Cervical myelopathy caused by atlantoaxial instability in a patient with an os odontoideum and total aplasia of the posterior arch of the atlas: a case report

✓ The authors placed titanium mesh cages to achieve posterior atlantoaxial fixation in five patients with atlantoaxial instability caused by rheumatoid arthritis or os odontoideum. A mesh cage packed with autologous cancellous bone was placed between the C-1 posterior arch and the C-2 lamina and was tightly connected with titanium wires. Combined with the use of transarticular screws, this procedure provided very rigid fixation. Solid fusion was achieved in all patients without major complications. The advantages of this method include more stable fixation, better control of the atlantoaxial fixation angle, and reduced donor-site morbidity compared with a conventional atlantoaxial arthrodesis in which an autologous iliac crest graft is used.

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Bifid Anterior and Posterior Arches of Atlas

Neurosurgery ◽

10.1227/neu.0000000000000761 ◽

2015 ◽

Vol 77 (2) ◽

pp. 296-306 ◽

Cited By ~ 22

Author(s):

Atul Goel ◽

Trimurti Nadkarni ◽

Abhidha Shah ◽

Raghvendra Ramdasi ◽

Neeraj Patni

Keyword(s):

Craniovertebral Junction ◽

Basilar Invagination ◽

Atlantoaxial Instability ◽

Os Odontoideum ◽

Occipital Condyle ◽

Posterior Arch ◽

Discrete Movements ◽

General Observation ◽

Fixation Techniques ◽

Group 1

Abstract BACKGROUND: On reviewing the database of patients with craniovertebral junction anomalies, the authors identified 70 patients with a bifid posterior arch of atlas. OBJECTIVE: To speculate on the pathogenesis of spondyloschisis of both the anterior and posterior arches of atlas, particularly as it relates to atlantoaxial instability. METHODS: Seventy patients with bifid anterior and posterior arches were identified by a retrospective review of the database from 2007 to 2013. RESULTS: The ages of the patients ranged from 14 months to 50 years. The patients were divided into 3 groups. Group 1 (3 patients) had multiple additional spinal bony and neural abnormalities. Group 2 (34 patients) had mobile and partially (5) or completely (29) reducible atlantoaxial dislocation. Group 3 (33 patients) had atlantoaxial instability and related basilar invagination. The os odontoideum was identified in 21 patients, and C2-3 fusion was seen in 24 patients. Two of 3 patients in group 1 were treated conservatively and without any surgery. All patients in groups 2 and 3 were surgically treated. Surgery was done using lateral mass plate/rod and screw fixation techniques. The general observation during surgery included identification of discrete movements of both halves of the atlas, lateral positioning of the facets of atlas in relation to the facets of the axis and occipital condyle and closer approximation of the occipital bone, atlas, and axis resulting in “crumpling” of bone and neural elements. CONCLUSION: Understanding of the pathogenesis and mechanical alterations in cases with a bifid arch of atlas can assist in evaluating the clinical implications and in conduct of surgery.

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Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study

Orphanet Journal of Rare Diseases ◽

10.1186/s13023-021-02031-9 ◽

2021 ◽

Vol 16 (1) ◽

Author(s):

Hidetomi Terai ◽

Koji Tamai ◽

Masatoshi Hoshino ◽

Hiromitsu Toyoda ◽

Akinobu Suzuki ◽

...

Keyword(s):

Cervical Myelopathy ◽

Posterior Longitudinal Ligament ◽

Atlantoaxial Instability ◽

Symptom Improvement ◽

Posterior Arch ◽

Type I ◽

Lysosomal Storage ◽

Japanese Orthopedic Association ◽

The Mean

Abstract Background Although the clinical efficacy of laminoplasty in adult cervical spondylotic myelopathy or ossification of posterior longitudinal ligament has been frequently reported, there are only few reports of laminoplasty for patients with lysosome storage diseases (LSDs). Therefore, this study aimed to report the midterm clinical and radiological outcomes of patients with LSDs after cervical laminoplasty. Methods Six patients with LSD who underwent laminoplasty with/without C1 laminectomy for cervical myelopathy were enrolled. Clinical evaluations, including the cervical Japanese Orthopedic Association (cJOA) score and visual analog scale (VAS) scores for upper extremity numbness, and radiographic parameters, including C2–C7 lordotic angle, atlanto-dens interval (ADI), and ⊿ADI, were evaluated preoperatively, at 2 years postoperatively, and at the final follow-up. Results Five patients had mucopolysaccharidoses (type I: n = 1, II: n = 3, VII: n = 1) and one patient had mucolipidoses type III. The mean age of patients at surgery was 27.5 years, and the mean postoperative follow-up period was 61 months. All mucopolysaccharidoses cases required C1 posterior arch resection with C2–C7 laminoplasty. No critical complications were observed postoperatively. There were no significant differences in C2–C7 angle (p = 0.724) and ⊿ADI (p = 0.592) between the preoperative and final follow-ups. The cJOA score and VAS for numbness significantly improved at the final follow-up (p = 0.004 and p = 0.007, respectively). Conclusions The cervical myelopathy in patients with LSD could be safely and effectively treated with laminoplasty with/without C1 posterior arch resection after excluding patients with atlantoaxial instability. Atlantoaxial stability and symptom improvement could be maintained at an average of 5 years postoperatively.

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Atlanto-axial subluxation with cervical myelopathy operated with occipital C2 fusion: A case report

Journal of Medical Research and Innovation ◽

10.15419/jmri.3 ◽

2017 ◽

Vol 1 (1) ◽

pp. 4-7

Author(s):

Rahul Kadam ◽

Vishal Bauva ◽

Krutarth Shah ◽

Sunil Yadav

Keyword(s):

Case Report ◽

Cervical Myelopathy ◽

Muscle Tone ◽

Rare Condition ◽

Posterior Arch ◽

Atlantoaxial Joint ◽

Posterior Aspect ◽

Posterior Displacement ◽

Grisel Syndrome ◽

Magnetic Resonance Imaging Mri

Background: Atlantoaxial subluxation with cervical myelopathy is a rare condition that can occur mainly by trauma followed by Rheumatoid arthritis, Grisel syndrome, Down’s syndrome and various other metabolic disorders. It is characterized by excessive movement of atlas (C1) over axis (C2) either by bony or ligamentous abnormality. Due to its laxity the spinal cord may get damaged and cause neurologic symptoms. Reduction and fixation is needed for such instability. Case Report: This 55-year-old gentleman was apparently all right 4 years back when he gradually developed difficulty in walking and imbalance. Bilateral Babinski sign was positive, All deep tendon reflexes were brisk; muscle tone was increased with clasp-knife spasticity present in all four limbs. Ankle and patellar clonus was present bilaterally. His X-ray cervical spine showed C1-C2 subluxation in flexion and extension views. Magnetic resonance imaging (MRI) of Cranio-vertebral junction. Mild subluxation of atlantoaxial joint (3.1 mm) with posterior displacement of dens causing narrowing of bony cervical spinal canal with reduced distance between posterior aspect of dens of C2 and anterior aspect of posterior arch of C1 vertebrae was noticed. Atlas was also slightly displaced anteriorly in relation to baso-occiput. We managed this patient with occipital cervical fusion after reduction from a posterior approach using screws and rods construct and fusion with bone graft from iliac crest. Post operatively the patient was able to walk without any support and tone of the muscles in lower limb decreased, no tingling or numbness are present, no signs of local infection or inflammation. Conclusion: We suggest to operate atlanto-axial subluxation and cervical myelopathy with occipital C2 fusion.

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Clinical Outcomes of Laminoplasty for Patients With Lysosomal Storage Disease Including Mucopolysaccharidosis and Mucolipidoses: a Retrospective Cohort Study

10.21203/rs.3.rs-651514/v1 ◽

2021 ◽

Author(s):

Hidetomi Terai ◽

Koji Tamai ◽

Masatoshi Hoshino ◽

Hiromitsu Toyoda ◽

Akinobu Suzuki ◽

...

Keyword(s):

Cervical Myelopathy ◽

Posterior Longitudinal Ligament ◽

Atlantoaxial Instability ◽

Symptom Improvement ◽

Posterior Arch ◽

Type I ◽

Lysosomal Storage ◽

Japanese Orthopedic Association ◽

Atlantoaxial Stability

Abstract Background:Although the clinical efficacy of laminoplasty in adult cervical spondylotic myelopathy or ossification of posterior longitudinal ligament has been frequently reported, there are only a few reports of laminoplasty for patients with lysosome storage diseases (LSDs). Thus, this study aimed to report the midterm clinical and radiological outcomes of patients with LSDs after cervical laminoplasty.Methods: Six LSD patients who underwent laminoplasty with/without C1 laminectomy for cervical myelopathy were enrolled. Clinical evaluations, including the cervical Japanese Orthopedic Association (cJOA) score and visual analog scale (VAS) for upper extremity numbness and radiographic parameters, including C2-7 lordotic angle, atlanto-dens interval (ADI), and ⊿ADI, were evaluated preoperatively, at 2 years postoperatively, and at the final follow-up. Results: Five patients had mucopolysaccharidoses (type I: n=1, II: n=3, VII: n=1) and one patient had mucolipidoses type III. Mean age at surgery was 27.5 years. Mean postoperative follow-up period was 61 months. All mucopolysaccharidoses cases required C1 posterior arch resection with C2-C7 laminoplasty. No critical complications were observed postoperatively. There were no significant differences between preoperative and final follow-up in C2-7 angle (p=0.724) and ⊿ADI (p=0.592). The cJOA score and VAS for numbness significantly improved at the final follow-up (p=0.004 and p=0.007, respectively).Conclusion: The cervical myelopathy of LSD patients could be safely and effectively treated with laminoplasty with/without C1 posterior arch resection after eliminating patients with atlantoaxial instability. The atlantoaxial stability and symptom improvement could be maintained at an average of 5 years postoperatively.

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Ossification of the Posterior Atlantoaxial Membrane Associated with an Os Odontoideum: A Case Report

Journal of Orthopaedic Surgery ◽

10.1177/230949901101900330 ◽

2011 ◽

Vol 19 (3) ◽

pp. 392-394 ◽

Cited By ~ 2

Author(s):

Junichi Ohya ◽

Hirotaka Chikuda ◽

Shurei Sugita ◽

Takashi Ono ◽

Yasushi Oshima ◽

...

Keyword(s):

Case Report ◽

Motor Vehicle ◽

Motor Vehicle Accident ◽

Atlantoaxial Instability ◽

Os Odontoideum ◽

Ectopic Ossification ◽

Vehicle Accident ◽

A Minor ◽

Atlantoaxial Arthrodesis

We report a case of ossification of the posterior atlantoaxial membrane associated with an os odontoideum in a 46-year-old woman. She developed myelopathy following a minor motor vehicle accident. The patient underwent posterior atlantoaxial arthrodesis and resection of the ossified lesion and recovered uneventfully. Long-standing atlantoaxial instability might have played a role in ectopic ossification of the posterior atlantoaxial membrane.

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Dystopic Os odontoideum causing cervical myelopathy: A rare case report and review of literature

Asian Journal of Neurosurgery ◽

10.4103/ajns.ajns_35_19 ◽

2020 ◽

Vol 15 (1) ◽

pp. 236

Author(s):

Aditya Raj ◽

SudhirKumar Srivastava ◽

Nandan Marathe ◽

Sunil Bhosale ◽

Shaligram Purohit

Keyword(s):

Case Report ◽

Cervical Myelopathy ◽

Rare Case ◽

Os Odontoideum ◽

Review Of Literature ◽

Rare Case Report

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Adult Morquio syndrome requiring occipito-thoracic fusion

Journal of Orthopaedic Surgery ◽

10.1177/2309499020918424 ◽

2020 ◽

Vol 28 (2) ◽

pp. 230949902091842

Author(s):

Ryosuke Okumura ◽

Keiji Hasegawa ◽

Shintarou Tsuge ◽

Katsunori Fukutake ◽

Kazumasa Nakamura ◽

...

Keyword(s):

Rare Condition ◽

Atlantoaxial Instability ◽

Os Odontoideum ◽

Posterior Arch ◽

Rare Entity ◽

Posterior Decompression ◽

Radiographic Outcomes ◽

Thoracic Spinal ◽

Morquio Syndrome ◽

Fragile Bone

Morquio syndrome is a relatively rare entity that is often associated with atlantoaxial instability from early childhood due to odontoid dysplasia based on a mucopolysaccharoidal disorder. Here, we present the case of a 55-year-old male patient with Morquio syndrome who developed cervical myelopathy, which is an extremely rare condition in the older population. Myelopathy developed gradually with upper-limb paresthesia and clumsiness of both hands. The patient had a characteristic “gargoyle-like” coarse face with a trunk shortening-type short stature. Imaging of the cervical spine demonstrated several problems, including diminutive structures called platyspondyly with small pedicles and fragile bone quality, hypoplasia of the C1 posterior arch that migrated into the spinal canal, and os odontoideum with atlantoaxial instability. With intraoperative navigation guidance, posterior decompression of C1 followed by occipito-cervico-thoracic spinal fusion was successfully performed in this complicated case. Clinical and radiographic outcomes were both excellent and have been maintained for 2 years postoperatively.

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