Traumatic Chronic Irreducible Atlantoaxial Rotatory Fixation in Adults: Review of the Literature, With Two New Examples

Abstract

Atlantoaxial rotatory fixation (AARF) is a rare condition in adults and is almost always due to an accompanying trauma. The first example of traumatic AARF in adults was reported by Corner in 1907 and since then only 55 adult cases with this rare traumatic scenario have been published so far. Approximately 80% of adults with traumatic AARF are diagnosed soon after the traumatic events. However, in the remaining casualties, the condition might be missed with some delay from the diagnosis to treatment. If this pathology is diagnosed early enough, the conservative attempts for a closed reduction are usually effective. After closed reduction, external immobilization is required to prevent recurrence of the dislocation. However, with delayed diagnosis, the condition may remain refractory to traction on manipulation and require an open reduction instead. In the literature, such irreducible chronic AARFs are rarely reported, being confined to only 14 adult examples, in whom surgical intervention for correction of the deformity will be required. In such cases, release of the atlantoaxial facet joints is the first surgical step. In the subsequent step, reduction of the dislocated facet joints can be done via one of the already described maneuvers. As the final step, C1-C2 fixation will be necessary for prevention of re-dislocation. Herein, 2 adult patients with chronic rotatory atlantoaxial dislocation of traumatic origin are presented. In both cases, cranial traction and manipulations were ineffective and therefore an open reduction procedure was proposed and accomplished via the posterior midline corridor. The transverse rod technique was implemented subsequent to the atlantoaxial facet release. After correction of the deformity, a C1-C2 fixation was accomplished followed by arthrodesis. In addition to the outlined procedure, an historical review of the literature on this subject from the beginning of 20th century is demonstrated.

Atlantoaxial rotatory subluxation presenting as acute torticollis after mild trauma

“…Treatment strategies for a case diagnosed early generally involve a closed reduction and immobilization with a halo-ring or cervical collar. For cases refractory to this treatment, as is common in chronic or recurrent cases with a delayed diagnosis, surgery may be indicated [2 , 5] .…”

“…Cervical spine injuries occur in 2.4% of blunt traumas [1] . Atlantoaxial rotatory subluxation (AARS) is a rare outcome of trauma in adults [2] . As reported by Rahimizadeh et al in 2010, in the 20th century, there were only 57 adults with reported cases of atlantoaxial rotatory fixation, a term that encompasses rotatory displacement arising from either subluxation or dislocation [2] .…”

“…Atlantoaxial rotatory subluxation (AARS) is a rare outcome of trauma in adults [2] . As reported by Rahimizadeh et al in 2010, in the 20th century, there were only 57 adults with reported cases of atlantoaxial rotatory fixation, a term that encompasses rotatory displacement arising from either subluxation or dislocation [2] . In the pediatric population, AARS is more common and can be spontaneous or seen in trauma, particularly with congenital malformation; 1 multicenter cohort showed that of children with blunt trauma related cervical spine injury, AARS accounted for 10% of injuries [3] .…”

Atlantoaxial rotatory fixation in childhood: a staged management strategy incorporating manipulation under anaesthesia

“…Atlantoaxial rotatory fixation (AARF) is an acquired fixed rotation abnormality of the C1 vertebra on C2 that impedes turning of the neck and typically manifests with painful torticollis. It is primarily a condition of childhood although adolescent and adult cases are seen [ 1 – 3 ]. Clinical diagnosis of AARF is suspected on the basis of a fixed rotational deformity of the neck, often with the typical “Cock-Robin” position created by cervical lateral flexion, forward flexion and rotation [ 1 , 4 , 5 ].…”

Systematic review of the nonsurgical management of atlantoaxial rotatory fixation in childhood

“…It is typically seen in childhood, with adolescent and adult cases occurring rarely. [1][2][3] Clinical diagnosis of AARF is based on fixed rotational deformity of the cervical spine with the head held in a "cock-robin" position with rotation and forward and lateral flexion. 1,4,5 Without treatment, the atlantoaxial joint is at risk of fusion in the abnormal position, which can cause cosmetic effects, facial asymmetry, functional disability, chronic pain, and compensatory deformity in the subaxial spine.…”