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 Table of Contents  
CASE REPORTS
Year : 2021  |  Volume : 4  |  Issue : 2  |  Page : 229-233

Midline solitary osteochondroma of C2 vertebra with myelopathy in a child: A case report with review of literature


1 Department of Spine Surgery, Park Clinic, 4, Gorky Terrace, Kolkata, India
2 Department of Spine Surgery, Park Clinic, 4, Gorky Terrace, Kolkata, India; Kothari Medical Center, 8/3, Alipore Road, Alipore, Kolkata, India

Date of Submission04-Jul-2020
Date of Decision11-Sep-2020
Date of Acceptance19-Oct-2020
Date of Web Publication24-May-2021

Correspondence Address:
Somashekar Doddabhadre Gowda
Kothari Medical Center, 8/3, Alipore Road, Alipore, Kolkata.
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/isj.isj_53_20

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  Abstract 

Spinal osteochondromas with neurological deficits are rare. Only 0.5% to 1% of spinal osteochondromas present with neurological dysfunction. We hereby present a case of osteochondroma of the C2 lamina with neurological deficit and unique presentation.A 10-year-old female presented with a history of severe weakness of all four limbs after a fall. Weakness had spontaneously improved over initial three days and she presented to us with mild weakness of the right upper limb about two weeks after the fall. On examination, neck movements were full and painless with spastic quadriparesis and a motor power of grade 4/5 in all four limbs with extensor plantars.On radiology, CT scan of the cervical spine showed an osseous outgrowth from the C2 lamina in the midline projecting into the spinal canal, directed superiorly toward the dens. An MRI showed spinal-cord compression with cord deformation and signal changes.She underwent en bloc excision of the tumor mass, which was severely impinging on the spinal cord. Histopathology confirmed a diagnosis of osteochondroma. At four-year follow-up, there was complete recovery with no signs of recurrence.Spinal osteochondroma is a rare but potential cause of spinal-cord compression in a child with varied presentation.

Keywords: Axis, myelopathy, osteochondroma


How to cite this article:
Basu S, Agarwal N, Gowda SD, Bhanta A. Midline solitary osteochondroma of C2 vertebra with myelopathy in a child: A case report with review of literature. Indian Spine J 2021;4:229-33

How to cite this URL:
Basu S, Agarwal N, Gowda SD, Bhanta A. Midline solitary osteochondroma of C2 vertebra with myelopathy in a child: A case report with review of literature. Indian Spine J [serial online] 2021 [cited 2021 Dec 4];4:229-33. Available from: https://www.isjonline.com/text.asp?2021/4/2/229/316666




  Introduction Top


Osteochondroma is a benign tumor that arises from cartilage cells of the growth plate near the surface of the bone. These tumors grow by enchondral ossification and the osseous part of the tumor is covered by a cartilaginous cap, which is unique to this tumor.[1] Osteochondroma is one of the most common bone tumors, accounting for 8.5% of all bone tumors and 36% of benign bone tumors.[2] They are diagnosed in the first or second decade of life without any gender predilection. Most of the osteochondromas originate in the appendicular skeleton as solitary or multiple entities.[3] However, only 1.3% to 4.1% of them occur in the spine. Spinal osteochondroma accounts for 4% of solitary spinal tumors. Most of the spinal osteochondromas are asymptomatic, with only 0.5% to 1% of them presenting with neurological dysfunction.[1] We present our case of C2 osteochondroma arising from lamina in the midline presenting with neurological deficit, a rare unique presentation.


  Case Report Top


A 10-year-old female presented with a history of severe weakness of all four limbs two weeks ago, which started after a fall. Fortunately, weakness improved significantly within three hours and almost recovered to normal in the next two days. Weakness was associated with neck pain, which also subsided in one day. The patient reported to us two weeks later with mild weakness of the right upper limb.

On examination, there was spasticity of all four limbs with weak bilateral hand grip (right more than left). All deep tendon reflexes of the upper and lower limb were exaggerated with plantars extensor and Hoffman’s sign positive on both sides. The neck range of movements was normal without any tenderness or sensory deficit.

X-rays were inconclusive [Figure 1]; hence, CT and MRI were done to evaluate the weakness and a skeletal survey was done to rule out multiple hereditary exostoses (MHE). The MRI and CT scan revealed large osseous outgrowth arising from the inner surface of the lamina of C2 in the midline and growing toward the odontoid process, causing significant compression of the spinal cord [Figure 2] and [Figure 3]. Preoperative CT angiography was also done to rule out vascular anomalies.
Figure 1: Preoperative X-ray showing (A) open mouth AP view and (B) neutral lateral X-ray of cervical spine

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Figure 2: Preoperative CT scan. (A) Sagittal view. (B) 3D Reconstruction shows osseous growth arising from C2 lamina and approaching toward dens through the spinal canal. (C) Axial view

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Figure 3: Preoperative MRI scan. (A) Sagittal view. (B) Axial view shows a large mass arising from posterior elements of C2 with significant cord compression and displacement of spinal cord

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Surgical treatment under intraoperative neural monitoring was planned. The tumor was approached by a posterior midline incision at the upper cervical region; gutters were made on both sides at the C2 lamina with a burr. Then, the ligamentum flavum along with the upper fourth of the C3 lamina was removed, after which the dura was exposed above and below the lesion. Thereafter, en block excision of the tumor mass along with the C2 lamina was performed, which was confirmed with X-rays, CT scan, and MRI postoperatively [Figure 4]. The sample was sent for histopathological examination.
Figure 4: Postoperative scan. (A) X-ray. (B) CT scan, and (C) MRI scan shows adequate tumor removal with postoperative changes

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Histopathology was consistent with a diagnosis of osteochondroma. Weakness also improved to normal power within three months following surgery. At two-year follow-up, mild instability at C2/3 was noted on dynamic X-rays [Figure 5] but it was not evident at four-year follow-up. However, there was complete recovery of neurology with full neck range of movements without any pain or restriction of daily activity. At four-year follow-up, X-ray, CT scan, and MRI scan show no evidence of recurrence [Figure 6]. Mild cervical kyphosis with disc degeneration from C2/3 to C5/6 was noted at four-year follow-up, without any clinical symptoms and with a pain-free full neck range of movements.
Figure 5: Two-year follow-up of dynamic X-rays shows instability at C2/3

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Figure 6: Four-year follow-up X-ray with dynamic views (A, B, and C), CT scan (D), and MRI scan (E) shows no major instability and no recurrence is noted. However, cervical spine kyphosis along with disc degeneration from C2/3 to C5/6 is noted

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The patient and their parents were informed that data from the case would be submitted for publication, and they gave their consent for the same.


  Discussion Top


Cervical spine is the most common site for spinal osteochondroma, with C2 being the most common vertebra to get affected.[4] Increased mobility at the cervical spine causes microtrauma and stress, which can stimulate exophytic growth at epiphysis.[3] In the cervical spine, posterior elements are commonly affected because of increased ossification centers.[3]

Solitary spinal osteochondroma are mostly asymptomatic and only a few cases present with neurological dysfunction.[5] Presenting symptoms can be pain, swelling, radiculopathy, or myelopathy. They are less likely to present with acute onset of symptoms after a fall, as in our case.

Rare symptoms such as pharyngeal mass, dysphagia, hoarseness, headache, obstructive sleep apnea, and vertigo can occur and sudden death may occur in very rare cases. Patients may also present with Horner’s syndrome, Arnolds neuralgia, or cranial nerve palsy.[2],[6],[7],[8],[9] We are presenting this case report, as our patient came with an unusual history of sudden onset of severe weakness of four limbs that was preceded by a minor fall. She had no history of pain or neurological symptoms in the past.

There are many case reports of spinal osteochondroma reported in the literature. In his paper, Albrecht et al. have shown 96 cases of solitary osteochondroma reported in the literature between 1843 and 1992.[5] Then, Gille et al. reviewed the literature and reported 54 cases of solitary spinal osteochondroma from 1992 to 2003.[10] A literature review by Lotfinia et al. showed 12 cases of spinal osteochondroma that presented with spinal-cord compression between 1843 and 2009.[1] Recently, in his case series with a literature review on solitary spinal osteochondroma, Yakkanti et al. disclosed 132 cases of solitary spinal osteochondroma in the literature from 2004 to 2016, out of which 36 cases presented with myelopathy.[4]

Complete excision of osteochondroma is the treatment of choice for patients presenting with neurological dysfunction.[4] Post-laminectomy kyphosis and disc degeneration should be kept in mind during follow-up. Follow-up along with radiological investigations is necessary because of the small risk for recurrence and malignant transformation.[10]


  Conclusion Top


Spinal osteochondroma most commonly arises in the cervical spine, and C2 is affected more than other vertebrae. They have a wide variety of presentations, some of which are rare, as in our case, and which should be kept in mind while evaluating a case of spinal-cord compression in a child. Complete excision of osteochondroma is the treatment of choice. Follow-up is necessary to monitor post-laminectomy kyphosis and disc degeneration.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Lotfinia I, Vahedi P, Tubbs RS, Ghavame M, Meshkini A. Neurological manifestations, imaging characteristics, and surgical outcome of intraspinal osteochondroma. J Neurosurg Spine 2010;12:474-89.  Back to cited text no. 1
    
2.
Kouwenhoven JW, Wuisman PI, Ploegmakers JF. Headache due to an osteochondroma of the axis. Eur Spine J 2004;13:746-9.  Back to cited text no. 2
    
3.
Tubbs RS, Maddox GE, Grabb PA, Oakes WJ, Cohen-Gadol AA. Cervical osteochondroma with postoperative recurrence: Case report and review of the literature. Childs Nerv Syst 2010;26:101-4.  Back to cited text no. 3
    
4.
Yakkanti R, Onyekwelu I, Carreon LY, Dimar JR II. Solitary osteochondroma of the spine—A case series: Review of solitary osteochondroma with myelopathic symptoms. Global Spine J 2018;8:323-39.  Back to cited text no. 4
    
5.
Albrecht S, Crutchfield JS, SeGall GK. On spinal osteochondromas. J Neurosurg 1992;77:247-52.  Back to cited text no. 5
    
6.
Murphey MD, Choi JJ, Kransdorf MJ, Flemming DJ, Gannon FH. Imaging of osteochondroma: Variants and complications with radiologic-pathologic correlation. Radiographics 2000;20:1407-34.  Back to cited text no. 6
    
7.
Grivas TB, Polyzois VD, Xarchas K, Liapi G, Korres D. Seventh cervical vertebral body solitary osteochondroma. Report of a case and review of the literature. Eur Spine J 2005;14:795-8.  Back to cited text no. 7
    
8.
Reckelhoff KE, Green MN, Kettner NW. Cervical spine osteochondroma: Rare presentation of a common lesion. J Manipulative Physiol Ther 2010;33:711-5.  Back to cited text no. 8
    
9.
Morard M, de Preux J. Solitary osteochondroma presenting as a neck mass with spinal cord compression syndrome. Surg Neurol 1992;37:402-5.  Back to cited text no. 9
    
10.
Gille O, Pointillart V, Vital JM. Course of spinal solitary osteochondromas. Spine (Phila Pa 1976) 2005;30: 13-9.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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