In case of
trauma more than 6-8 weeks ago, most authors prefer surgical treatment [11-14].
The combination of AARF with a fracture of the bone structures of the upper
cervical spine is an even rarer situation. In this case, as a rule, a surgical
method of treatment is chosen, regardless of the duration of the injury [15].
Often the reason for the difficulties in making a decision is the lack of
understanding of the methods of diagnosis, tactics and methods of treatment of
these injuries.
The objective of this
report is to present the outcome of successful treatment of a patient with
post-traumatic AARF associated with a C2 fracture, as well as existing
approaches to the treatment of such injuries in adults based on literature
data.
MATERIALS AND METHODS
The literature search was carried out using the databases of scientific electronic libraries eLibrary and PubMed for the period 2001-2020. Criteria for inclusion and exclusion of sources in the study were defined for analysis in the evaluation of literature data. For the first stage of the literature search, the keywords were (in English): "atlanto-axial instability". At the second stage, only full-text articles were selected from the sources. At the third stage, sources containing information were identified: systematic and literary reviews, meta-analyses, clinical cases. At the last stage of the search, studies were selected containing data on post-traumatic atlanto-axial fixations (types 1, 2 according to Fielding JW, Hawkins RJ) in adult patients with a follow-up period of at least 6 months. As a result, 24 full-text articles were identified (Tables 1, 2), which described 41 cases of treatment of patients aged 19 to 70 years with post-traumatic AARF, 11 of which had fractures of the C2 vertebra. Treatment was carried out up to 6 weeks after injury in 21 patients, more than 6 weeks in 20 patients.
Table 1. Post-traumatic atlanto-axial rotational fixation
|
N |
Authors of publications |
Year |
male/female |
Age (years) |
Type of injury |
Diagnosis |
Time of injury (weeks) |
Type of treatment |
Favorable / poor outcomes |
|
1 |
Castel et al. [30] |
2001 |
1/0 |
41 |
Sport |
AAF |
> 6 |
Conservative |
1/0 |
|
2 |
Weisskopf et al. [29] |
2005 |
3/2 |
38, 36, 26 / 21, 23 |
N/D |
AAF |
> 6 |
Surgical. |
5/0 |
|
3 |
Sinigaglia et al. [31] |
2008 |
1/2 |
29 / 26, 21 |
TA |
AAF |
> 6 |
Halo |
2/1 (after 45 days onset of treatment, headache) |
|
4 |
Jeon et al. [13] |
2009 |
0/1 |
0/25 |
TA |
AAF |
< 6 |
C
|
1/0 |
|
5 |
Singh et al. [9] |
2009 |
0/1 |
0/25 |
TA |
AAF + hemiparesis |
< 6 |
C |
1/0 |
|
6 |
Goel et al. [11] |
2010 |
1/0 |
28/0 |
CT |
AAF |
< 6 |
Surgery + Halo |
1/0 |
|
7 |
Meza Escobar et al. [20] |
2012 |
0/1 |
0/19 |
ДТ TA |
ААРФ AAF |
< 6 |
К C |
1/0 |
|
8 |
Venkatesan et al. [17] |
2012 |
0/2 |
0 / 20, 52 |
TA |
AAF |
< 6 |
Halo |
1/0 |
|
9 |
Maida et al. [21] |
2012 |
1/0 |
27/0 |
TA |
AAF |
< 6 |
C |
1/0 |
|
10 |
Han et al. [22] |
2014 |
1/0 |
22/0 |
TA |
AAF |
< 6 |
C |
1/0 |
|
11 |
Qi et al. [33] |
2015 |
4/1 |
19, 51, 33, 34 / 29 |
N/D |
AAF |
> 6 |
S |
5/0 |
|
12 |
Garcia Pallero et al. [7] |
2019 |
0/1 |
28 |
TA |
AAF |
< 6 |
C |
1/0 |
|
13 |
Abolfazl Rahimizadeh et al. [10] |
2019 |
1/1 |
56/39 |
TA |
AAF |
> 6 |
S |
2/0 |
|
14 |
Barimani et al. [ |
2019 |
0/1 |
0/66 |
R |
AAF (type 1) |
> 6 |
X |
1/0 |
|
15 |
Singla et al. [32] |
2020 |
1/0 |
25/0 |
TA |
AAF (type 1) |
> 6 |
X |
1/0 |
|
|
Hugo et al. |
2020 |
0/1 |
65 |
CT |
AAF |
< 6 |
Х |
1/0 |
|
16 |
Eghbal et al. [6] |
2017 |
1/0 |
35/0 |
CT |
AAF (type 1) |
< 6 |
X |
1/0 |
|
17 |
Hawi et al. [ |
2015 |
0/1 |
0/34 |
TA |
ААРФ |
< 6 |
K |
1/0 |
Note: AAF – atlanto-axial rotational fixation; TA - traffic accident; CT - catatrauma; R - during ritual; C - conservative treatment; S - surgery; Halo - halofixation.
Table 2. Posttraumatic atlanto-axial rotational fixation in combination with C2 fracture
|
N |
Authors |
Year |
male /female |
Age (years) |
Type of injury |
Diagnosis |
Time of injury (weeks) |
Type of treatment |
Favorable /poor outcomes |
|
1 |
Chaundhary et al. [26] |
2015 |
1/0 |
26 |
TA |
AARF + C2 |
< 6 |
S |
1/0 |
|
2 |
Bellil et al. [5] |
2014 |
0/1 |
0/56 |
TA |
AARF + C2 |
< 6 |
Halo |
1/0 |
|
3 |
Seybold et al. [23] |
2003 |
0/1 |
0/21 |
TA |
AARF + C2 |
< 6 |
Halo |
1/0 |
|
4 |
Oh et al. [25] |
2010 |
1/0 |
37/0 |
CB |
AARF + C2 |
< 6 |
Halo |
1/0 |
|
5 |
Kim et al. [24] |
2007 |
1/0 |
0/34 |
CT |
AARF + C2 |
< 6 |
S |
1/0 |
|
|
Yang et al. [] |
2015 |
1/0 |
70/0 |
TA |
AARF + C2 |
< 6 |
C |
1/0 |
|
|
Peyriere et al[] |
2017 |
2/3 |
60 |
TA – 1, |
AARF + C2 |
< 6 |
S |
5/0 |
Note: AAF – atlanto-axial rotational fixation; TA - traffic accident; CT - catatrauma; CB -cervical blow; ES - epileptic seizure; C - conservative treatment; S - surgery; Halo - halofixation.
DESCRIPTION OF THE CLINICAL CASE
Study
approved by Biomedical Ethics Committee of Tsivyan Novosibirsk Research Institute of Traumatology
and Orthopaedics (the statement 032/22 from the protocol 007/22, October 27,
2022). It complies with WMA Declaration of Helsinki - Ethical Principles for
Medical Research Involving Human (2013), and the Order by Health Ministry of RF
(April 4, 2016, No. 200n) - On approval of the rules of good clinical practice.
All participants
gave
voluntary
consent
to
the
study.
The
female patient (age of 21) was injured in a traffic accident in which she was
thrown from a car. She lost consciousness, after the restoration of which she
noted an extreme turn of the head to the right, the impossibility of any
movements in the cervical spine due to sharp pain in the occipital-cervical
region, more on the left. In conditions of immobilization with a collar, she
was taken from the scene and hospitalized in one of the central regional
hospitals, where an examination was carried out and a diagnosis was made: "Concomitant
injury, traumatic brain injury (TBI), fracture of the C2 odontoid process,
rotational subluxation of the C1 vertebra, torticollis." Conservative
treatment was carried out in the form of fixation of the cervical spine with an
impromptu cotton-gauze collar of the Shants type, and symptomatic therapy.
Despite the pathological position of the head, limitation of movements and pain
in the cervical spine, the neurosurgeons of the medical institutions chose
expectant treatment tactics.
Eight
weeks after the injury, the patient with persistent rough rotation of the head
to the right came to the attention of a traumatologist from Novosibirsk
Research Institute of Traumatology and Orthopaedics. The patient has been on
bed rest since the day of the injury. Despite the age of the injury, attempts
to independently turn the head to the left were impossible due to severe pain
in the cervical-occipital region. At the same time, they were accompanied by
the appearance of dizziness, a decrease in sensitivity in the right half of the
trunk and legs. The same phenomena arose when trying to sit. According to the
initial data and additional examination data (MSCT (Fig. 1) and MRI of the
cervical spine (Fig. 2), MSCT of the neck with contrast (angiography) (Fig. 3)),
the diagnosis was made: “Old
atlanto-axial fixation (Fielding type 1) ), a consolidating fracture of the
roots of the arches of the C2 vertebra (on the right with the transition to the
body, the articular facet of C2) with displacement (type 1 according to
Effendi)”.
The rotation
of C1 in relation to C2 was 50 degrees. MRI showed no signs of ruptures of the
pterygoid and transverse ligaments of the atlas. Under conditions of manual
traction and sequentially performed rotation, flexion and extension, the
vicious position of the head was eliminated to a neutral position, followed by
fixation with a Philadelphia-type collar. MSCT of the cervical spine
immediately after reduction and in the position of turning the head to the left
showed the normalization of the relationship at the level of C1-C2 to neutral.
Figure 1. MSCT of the cervical spine after injury: a) sagittal
reconstruction: fracture of the C2 arch root on the right, with transition to
the articular facet; displacement of the C1 lateral mass posteriorly; b) sagittal
reconstruction: displacement of the lateral mass of the C1 vertebra from the
left anteriorly; c) axial reconstruction: rotational displacement of C1
vertebra to the right in relation to C2 50 degrees
Figure 2. MRI of the cervical spine after trauma. Sagittal
view, center: stenosis of the spinal canal at the level of the C2 tooth, with
almost complete absence of anterior "reserve space".
Figure 3. 3D reconstruction of MSCT angiography of the
cervical spine 2 months after injury, rotational displacement of the C1
vertebra is preserved
During the
study, a head turn of 40 degrees to the left was achieved, which was limited by
the appearance of pain in the cervical-occipital region. The volume of rotation
between C1 and C2 was only 8 degrees (Fig. 4, 5). A slight asymmetry of the
spaces between the tooth of the C2 vertebra and the lateral masses of the C1
vertebra was preserved. The displacement of the bone fragments of the C2
vertebra did not worsen. Previous
dizziness, and incoming sensory disturbances disappeared. The vicious position
of the head was eliminated. She began to sit up and walk on her own.
Figure 4. 3D reconstruction of MSCT of the cervical spine
after reduction and elimination of AARF: slight asymmetry of the spaces between
the odontoid process of the C2 vertebra and the lateral masses of the C1
vertebra: on the right – 2.8 mm, on the left – 3.5 mm. Relationships
in the lateral atlanto-axial joints restored.
Figure 5. MSCT of the cervical spine on the second day
after closed elimination of AARF with the head turned to the left. Axial:
8 degree
rotation
between C1 (a) and C2 (b)
From the
first day after the elimination of AARF, physiotherapy exercises were started,
which included a combination of isometric gymnastics with a mandatory daily
increase in head rotation to the left, flexion and extension, without attempts
to rotate to the right. Immobilization with a Philadelphia-type collar with a
halo-fixator was carried out for the period of isometric gymnastics, as well as
between periods of active movements and during sleep. Treatment continued on an outpatient basis for 2 months.
5 months
after the simultaneous elimination of AARF, MSCT of the cervical spine revealed
consolidation of the C2 fracture and complete restoration of the anatomy of the
upper cervical spine (Fig. 6). By this time, the adjusting tilt of the head to
the right, which was observed after the termination of immobilization with the
collar, was eliminated, and movements in the cervical spine were restored in
full range. The pain score on VAS was 0.
Figure 6. 3D reconstruction of the control MSCT of the
cervical spine 5 months after the removal of AARF. Consolidated non-displaced
fracture of the roots of the C2 vertebral arch. The relationship of C1-C2
vertebrae is not changed. Lateral atlanto-dental intervals: D = S = 2.4 mm
DISCUSSION
AARF usually
occurs in children, especially in the presence of diseases that cause ligament
failure [8, 16]. Posttraumatic AARF in adults is a very rare occurrence, with
relatively few case reports or examples in the English-language literature [5,
6, 7].
For many
years of providing emergency care to victims with spinal injury (more than 30
years), we have not identified a single fact of post-traumatic AARF. The case
described in this article was probably the first in the clinics where the
victim applied from the day of the injury. There are no recommendations that
determine the direction of treatment of such patients. In this case, the articular
facets C1 and C2 retained the contact of the cartilaginous surfaces in limited
areas on the right, and on the left, on the border of the articular surfaces.
We must agree with Venkatesan M. et al. (2012), who suggested that these
injuries be called subluxations, based on the concept of articular surface
contact in damaged joints [17]. But the terminology and classification proposed
by Fielding JW, Hawkins RJ. (1977) is used more often in practice, although
they also define this condition as subluxation [4]. These
authors described four different types of atlanto-axial instability depending
on the severity of the injury. Type 1 refers to movement of the facet joints
within the normal range of atlantoaxial rotation without an increase in the
anterior atlantodental interval, which can lead to narrowing of the spinal
canal at significant degrees of rotation, as in the case described. In the
available literature, no gradations of these displacements were found as the
severity of AARF. This definition is controversial when there is no contact of
the articular surfaces on one side, and this condition is defined as a
dislocation, which in the English literature is called as dislocation.
When
unilateral atlantoaxial rotation is outside the normal range and the anterior
atlantodental distance increases, this type of injury is defined as the second
type. The anterior bilateral dislocation, caused by a rupture of the transverse
ligament, and the posterior one, which occurs more often with a fracture of the
odontoid process, are called as types 3 and 4, respectively. As a more severe
injury, types 3 and 4 are accompanied by spinal stenosis [4]. The most common
type is the first type, which is much more often recorded in children, while
the rest are much less common and require a more aggressive approach in
choosing treatment tactics [8]. Unlike children, in adults, post-traumatic AARF
occurs in high-energy trauma: traffic accidents or falls from a height [8, 14].
The likelihood
of fatal outcomes in adults is extremely rare, but higher than in pediatric
cases [8, 14]. The typical clinical picture of AARF is accompanied not only by
neck pain, but also by torticollis with a contralateral bending of the neck,
known as the Cock-Robin position [9, 18]. There are reports of atypical
clinical manifestations of AARF, manifested by dyscirculatory phenomena in the
basin of the vertebral arteries [6].
In the above
mentioned observation, an assessment of the totality of clinical manifestations
of injury, and data from a full range of diagnostic methods (MRI, MSCT,
angiography) made it possible to obtain a complete picture of the morphology of
the injuries that occurred. If the victims have a clinical picture of AARF,
diagnostic measures should be carried out urgently. It is impossible to allow a
decrease in the volume of this examination in patients with multiple and
combined injuries and diagnose only on the basis of clinical manifestations.
This leads to errors in the diagnosis and, consequently, to failures in the
treatment of these lesions [10,15]. Unfortunately,
there are no guides available for diagnosing these conditions. It should be
noted that CT scan data of the cervical spine are commonly used to assess bone
structures and determine the type of AARF. The side of the subluxation and the
amount of rotational displacement will be best seen on axial views, while
linear displacements in the facet joints are better visualized on sagittal
reconstructions [2]. In cases of instability at the C1-C2 level, MRI will be
most useful in assessing the ligamentous structures of the upper cervical
spine. At the same time, the main stabilizers of the atlanto-axial joint are
evaluated − the pterygoid and transverse ligaments [12, 19].
AARF is
usually accompanied by severe pain. Treatment should be aimed not only at its
elimination, but also at the restoration of normal anatomical relationships and
the prevention of neurological deficit or its aggravation. This is the main
goal of the chosen treatment strategy. The lack of direction of treatment from
the moment of injury, including the diagnostic period, leads to the appearance
of chronic cases that reduce the possibilities and effectiveness of
conservative treatment [8, 14]. The given clinical observation is not an
exception to this due to the delay of pathogenetically directed treatment. The
choice of method to eliminate AARF is still controversial due to its rare
occurrence, and an individual approach is preferred in each case [8, 14]. Early
diagnosis is of great importance in the timeliness of the treatment plan and
can increase the success rate of closed reduction, which reduces the need for
further surgical interventions [4]. In recent cases, closed reduction with or
without prior traction behind the head is preferred [9, 13, 14, 17, 20, 21,
22]. According to the literature, the duration of the injury is important for
performing reduction by traction [5, 9, 17, 18, 20-28]. A total of 18 cases of
such lesions in adults have been described.
Comparing the
data of these authors, it can be noted that if the elimination of AARF was
carried out up to 6 weeks after injury, then reduction was obtained in 64 % (7
patients out of 11). Moreover, two patients underwent reduction under
conditions of halofixation due to the inefficiency of traction for the head.
For the same reason, two patients underwent open reduction from the dorsal
approach with stabilization. All victims received a good long-term result [9,
14, 17, 20, 21, 22, 28]. Even if the dislocation is associated with a fracture
of the C2 vertebra, then traction is also preferred in the initial stages.
It should be
noted that a unilateral fracture of the C2 facet with displacement in the form
of an outward and anterior inclination can lead to the appearance of AARF. This
situation requires reduction under conditions of instrumental fixation [5, 27].
The initial absence of AARF in such non-displaced fractures does not exclude
its occurrence in the late period with external fixation with a collar as a
result of the displacement of a fragment of the C2 articular facet with the
appearance of limitation of movements, pain and torticollis [27].
With
post-traumatic AARF in adult patients more than 6 weeks, the possibility of its
elimination with the use of conservative treatment remains, which, as the
statute of limitations increases, loses its potential to achieve success.
Weisskopf M.
et al. (2005) described 26 cases of treatment of patients with AARF of various
etiologies with duration of at least 15 months, in five of which the cause was
trauma. The last one, due to the rigidity of the deformity, underwent two types
of surgical interventions: three - transoral fusion, two
- occipitospondylodesis. Long-term results of treatment did not differ and were
good. However, patients who underwent occipitospondylodesis noted a significant
deterioration in the function of the cervical spine [29].
Castel et al.
(2001) described a case of AARF in an athlete who sought help for torticollis
and neck pain 6 weeks after an injury while playing baseball. Long-term
traction for the head and staged manual reduction were performed. Subsequent
long-term physiotherapy and external immobilization ensured the patient's
recovery [30]. Traction under halofixation, used up to 6 weeks after injury, is
effective in eliminating AARF [9, 11, 17, 31]. More justified is the use of
this method of treatment in this pathology, combined with transfacet fractures
of C2 and its arches [5, 23, 25]. However, after the cessation of halotraction,
a longer time is required to restore the function of the cervical spine, with
an intermediate period of wearing a collar [31].
Surgical
treatment was necessary for patients due to the ineffectiveness of conservative
treatment of chronic AARF, when the time after injury was more than 6 weeks.
Five authors of various publications reported on the surgical treatment of 17
patients with AARF [10, 28, 29, 32, 33]. Among the methods of surgical
treatment, open reduction, atlanto-axial fixation, and spinal fusion prevailed
[10, 28, 33].
The
ineffectiveness of conservative treatment in the early period and the longevity
of the occurrence of post-traumatic AARF are not only the reasons for the need
for surgical treatment of this pathology. Qi et al. determined that not all
types of AARF can be described by Fielding's (1977) classification, since the
unilateral type of rotation sometimes leads to blockage at the border of the
C1-C2 facet joint of this side. In these cases, it is not possible to apply
effective traction and/or closed manual reduction even in the early period
after injury. Therefore, the authors believe that reduction can only be
achieved by staged surgical treatment from the refarengial approach, followed
by posterior C1-C2 fixation and fusion [33].
In our case,
clinical manifestations of incoming positional vestibular and neurological
disorders were recorded. A comprehensive examination revealed stenosis of the
spinal canal, deformation of the vertebral arteries without disturbing blood
flow. The key points in the choice of treatment tactics were also: the absence
of displacement of the fragments of the transfacet fracture and C2 arches, the
absence of damage to the transverse ligament of the atlas and pterygoid
ligaments, and the blockage described by Qi et al. (2015) [33]. Despite the
injury being 8 weeks old, the performed closed reduction ensured the
elimination of AARF. As shown by MSCT data of the cervical
spine, in the early period after reduction, rotation in the opposite direction
of the dislocation is not due to movements in C1-C2. Head rotation was provided
by movements in the segments of the lower cervical region. Undoubtedly,
physiotherapy started early after elimination of AARF played a positive role in
restoring the function of the atlanto-axial joint. Ultimately, the chosen
treatment strategy, which was based on an objective assessment of clinical and
radiographic examination data, allowed us to obtain a good result.
CONCLUSION
Diagnosis of
post-traumatic AARF should be carried out on the basis of a comprehensive
examination and at an earlier date, including in patients with concomitant
injuries, which makes it possible to successfully implement conservative
treatment. When AARF is combined with fractures of the C2 vertebra without displacement
of the fragments and without rupture of the main stabilizing ligaments of the
upper cervical region, it remains possible to avoid surgical treatment by
closed reduction even in the late stages after the injury. In the future,
physiotherapy is required to restore the function of this part of the spine.
If
conservative treatment is ineffective and surgical treatment of AARF is
necessary, preference should be given to methods that allow stopping pain,
eliminating the pathological position of the head, by restoring the anatomical
relationship of the atlas and axis, with instrumental stabilization. When
performing occipitospondylodesis, aggravation of the function of this part of
the spine is expected.
Funding and conflict of interest information
The study was
not sponsored.
The authors declare the absence of obvious and potential
conflicts of interest related to the publication of this article.
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