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Table of Contents
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 1  |  Page : 172-173

Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report


1 Neurological Surgery Resident Physician, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
2 Spine Surgery, Department of Orthopedics, Davao Doctors Hospital, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
3 Program Director, Department of Neurosurgery, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines
4 Training Officer, Department of Neurosurgery, Southern Integrated Neurological Surgery Training Program, Davao City, Philippines

Date of Web Publication4-Jul-2017

Correspondence Address:
P Reyes Rechilda Rhea
Neurological Surgery Resident Physician, Southern Philippines Integrated Neurological Surgery Training Program, Davao City
Philippines
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Source of Support: None, Conflict of Interest: None


DOI: 10.5530/ami.2015.1.31

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  Abstract 


Introduction: Os odontoideum is a rare lesion of the axis. Described as an ossicle, consisting of smooth and separate caudal portions of the odontoid process wherein its etiology has remained controversial. Os odontoideum complicating a possible osteogenesis imperfecta has not been reported before in the literature. Methods: We report the case of a 14-yr male patient presented with progressive weakness of both lower and upper extremities after a head trauma 10 months prior to admission, which presented as transient quadreparesis. Results: Magnetic resonance imaging of cervical spine showing cervicomedullary junction compression. Patient underwent surgical intervention and 3 months post operation, patient was reported to have steady gait and muscle grading of 5/5 on all extremities. Conclusion: Surgical fixation and fusion in patients with instability may prevent catastrophic neurologic insult after minor trauma in the future.

Keywords: Dystropic Os Odontoideum, Osteogenesis imperfect, Occipito-thoracic fusion, Suboccipital craniectomy


How to cite this article:
Rhea P R, Ronald P T, James A S, Paul G L. Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report. Acta Med Int 2015;2:172-3

How to cite this URL:
Rhea P R, Ronald P T, James A S, Paul G L. Occipito-thoracic fusion in incidental dystropic os odontoideum in a patient suspected with osteogenesis imperfecta: A case report. Acta Med Int [serial online] 2015 [cited 2020 Nov 25];2:172-3. Available from: https://www.actamedicainternational.com/text.asp?2015/2/1/172/209442




  Introduction Top


Os odontoideum is a rare lesion of the axis described in 1886 by Giacomini as an ossicle, consisting of smooth and separate caudal portions of the odontoid process wherein its etiology has remained controversial.[1],[2] To the best of our knowledge, Os odontoideum complicating a possible osteogenesis imperfecta[3],[4],[5] has not been reported before in the literature.


  Case Report Top


We report a case of a 14-year-old male presented with progressive weakness of both lower and upper extremities. He reported an unsteady gait requiring assistance associated with lack of fine motor coordination in his hands. He had a trauma of his head and neck 10 months prior to admission resulting to a transient quadriparesis. Physical examination revealed limited cervical spine flexion and extension. On neurological examination, his gait was wide-based, unsteady and spastic. Both lower and upper extremities had a muscle weakness grade 3 of 5. Sensory deficits are present in both upper and lower extremities. He had a symmetrical hyperreflexia, as well as positive Hoffman sign, Babinski sign and clonus bilaterally. Radiographs of the cervical spine showed atlantoaxial instability from a dystrophic Os odontoideum with associated cervicomedullary compression [Figure 1].
Figure 1: Noncontrast computed tomography of cervical spine demonstrates the ossicle displaced near the basion and deformed bodies of C3 to T1

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CT scan studies demonstrated a displaced ossicle located near the basion. The bodies of C3 to T1 are deformed with areas of nonfusion [Figure 2].
Figure 2: Magnetic resonance imaging of cervical spine showing cervicomedullary junction compression

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The patient underwent suboccipital craniectomy, C1 laminectomy, occipito-thoracic fusion using rods and screws [Figure 3] and [Figure 4]. At the time of the latest follow-up at 3 months, his unsteady gait had fully recovered and the muscle grade has improved to 5 of 5.
Figure 3: Intraoperative Occipito-thoracic fixation and fusion using rods and screws

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Figure 4: Postoperative lateral radiograph showing Occipito-thoracic fixation and fusion using rods and screws

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  Discussion Top


Os odontoideum is rare and an important cervical spine abnormality that can be associated with significant spinal cord injury and should be considered when a young patient has symptoms consistent with upper cervical spine compression.[6],[7] Surgical fixation and fusion in patients with instability may prevent catastrophic neurologic insult after minor trauma in the future.



 
  References Top

1.
Wollin DG: The osodontoideum. Separate odontoid process. J Bone Joint Surg Am 45:1459–1471, 1963.  Back to cited text no. 1
    
2.
Giacomini C: Sull' esistenza dell' “osodontoideum” nell' vomo. Gior Accad Med Torino 49:24–28, 1886.  Back to cited text no. 2
    
3.
Gordon CM, Bachrach LK, Carpenter TO, Crabtree N, El-Hajj Fuleihan G, Kutilek S, et al. Dual energy X-ray absorptiometry interpretation and reporting in children and adolescents: the 2007 ISCD Pediatric Official Positions. J Clin Densitom. Jan-Mar 2008;11(1):43–58. [Medline].  Back to cited text no. 3
    
4.
Rauch F, Plotkin H, DiMeglio L, Engelbert RH, Henderson RC, Munns C, et al. Fracture prediction and the definition of osteoporosis in children and adolescents: the ISCD 2007 Pediatric Official Positions. J Clin Densitom. Jan-Mar 2008;11(1):22–8. [Medline].  Back to cited text no. 4
    
5.
Baim S, Leonard MB, Bianchi ML, Hans DB, Kalkwarf HJ, Langman CB, et al. Official Positions of the International Society for Clinical Densitometry and executive summary of the 2007 ISCD Pediatric Position Development Conference. J Clin Densitom. Jan-Mar 2008;11(1):6–21. [Medline].  Back to cited text no. 5
    
6.
Clements WD, Mezue W, Mathew B. Osodontoideum: congenital or acquired? That's not the question. Injury. 1995;26(9):640–642.  Back to cited text no. 6
    
7.
Fielding JW, Hensinger RN, Hawkins RJ. Osodontoideum. J Bone Joint Surg Am. 1980;62(3):376–383.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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