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Subluxacion atlantoaxial rotatoria traumatica en adultos reporte de dos casos y revision de la literatura.

Traumatic Atlantoaxial Rotatory Subluxation in Adults. Report of Two Cases and Literature Review.

Introduction

Atlantoaxial subluxation is defined as an instability of the atlas (C1) over the axis (C2), due to failure or rupture of the ligament complex in the C1-C2 joint. Rare in adults, several conditions associated with abnormalities in the atlantoaxial joint or ligament laxity course with a higher incidence of this entity, such as rheumatoid arthritis, Sfndrome de Down, Sfndrome de Marfan, Sfndrome de Morquio e Sfndrome de Grisel (1, 2). Down syndrome, Marfan syndrome, Morquio's syndrome and Grisel's syndrome (4,8).

In this study we describe two cases of atlantoaxial subluxation of traumatic origin in adults and review the literature regarding the main aspects of this entity.

Case 1

A female patient, 27 years old, was victim of an accidental fall to the ground while holding her son on her lap. The patient acquired a vicious posture with head rotation to the right. She showed functional limitation and pain when attempting to raise head in neutral position.

She presented with the mental state preserved without motor deficit, ASIA E. A computed tomography (CT) scan of the neck was performed and showed atlanto-axial rotatory dislocation type II according to Fielding and Hawkins (Figure 1A). The patient was submitted to conservative treatment with Gardner Wells skull traction for 12 days. After dislocations reduction, a halovest immobilization was maintained for 03 months. After that, she presented without deficits or pain.

Case 2

A 32 years old man was victim of aggression. On admission he presented an intense bleeding in the upper airway due to face fractures and nine points in Glasgow Coma Scale. After intubation for airway protection CT scan of the brain was performed and showed no abnormalities.

Fielding e Hawkins a direita (figuras 2 e 3) The cervical spine CT scan revealed rotator subluxation type I Fielding and Hawkins's classification (Figure 1B and 2A). The patient was immobilized with a Philadelphia neck collar during 15 days for deformity's reduction (Figure 2B). After this period, he presented without pain or neurologic deficit.

Discussion

The atlantoaxial rotatory subluxation is a rare clinical condition that may have traumatic or nontraumatic causes (5). It occurs most frequently in childhood, due to atlantoaxial ligament laxity, a common condition at this age group (5). The first classical description of this entity was made by Corner in 1907 (5). The main non-traumatic origins include rheumatoid arthritis, Down syndrome, Marfan syndrome, Morquio's syndrome, Grisel's syndrome, and after head and neck surgical manipulation (4,6,13). Atlantoaxial complex has morphological and functional properties that differ it from the rest of the cervical spine. The absence of intervertebral disc, the horizontal orientation of facets and the presence of complex ligament inserted into the odontoid are features which allow the joint and the creation of rotational motion. The rotational mobility of the cervical spine is approximately 90[degrees], a half occurs in the atlantoaxial joint. The transverse ligament acts as the main stabilizing factor, preventing the anterior-posterior displacement of the atlas on the axis. The alar ligaments are in number of two and its main function is to limit the axial rotation, the right alar ligament limits left rotation and vice versa (5,6,8,10).

[FIGURE 1 OMITTED]

[FIGURE 2 OMITTED]

The magnetic resonance imaging (MRI) findings suggest that the alar ligament injury is the most important cause for atlantoaxial subluxation (11,14). In 1977, a retrospective study of 17 patients conducted by Fielding and Hawkins (6) proposed a classification of this condition that is widely used currently in medical practice (4-8,10,11,13). The rotary subluxation is classified into four types, based on the evaluation of atlanto-dental interval (ADI) view through the lateral cervical radiography. The type I lesion, the most frequently seen, is characterized by rotation without subluxation. The atlantodental interval is less than 3 mm, which is considered the limit of the normal physiological motion. In Type II lesions, there is a shift of the atlas over the axis between 3 to 5 mm, which potentially indicates a failure of the primary stabilizing component: the transverse ligament. In type III lesions, there is an atlanto-dental interval greater than 5 mm, implying a complete rupture of the transverse ligament and the contralateral alar ligament.

The dislocation type IV is the less common type and corresponds to the displacement of the atlas over the axis. The latter lesion occurs in association with odontoid fractures or in patients with rheumatoid arthritis who have erosion of this structure. Fielding and Hawkins found that the atlanto-axial dislocation would occur at 65 degrees of rotation if the transverse ligament integrity is present. In case of injury of the transverse ligament associated with a displacement of 5 mm of the atlas over the axis, the shift will occur at 45 degrees of axial rotation (6).

The clinical features of this entity is the presence of upper cervical pain, limitation of neck mobility, torticollis and muscle spasm. The spasm is an attempt to restore the neutral position and correction of the deformity. The patient presents with the classic "Cock Robin's" position, which consisted of an axial rotation around 20-30 degrees, associated with lateral flexion of 20-30 degrees in the opposite direction of the rotation (6,8). The cervical spine radiographies are mandatory in suspected alterations of atlantoaxial lesion, in order to measure atlanto-dental interval and seek for changes in the lateral mass. The CT scan in coronal sections shows asymmetry of the lateral masses relation to the odontoid. The lateral mass superiorly dislocated, shall be shown larger and closer to the midline, while the contralateral lateral mass appears more distant and smaller in diameter. Another indirect sign is the misalignment of the spinal processes. The three-dimensional CT reconstruction, allows a more detailed assessment of the craniocervical junction in compared to x-ray, being used more frequently in the evaluation of the injuries affecting the cervical spine. The treatment of rotatory subluxation should be individualized because there is no evidence in the literature showing the superiority of a particular therapeutic proposal.

The main factors that should be considered in the decision are the presence of neurological deficit, the integrity of the transverse ligament, the presence of odontoide fracture and other associated injuries. In injuries type II and III, Fielding and Hawkins (6) propose C1-C2 arthrodesis after a failed skull traction and reduction. Crockard and Rogers in 1996 (3) recommend facetectomy followed by fixation C1-C2 in theses cases. Recently, Kim et al (10), proposed open reduction and fixation with C1-C2 transpedicular screw, mentioning some advantages over other techniques. In lesions types I and II, skull traction reduction followed by external immobilization or even with the use of Philadelphia collar showed good performance in some cases (8,9,12). Other authors consider that the lesions type I and II have considerable possibility of recurrence, thereby advocating the early C1-C2 fixation (7,10,11).

Conclusion

These cases presented lesions types I and II, and obtained a satisfactory outcome in both with the non-operative therapy. We took into account biomechanical principles, functional and mortality associated with the given method. Regarding the few studies demonstrating the superiority of certain method in the treatment of traumatic rotatory subluxation, a carefull individulalized evaluation of each case is probably the most effective way to achieve therapeutic success.

Recibido: 07.10.11

Aceptado: 07.12.11

References

(1.) Born CT, Mure AJ. Born CT, Mure AJ, Iannacone WM, DeLong WG Jr: Three dimensional computerized tomographic demonstration of bilateral atlantoaxial rotatory dislocation in an adult. Three-dimensional computerized tomographic demonstration of bilateral atlantoaxial rotatory dislocation in an adult: report of a case and review of the literature. J Orthop Trauma 1994;8:67-72. J Orthop Trauma 1994; 8 (1): 67-72.

(2.) Bouillot P, Fuentes S. Bouillot P, Fuentes S, Dufour H, Manera L, Grisoli F:Imaging features in combined atlantoaxial and occipitoatlantal rotatory subluxation. J Neurosurg 1999;90:258-60. Imaging features in combined atlantoaxial and occipitoatlantal rotatory subluxation: a rare entity. Case report. J Neurosurg 1999; 90 (2 Suppl): 258-260.

(3.) Crockard HA, Rogers MA. Crockard HA, Rogers MA:Open reduction of traumatic atlantoaxial rotatory dislocation with the use of extreme lateral approach. J Bone Joint Surg Am, 1986;78:431-6 Open reduction of traumatic atlanto-axial rotatory dislocation with use of extreme lateral approach. A report of two cases. J Bone Joint Surg Am 1996; 78 (3): 431-436.

(4.) Crook TB, Eynon CA. Crook TB, Eynon CA: Traumatic atlantoaxial rotatory subluxation. Emerg Med J 2005; 22: 671- 672. Traumatic atlantoaxial rotatory subluxation. Emerg Med J 2005; 22 (9): 671-672.

(5.) El-Khoury GY, Clark CR Gravett AW. El-Khoury GY, Clark CR, Gravett AW: Acute traumatic rotatory atlanto-axial dislocation in children. Acute traumatic rotatory atlanto-axial dislocation in children. A report of three cases. J Bone Joint Surg Am 1984;66:774-7. A report of three cases. J Bone Joint Surg Am 1984; 66 (5): 774-777.

(6.) Fielding JW, Hawkins RJ. Fielding JW, Hawkins RJ: Atlantoaxial rotatory fixation. J Bone Joint Surg Am 1977;59A:37-44. Atlantoaxial rotatory fixation. J Bone Joint Surg Am 1977; 59-A: 37-44.

(7.) Fuentes S, Bouillot P, Palombi O, Ducolombier A, Desgeorges M: Traumatic atlantoaxial rotatory dislocation with odontoid fracture: Case report and review. Spine 2001;26:830-834. Fuentes S, Bouillot P, Palombi O, Ducolombier A, Desgeorges M: Traumatic atlantoaxial rotatory dislocation with odontoid fracture: Case report and review. Spine 2001; 26 (7): 830-834.

(8.) Haliasos N, NorrisHaliasos N, Norris J: Conservative management of a traumatic unilateral rotatory atalntoaxial subluxation in na adult: Case report and review. Injury Extra 2007; 38: 381-391. Conservative management of a traumatic atlantoaxial unilateral rotatory subluxation in the adult: Case report and review. Injury Extra 2007; 38: 381-391.

(9.) Johnson DP, Fergusson CM. Early diagnosis of atlantoaxial rotatory fixation. J Bone Joint Surg Br 1986;68:698-701. Johnson DP, Fergusson CM: Early diagnosis of atlanto-axial rotatory fixation. J Bone Joint Surg Br 1986; 68 (5): 698-701.

(10.) Kim YS, Lee JK, Kim JH, Kim SH. Kim YS, Lee JK, Kim JH, Kim SH:Post-traumatic atlantoaxial rotatory dislocation in an adult treated by open reduction and C1-C2 transpedicular screw fixation. J Korean Neurosurg Soc 2007;41: 248-251. Post-traumatic atlantoaxial rotatory dislocation in an adult treated by open reduction and C1-C2 transpedicular screw fixation. J Korean Neurosurg Soc 2007; 41: 248-251.

(11.) Niibayashi H. Atlantoaxial rotatory subluxation. Niibayashi H: Atlantoaxial rotatory subluxation. A case report. Spine 1998;23:1494-6. A case report. Spine 1998; 23 (13): 1494-1496.

(12.) Robertson PA, Swan H. Robertson PA, Swan H: Traumatic bilateral rotatory facet dislocation of the atlas on the axis. Spine 1992;17:12524. Traumatic bilateral rotatory facet dislocation of the atlas on the axis. Spine 192; 17 (10): 1252-1254.

(13.) Rocha R, et al. Atlantoaxial rotatory subluxation with ligamentous disruption: A biomechanical comparison of current fusion methods. Neurosurgery 2009;64:137-144. Rocha R, Sawa AG, Baek S, et al: Atlantoaxial rotatory subluxation with ligamentous disruption: A biomechanical comparison of current fusion methods. Neurosurgery 2009; 64 (3 Suppl): 137-144.

(14.) White AA, Panjabi MM. Willauschus WG, Kladny B, Beyer WF. Willauschus WG, Kladny B, Beyer WF, Gluckert K, Arnold H, Scheithauer R: Lesions of the alar ligaments in vivo and in vitro studies with magnetc resonance imaging. Spine 1995;20:2493-8. Lesions of the alar ligaments. In vivo and in vitro studies with magnetic resonance imaging. Spine 1995; 20 (23): 2493-2498.

Corresponding author:

Guilherme Brasileiro de Aguiar

Rua Abflio Soares, 121--84/Parafso.

04005-000. Sao Paulo--SP, Brazil

Email: guilhermebraguiar@yahoo.com.br

Joao Luiz Vitorino Araujo *, Guilherme Brasileiro de Aguiar *, Luciano Haddad *, Jefferson Walter Daniel *, Jose Carlos Esteves Veiga **

* Department of Surgery, Division of Neurosurgery, Santa Casa Medical School, Sao Paulo, Brazil.

** Chief of the Division of Neurosurgery, Santa Casa Medical School, Sao Paulo, Brazil.
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Title Annotation:reporte de casos
Author:Vitorino Araujo, Joao Luiz; Brasileiro de Aguiar, Guilherme; Haddad, Luciano; Walter Daniel, Jeffers
Publication:Revista Chilena de Neurocirugia
Date:Jan 1, 2012
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