Cervical juxtafacet cyst

Cervical juxtafacet cyst

A literature review till 2010 described only 28 symptomatic cervical synovial cyst cases 1)

A literature review till 2013 identified 35 studies with 89 previously reported cases of surgically treated subaxial juxtafacet cysts (JFCs) 2).

Attwell et al. presented an unusual case of acute symptomatology secondary to spontaneous haemorrhage into a cervical facet joint cyst 3)

Sasamori et al. report a case of cervical juxtafacet cyst with extensive rim enhancement on magnetic resonance imaging 4).

see atlantoaxial juxtafacet cyst

Juxtafacet cysts (JFCs) seem to be a degenerative change of the cervical spine rather than a traumatic event. Similar to their counterparts in the lumbar spine, they tend to arise in segments with increased mobility.

Sivakumar et al. have reported on the development of JFCs adjacent to anterior cervical fusion constructs, and consideration of JFCs as a form of adjacent level disease (ALD) has been hypothesized 5).

Moon et al. reported one patient that developed a C5/6 JFC 20 months after C4/5 anterior fusion and C5/6 anterior foraminotomy. In this case, despite progressive subluxation at C5/6 and solid C4/5 fusion demonstrated on flexion films 20 months after the original surgery, the patient underwent partial hemilaminectomy alone for cyst decompression. Outcome was favorable at 4 months follow-up 6)

Trauma

After a cervical spine fracture and, hence, was probably related to trauma. Surgical therapy resulted in a satisfactory recovery 7).

Chronic expansion of the extradural mass may lead to compression of the nerve root, thecal sac, or both, and may follow long periods of axial back pain without neurological deficit 8).

They are rare causes of neurological deficits. Their imaging characteristics, relationship to segmental instability, and potential for inducing acute symptomatic deterioration have only been described in a few case reports and small case series 9).

Less commonly, neurological deterioration has been attributed to rapid cystic growth with hemorrhage 10) 11)

Attwell et al., reported acute symptomatology secondary to spontaneous haemorrhage into a cervical facet joint cyst 12).

Combination with discal herniation and spina bifida occulta was diagnosed with computed tomography (CT) and magnetic resonance imaging (MRI) in one case 13).

Myelopathy

In the series of Christophis all cervico-thoracic or thoracic cysts presented myelopathy 14).

Till 1999 there have been only two previously reported cases of subaxial degenerative synovial cysts of the cervical spine in patients who presented with a clinical picture of spinal cord compression. Cudlip et al. report three additional patients treated for degenerative cervical synovial cysts who presented with myelopathy. In all three patients the cyst was successfully excised and a good clinical outcome achieved 15).

Cho el al. describe a case of an 80-year-old man with a gradual weakness of the lower extremities not linked to any known traumatic episode over the 2 weeks before admission. CT scan and MRI of the spine revealed a cystic formation, measuring about 1 cm in diameter, at C7-T1 at the left posterolateral site at the level of the articular facet. During surgery, the mass appeared to be in the ligamentum flavum at the level of the articular facet and was in contact with the dura mater. After the removal of the mass, there was an immediate and significant improvement of the patient’s symptoms. Histopathologic examination showed the cyst to be composed of nonspecific degenerative fibrous tissue with mild inflammatory change and confirmed the cyst as a synovial cyst. Synovial cyst in the cervical region is a very rare lesion causing myelopathy. Surgical removal of the cyst and decompression of the spinal cord results in good neurological recovery 16).

Brown-Sequard syndrome

Cheng et al. published a rare case of a patient with a ganglion cyst of the lower cervical spine presenting with acute Brown-Sequard syndrome. The patient had no history of trauma. Magnetic resonance imaging of the cervical spine showed a cystic lesion connecting to the synovial joint C6-7 and compressing the posterior aspect of the spinal cord. The patient underwent emergent C6-7 laminectomy with total removal of the cyst. Neurological function recovered completely 4 months after operation 17).

Magnetic resonance imaging reveal an intraspinal extradural cystic lesion in contact with the facet joint. The spinal cord can severely compressed by this lesion which is hypointense on T1-weighted imaging and hyperintense on T2-weighted imaging and short T1 inversion recovery. The cyst wall can strongly enhance after contrast injection 18).

Sasamori et al. report a case of cervical juxtafacet cyst with extensive rim enhancement on gadolinium-diethylenetriamine pentaacid magnetic resonance imaging.

Operative finding revealed the epidural space around the mass filled with abundant venous plexus. Histological examination demonstrated that cyst wall was composed of the well-vascularized fibrous connective tissue with some inflammatory changes. They speculate that extensive rim enhancement of juxtafacet cyst may be attributed not only to the chronic inflammatory changes of cyst wall, but to engorged venous plexus within the widened epidural space 19)

Surgical treatment is effective 20).

Colen and Rengachary report a spontaneous resolution of a cervical synovial cyst 21)

Technique

The head is positioned in Mayfield pins under gentle capital flexion, and the patient was positioned prone on gel rolls. Dissection proceeded in the subperiosteal plane, either unilaterally (e.g., hemilaminectomy) or bilaterally, depending on the goals of the decompression and the extent of spinal canal compromise. During resection of the lesion, the lateral facet and capsule were preserved as much as possible. When deemed necessary for complete decompression or visualization of the lesion, the laminectomy was extended to include a conservative medial facetectomy on the affected side.

The putative medial facet joint is carefully cauterized to minimize risk of cyst regrowth.

Instrumentation and fusion can be performed at the discretion of the operating surgeon. Loss of cervical lordosis, spondylolisthesis, hypermobility, index level neck pain, and iatrogenic instability following decompression are each relative indications for fusion.

Fixation can be accomplished using bilateral lateral mass/pedicle screw and rod constructs. Fusion can be augmented with morselized local autograft, with or without bone allograft.

12 consecutive patients (mean age 63.4 years, range 52-83 years) harboring 14 JFCs treated across 9 years was retrospectively reviewed. Clinical history, neurological status, preoperative imaging, operative findings, pathology, and postoperative outcomes were obtained from medical records. The mean follow up was 9.2 ± 7.8 months.

Most JFCs in this series involved the C7/T1 level. Nine patients reported axial neck pain, 12 patients had radicular symptoms, four patients had myelopathy, and one patient experienced rapid neurological decline attributable to cystic hemorrhage. Cyst expansion without hemorrhage caused subacute deterioration in one patient. All patients experienced sensory and/or motor improvement following surgical decompression. Preoperative axial neck pain improved in eight of nine patients (89 %). Seven out of 12 patients (58 %) underwent fusion either at the time of decompression (six patients) or at a delayed timepoint within the follow-up period (one patient). Prior history of cervical instrumentation, hypermobility on dynamic imaging, and other risk factors for segmental instability were more common in this series than in previous reports 22).

13 patients with synovial or ganglion cysts of the spinal facet joints causing nerve root compression. These cysts were found in both the cervical and the lumbar spine, and the anatomical location of each cyst corresponded to the patient’s signs and symptoms. In no case was there evidence of intervertebral disc abnormality found at operation. The patients ranged from 49 to 77 years of age and included 4 men and 9 women. Radiographic evidence of facet degenerative change and degenerative spondylolisthesis was frequently but not invariably noted. The extradural defects defined with positive contrast myelography or postmyelography computed tomographic scanning were usually posterior or posterolateral to the common dural sac and were misinterpreted as extruded discs in the majority of cases. Treatment consisted of laminectomy and surgical excision of cysts. All patients reported improvement or resolution of their presenting symptoms 23).

Chun et al. described an interesting case of cervical juxtafacet that developed outside the intervertebral foramen, compressing the cervical medial branch and causing neuropathic pain in the posterior inferior neck pain. A 61-year-old woman visited a local pain clinic due to neuropathic pain with a tingling and burning nature (numeric rating scale [NRS]: 5 out of 10) on the left posterior inferior neck area for 4 months. Paresthesia was observed in the left posterior inferior neck area. On cervical radiography, segmental instability was observed at the C3-4 and C4-5 levels. Moreover, on the magnetic resonance imaging (MRI) of the cervical spine, a cyst (size: 1.3 cm × 0.7 cm × 1 cm) was outside the intervertebral foramen, contacting the left C4-5 facet joint and left C5 articular pillar. We thought that compression of the left C5 medial branch by the cyst could cause the patient’s pain. We conducted computed tomography (CT)-guided percutaneous needle aspiration of a cervical juxtafacet cyst. An 18-gauge needle was advanced under the guidance of CT into the largest portion of the cyst through a posterolateral oblique approach. Gelatinous mucoid fluid (approximately 0.5 cc) was aspirated. Immediately after the aspiration, 80% of the patient’s pain was disappeared, and dysesthesia was completely disappeared. At the 1-, 3-, and 6-month follow-ups, the patient reported slight pain (NRS: 1) on the left posterior inferior neck. Cervical juxtafacet cysts can develop outside of the intervertebral foramen and spinal canal. Percutaneous needle aspiration can be a useful therapeutic tool for the treatment of such cysts 24)


Third reported case of a degenerative articular cyst of the upper cervical spine, involving the quadrate ligament of the odontoid process. Magnetic resonance examination reveals typical images. A new, more general terminology is proposed 25).


1)

Costa F, Menghetti C, Cardia A, Fornari M, Ortolina A. Cervical synovial cyst: case report and review of literature. Eur Spine J. 2010 Jul;19 Suppl 2:S100-2. doi: 10.1007/s00586-009-1094-6. Epub 2009 Jul 15. Review. PubMed PMID: 19603197; PubMed Central PMCID: PMC2899642.
2) , 9) , 22)

Uschold T, Panchmatia J, Fusco DJ, Abla AA, Porter RW, Theodore N. Subaxial cervical juxtafacet cysts: single institution surgical experience and literature review. Acta Neurochir (Wien). 2013 Feb;155(2):299-308. doi: 10.1007/s00701-012-1549-0. Epub 2012 Nov 17. Review. PubMed PMID: 23160630.
3) , 12)

Attwell L, Elwell VA, Meir A. Cervical synovial cyst. Br J Neurosurg. 2014 Dec;28(6):813-4. doi: 10.3109/02688697.2014.913782. Epub 2014 May 6. PubMed PMID: 24801806.
4) , 19)

Sasamori T, Hida K, Anzai K, Yano S, Kato Y, Tanaka S, Saito H, Houkin K. A case of cervical juxtafacet cyst with extensive rim enhancement on Gd-DTPA MRI. Clin Imaging. 2014 Mar-Apr;38(2):199-201. doi: 0.1016/j.clinimag.2013.10.002. Epub 2013 Nov 7. PubMed PMID: 24332973.
5)

Sivakumar W, Elder JB, Bilsky MH. Cervical juxtafacet cyst after anterior cervical discectomy and fusion. Neurosurg Focus. 2011 Oct;31(4):E19. doi: 10.3171/2011.8.FOCUS11119. Review. PubMed PMID: 21961863.
6)

Moon HJ, Kim JH, Kim JH, Kwon TH, Chung HS, Park YK. Cervical juxtafacet cyst with myelopathy due to postoperative instability. Case report. Neurol Med Chir (Tokyo). 2010;50(12):1129-31. PubMed PMID: 21206195.
7)

Cartwright MJ, Nehls DG, Carrion CA, Spetzler RF. Synovial cyst of a cervical facet joint: case report. Neurosurgery. 1985 Jun;16(6):850-2. PubMed PMID: 4010912.
8)

Boviatsis EJ, Stavrinou LC, Kouyialis AT, Gavra MM, Stavrinou PC, Themistokleous M, Selviaridis P, Sakas DE (2008) Spinal synovial cysts: pathogenesis, diagnosis and surgical treatment in a series of seven cases and literature review. Eur Spine J 17:831– 837
10)

Akhaddar A, Qamouss O, Belhachmi A, Elasri A, Okacha N, Elmostarchid B, Boucetta M (2008) Cervico-thoracic juxtafacet cyst causing spinal foraminal widening. Joint Bone Spine 75:747–749
11)

Jabre A, Shahbabian S, Keller JT (1987) Synovial cyst of the cervical spine. Neurosurgery 20:316–318
13)

Vastagh I, Palásti A, Nagy H, Veres R, Bálint K, Karlinger K, Várallyay G. Cervical juxtafacet cyst combined with spinal dysraphism. Clin Imaging. 2008 Sep-Oct;32(5):387-9. doi: 10.1016/j.clinimag.2008.02.034. PubMed PMID: 18760727.
14)

Christophis P, Asamoto S, Kuchelmeister K, Schachenmayr W. “Juxtafacet cysts”, a misleading name for cystic formations of mobile spine (CYFMOS). Eur Spine J. 2007 Sep;16(9):1499-505. Epub 2007 Jan 4. PubMed PMID: 17203271; PubMed Central
15)

Cudlip S, Johnston F, Marsh H. Subaxial cervical synovial cyst presenting with myelopathy. Report of three cases. J Neurosurg. 1999 Jan;90(1 Suppl):141-4.Review. PubMed PMID: 10413141.
16)

Cho BY, Zhang HY, Kim HS. Synovial cyst in the cervical region causing severe myelopathy. Yonsei Med J. 2004 Jun 30;45(3):539-42. PubMed PMID: 15227744.
17)

Cheng WY, Shen CC, Wen MC. Ganglion cyst of the cervical spine presenting with Brown-Sequard syndrome. J Clin Neurosci. 2006 Dec;13(10):1041-5. PubMed PMID:17113987.
18)

Cheng YY, Chen CC, Yang MS, Hung HC, Lee SK. Intraspinal extradural ganglion cyst of the cervical spine. J Formos Med Assoc. 2004 Mar;103(3):230-3. PubMed PMID: 15124052.
20)

Krauss WE, Atkinson JL, Miller GM. Juxtafacet cysts of the cervical spine.Neurosurgery. 1998 Dec;43(6):1363-8. Review. PubMed PMID: 9848850.
21)

Colen CB, Rengachary S. Spontaneous resolution of a cervical synovial cyst. Case illustration. J Neurosurg Spine. 2006 Feb;4(2):186. PubMed PMID: 16506489.
23)

Onofrio BM, Mih AD. Synovial cysts of the spine. Neurosurgery. 1988 Apr;22(4):642-7. PubMed PMID: 3374775.
24)

Chun YM, Boudier-Revéret M, Lee SH, Chang MC. Neuropathic Pain due to Compression of Cervical Medial Branch by Cervical Juxtafacet Cyst: A Case Report. Pain Pract. 2022 May 24. doi: 10.1111/papr.13129. Epub ahead of print. PMID: 35607892.
25)

Goffin J, Wilms G, Plets C, Bruneel B, Casselman J. Synovial cyst at the C1-C2 junction. Neurosurgery. 1992 Jun;30(6):914-6. PubMed PMID: 1614595.

Aggressive vertebral hemangioma

Aggressive vertebral hemangioma

Accurate preoperative diagnosis is essential because they are highly vascular with a high tendency for intraoperative bleeding.

Management of aggressive VHs involves pre-op embolization, spinal surgery, and reconstruction. Pain management, physical rehabilitation, and close neurological follow-up are imperative to near-total recovery 1).

Surgery is required in cases of rapid or progressive neurological symptoms like compressive myelopathy or radiculopathy.

Vertebral hemangioma resection can be a real challenge for spine surgeons, given the high potential of massive intraoperative bleeding. For this reason, preoperative transarterial embolization of this tumor is supported by the available literature 2).


A navigation-guided drill is highly helpful for real-time monitoring of ongoing tumor resection. It enables safely resection of the tumor, especially in the anterior cortical surface of the vertebral body, and easily resections even hard tumors. This method results in reducing residual tumors and maintaining safe resection 3).

Radiotherapy can be used in patients with slowly progressive neurological deficits.

While CT-guided direct alcohol injection is effective in the management of symptomatic and aggressive vertebral hemangiomas, spinal angiography and trans-arterial embolization of the blood supply to the vertebral body hemangioma, prior to the direct transpedicular alcohol embolization of the lesion, improves the safety of the procedure 4).

Other emerging options in cases of aggressive hemangiomas include radiofrequency ablation with a hemostatic agent (e.g. FLOSEAL, Baxter, USA), and bone autograft placement 5).


Minimally invasive procedures may be successful in smaller lesions 6).

The case of a pregnancy who was diagnosed with an aggressive vertebral hemangioma that further led to progressive paraparesis. We had to take the fact that she was pregnant into account in the diagnostic procedure, the choice of examination method, and also the method of therapy. The goal of this case report is threefold: (1) provide an overview of the possible methods of management, specifically imaging, which will aid in diagnosis and based on that, (2) determine the appropriate therapy, and (3) review the risks and benefits of each will be presented when choosing individual approaches 7).


1)

Goraya GS, Singhal S, Paul BS, Paul G. Aggressive Vertebral Hemangioma: The Mystery of Spastic Legs Unveiled by a Purple Shoulder. Cureus. 2022 Jan 24;14(1):e21568. doi: 10.7759/cureus.21568. PMID: 35228927; PMCID: PMC8873442.
2)

Fiore G, Bertani GA, Tariciotti L, Borsa S, Paolucci A, Taramasso L, Locatelli M, Pluderi M. Vertebral Body Infarction after Transarterial Preoperative Embolization of a Vertebral Hemangioma. J Neurol Surg A Cent Eur Neurosurg. 2021 Dec 12. doi: 10.1055/s-0041-1739215. Epub ahead of print. PMID: 34897610.
3)

Nagashima Y, Nishimura Y, Haimoto S, Eguchi K, Awaya T, Ando R, Akahori S, Hara M, Natsume A. Piecemeal resection of aggressive vertebral hemangioma using real-time navigation-guided drilling technique. Nagoya J Med Sci. 2021 Nov;83(4):861-868. doi: 10.18999/nagjms.83.4.861. PMID: 34916728; PMCID: PMC8648519.
4)

Srinivasan G, Moses V, Padmanabhan A, Ahmed M, Keshava SN, Krishnan V, Joseph BV, Raju KP, Rajshekhar V. Utility of spinal angiography and arterial embolization in patients undergoing CT guided alcohol injection of aggressive vertebral hemangiomas. Neuroradiology. 2021 Nov;63(11):1935-1945. doi: 10.1007/s00234-021-02788-7. Epub 2021 Aug 24. PMID: 34427707.
5)

Canbay S, Kayalar AE, Gel G, Sabuncuoğlu H. A novel surgical technique for aggressive vertebral hemangiomas. (2019) Neurocirugia (Asturias, Spain). 30 (5): 233-237. doi:10.1016/j.neucir.2018.08.003
6)

Vasudeva VS, Chi JH, Groff MW. Surgical treatment of aggressive vertebral hemangiomas. Neurosurg Focus. 2016 Aug;41(2):E7. doi: 10.3171/2016.5.FOCUS16169. PMID: 27476849.
7)

Ridzoňová L, Fedičová M, Andráš T, Urdzík P, Gdovinová Z. Lower-limb progressive paraparesis management and diagnosis overview in a pregnant woman with vertebral haemangioma. Womens Health (Lond). 2022 Jan-Dec;18:17455057221099018. doi: 10.1177/17455057221099018. PMID: 35574823.

Atlantoaxial rotatory subluxation

Atlantoaxial rotatory subluxation

● typically seen in children

● associations: trauma, RA, respiratory tract infections in peds (Grisel syndrome)

● often present with cock-robin head position (tilt, rotation, sl. flexion)

● Tx: early traction often successful. Treat infection in Grisel syndrome. Subluxation unreducible in traction may need transoral release then posterior fusion.


Rotational deformity at the atlanto-axial junction is usually of short duration and easily corrected. Rarely, the atlantoaxial joint locks in rotation (AKA atlantoaxial rotatory fixation 1)

Usually seen in children. May occur spontaneously (with rheumatoid arthritis 2) or with congenital dens anomalies), following major or minor trauma (including neck manipulation or even with neck rotation while yawning 3), or with an infection of the head or neck including upper respiratory tract (known as Grisel syndrome 4): inflammation may cause mechanical and chemical injury to the facet capsules and/or Transverse Ligament of the Atlas.

Atlantoaxial rotatory fixation (AARF) is a rare condition that occurs most commonly in children.

Could occur due to congenital bony malformation, minor trauma, upper respiratory tract infections (Grisel’s syndrome), postoperatively after head and neck (ENT) surgery, and unknown reasons. AARF in the postoperative patient is a rare and poorly understood entity.

A serious and often unrecognized complication of rheumatoid arthritis or ankylosing spondylitis.

The dislocation may be at the occipito-atlantal and/or the atlanto-axial articulations 5). The mechanism of the irreducibility is poorly understood. With an intact TAL, rotation occurs without anterior displacement. If the TAL is incompetent as a result of trauma or infection, there may also be anterior displacement with more potential for neurologic injury. Posterior displacement occurs only rarely 6)

Fielding & Hawkins

type I: the atlas is rotated on the odontoid with no anterior displacement

type II: the atlas is rotated on one lateral articular process with 3 to 5 mm of anterior displacement

type III: comprises a rotation of the atlas on both lateral articular processes with anterior displacement greater than 5 mm

type IV: characterised by rotation and posterior displacement of the atlas vertical subluxation

The neurologic deficit is rare. Findings may include: neck painheadachetorticollis—characteristic “cock robin” head position with ≈ 20° lateral tilt to one side, 20° rotation to the other, and slight (≈ 10°) flexion, reduced range of motion, and facial flattening.

The torticollis caused usually presents as abnormal posturing of the head and neck, with rotation of the chin to the opposite side.

Although the patient cannot reduce the dislocation, they can increase it with head rotation towards the subluxated joint with potential injury to the high cervical cord.

Brainstem and cerebellar infarction and even death may occur with the compromise of circulation through the VAs 7).

Pediatric emergency physicians must have a high clinical suspicion for atlantoaxial rotatory subluxation (AARS), particularly when a child presents with neck pain and an abnormal head posture without the ability to return to a neutral position. As shown in the neurosurgical literature, timely diagnosis and swift initiation of treatment have a greater chance of treatment success for the patient. However, timely treatment is complicated because torticollis can result from a variety of maladies, including: congenital abnormalities involving the C1-C2 joint or the surrounding supporting muscles and ligaments, central nervous system abnormalities, obstetric palsies from brachial plexus injury, clavicle fractures, head and neck surgery, and infection. The treating pediatrician must discern the etiology of the underlying problem to determine both timing and treatment paradigms, which vary widely between these illnesses.

Kinon et al., present a comprehensive review of AARS that is intended for pediatric emergency physicians. Management of AARS can vary widely bases on factors, such as duration of symptoms, as well as the patient’s history. The goal of this review is to streamline the management paradigms and provide an inclusive review for pediatric emergency first responders 8).

X-rays : Findings (may be confusing) include:

● pathognomonic finding on AP C-spine X-ray in severe cases: frontal projection of C2 with simultaneous oblique projection of C1. In less severe cases, the C1 lateral mass that is forward appears larger and closer to the midline than the other

● asymmetry of the atlantoaxial joint that is not correctable with head rotation, which may be demonstrated by persistence of asymmetry on open mouth odontoid views with the head in neutral position and then rotated 10–15° to each side

● the spinous process of the axis is tilted in one direction and rotated to the other (may occur in torticollis of any etiology)

CT scan: Demonstrates rotation of the atlas.

MRI: May assess the competence of the transverse ligament.

Deformity of the superior facet of the axis (C2) and >20° of lateral inclination of the atlas observed on 3D CT are known factors in the progression to intractable 9).

The vertebral artery (VA) may be compromised in excessive rotation, especially if it is combined with anterior displacement.

Atlanto-occipital rotatory subluxation and facet deformity in the atlanto-occipital joint may occur after prolonged Atlantoaxial rotatory fixation . It is necessary to pay attention to pathological changes not only in the atlantoaxial joint but also in the atlanto-occipital joint, when treating patients with Atlantoaxial rotatory fixation 10).

In seventeen cases of irreducible atlanto-axial rotatory subluxation (here called fixation), the striking features were the delay in diagnosis and the persistent clinical and roentgenographic deformities. All patients had torticollis and restricted, often painful neck motion, and seven young patients with long-standing deformity had flattening on one side of the face. The diagnosis was suggested by the plain roentgenograms and tomograms and confirmed by persistence of the deformity as demonstrated by cineroentgenography. Treatment included skull traction, followed by atlanto-axial arthrodesis if necessary. Of the thirteen patients treated by atlanto-axial arthrodesis, eleven had good results, one had a fair result, and one had not been followed for long enough to determine the result. Of the remaining four patients, one treated conservatively had not been followed for long enough to evaluate the result, two declined surgery, and one died while in traction as the result of cord transection produced by further rotation of the atlas on the axis despite the traction 11).


Atlantoaxial rotational fixation (AARF) is a rare entity in adults, with only a few cases reported in the English literature and often associated with a traumatic mechanism. It is an underdiagnosed condition that must be taken into account in the initial assessment of all craniocervical trauma. Both diagnostic and therapeutic delay may be a potential cause of severe neurological damage or even death of the patient. The therapeutic management is controversial given the difficulty of achieving optimum stability and permanent reduction.

The atlantoaxial rotatory fixation (AARF) and the atlantoaxial rotatory subluxation (AARS) are the most frequent manifestations of atlantoaxial rotatory dislocation (AARD) in children, and conservative treatment has proved to be suitable in many cases, considering the pathological features of these type of injuries. In literature, there is no agreement on the treatment modalities and the timing of conservative treatment.


A 28-year-old woman was involved in a traffic accident a week before coming to the emergency with rotation and irreducible cervical flexion from trauma and severe neck pain. CT and MRI column were performed and showed a cervical spinal AARF with transverse and alar ligaments intact and preserved atlantoaxial distance (Fielding I). The patient was treated by progressive cervical traction with 5 kg and manual reduction was completed in 24 h. Subsequently, an external immobilization was performed by cervical rigid collar for 16 weeks. The clinical course was good, with the patient regaining full mobility with cervical neck pain improvement.

The purpose of this paper is to show a case of a young woman with a posttraumatic AARF successfully treated conservatively. This case delineates the difficulties in diagnosing this pathology, as well as the challenges encountered in its management 12).

A 2-year-old boy with Crouzon Syndrome undergoing posterior calvarial vault expansion (PVE) surgery developed AARF as a complication.

Results: The authors believe that cranial vault surgery should be considered a potential risk procedure for AARF, especially if it is done in susceptible populations (syndromic craniosynostosis patients) with other underlying sequelae (tonsillar ectopia or syringomyelia). During surgery, careful attention should be paid to maintaining a neutral alignment of the patient’s cervical spine as rotatory movements under anesthesia and muscle relaxation may be contributory factors.

AARF should be suspected and investigated in children with painful torticollis after craniofacial surgery 13).


1) , 3) , 6)

Fielding JW, Hawkins RJ. Atlanto-Axial Rotatory Fixation. (Fixed Rotatory Subluxation of the Atlanto- Axial Joint). J Bone Joint Surg. 1977; 59A:37–44
2)

Lourie H, Stewart WA. Spontaneous atlantoaxial dislocation: a complication of rheumatic disease. N Engl J Med. 1961; 265:677–681
4)

Wetzel FT, La Rocca H. Grisel’s syndrome. Clin Orthop. 1989:141–152
5)

Fielding JW, Stillwell WT, Chynn KY, et al. Use of computed tomography for the diagnosis of atlantoaxial rotatory fixation. J Bone Joint Surg. 1978; 60A: 1102–1104
7)

Schneider RC, Schemm GW. Vertebral artery insufficiency in acute and chronic spinal trauma. With special reference to the syndrome of acute central cervical spinal cord injury. J Neurosurg. 1961; 18: 348–360
8)

Kinon MD, Nasser R, Nakhla J, Desai R, Moreno JR, Yassari R, Bagley CA. Atlantoaxial Rotatory Subluxation: A Review for the Pediatric Emergency Physician. Pediatr Emerg Care. 2016 Oct;32(10):710-716. PubMed PMID: 27749670.
9)

Ishii K, Chiba K, Maruiwa H, Nakamura M, Matsumoto M, Toyama Y. Pathognomonic radiological signs for predicting prognosis in patients with chronic atlantoaxial rotatory fixation. J Neurosurg Spine. 2006 Nov;5(5):385-91. doi: 10.3171/spi.2006.5.5.385. PMID: 17120886.
10)

Kashii M, Masuhara K, Kaito T, Iwasaki M. Rotatory Subluxation and Facet Deformity in the Atlanto-occipital Joint in Patients with Chronic Atlantoaxial Rotatory Fixation: Two Case Reports. J Orthop Case Rep. 2017 Nov-Dec;7(6):59-63. doi: 10.13107/jocr.2250-0685.950. PMID: 29600213; PMCID: PMC5868886.
11)

Fielding JW, Hawkins RJ. Atlanto-axial rotatory fixation. (Fixed rotatory subluxation of the atlanto-axial joint). J Bone Joint Surg Am. 1977 Jan;59(1):37-44. PubMed PMID: 833172.
12)

García-Pallero MA, Torres CV, Delgado-Fernández J, Sola RG. Traumatic atlantoaxial rotatory fixation in an adult patient. Eur Spine J. 2017 Jan 11. doi: 10.1007/s00586-016-4916-3. [Epub ahead of print] PubMed PMID: 28078473.
13)

Saenz A, Silva AHD, Jeelani NUO, James G, Tahir MZ. A rare case of atlantoaxial rotatory fixation after posterior calvarial vault expansion surgery in a Crouzon patient. Childs Nerv Syst. 2022 May 16. doi: 10.1007/s00381-022-05554-z. Epub ahead of print. PMID: 35578116.
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