Volume 6, Issue 1 (Winter 2020)                   Iran J Neurosurg 2020, 6(1): 35-40 | Back to browse issues page


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Farrokhi M R, Tarokh A, Safaei J. Can C2 Vertebroplasty Restore Alignment in Pathologic Odontoid Fracture?Odontoid Fracture?. Iran J Neurosurg 2020; 6 (1) :35-40
URL: http://irjns.org/article-1-197-en.html
1- Professor of Neurosurgery, Shiraz Neuroscience Research Center, Shiraz University of Medical Sciences, Shiraz, Iran And Department of Neurosurgery, Shiraz University of Medical Sciences, Shiraz, Iran , farrokhimr@yahoo.com
2- Neurosurgeon, Shiraz Neuroscience Research Center, Shiraz University of Medical Sciences, Shiraz, Iran And Department of Neurosurgery, Shiraz University of Medical Sciences, Shiraz, Iran
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1. Introduction
olitary plasmacytoma of bone is a rare plasma cell tumor that mainly involves the axial skeleton which progresses indolently and may result in pathologic fracture, deformity, instability neurologic deficit and pain [1]. Plasmacytoma is more likely to involve the spine (up to 50% of cases), and is most commonly found within the thoracic spine. Up to half of patients diagnosed with plasmacytoma progress to develop multiple myeloma within 2 years. The diagnosis of solitary plasmacytoma is established histologically by needle or open bone marrow biopsy. Because plasmacytoma is a radiosensitive lesion, conventional external beam radiotherapy is the primary treatment modality [1]. Surgical intervention is reserved for acute instability and neurologic compromise [1]. Pain is usually localized and constant and aggravates with the recumbent position. The management of pain mainly consists of medical therapy with non-steroidal anti-inflammatory agents, corticosteroid injection, hormone replacement therapy, and therapeutics with the osteogenic property such as calcitonin [2].
Percutaneous vertebroplasty was first described by Galibert and colleagues in 1984 [3] for treatment of vertebral angioma, which is now recognized as a standard treatment for painful osteoporotic fractures and vertebral metastases. Vertebroplasty has been shown to induce immediate and durable pain relief as much as 80% to 90%, with an acceptable complication rate of 2%-4% [2].
Lower and midcervical vertebroplasty is typically addressed with the anterolateral approach, introducing the needle between the upper airways medially and carotid-jugular vessels laterally [2]. Vertebroplasty of C2 has been practiced via percutaneous [4, 5] transpedicular and direct trans-oral routes [3]. Post-operative infection, excessive bleeding and retro-pharyngeal abscess formation may occur in trans-oral vertebroplasty, which is less common in anterior percutaneous approach [3]. In addition to pain reduction through an exothermic reaction at polymethyl methacrylate (PMMA) bone [6] interface via destroying sensitized nociceptor endings, PMMA has shown to possess oncolytic properties [7]. Herein, we present a case of solitary plasmacytoma with osteolytic C2 lesion, for whom fluoroscopically guided anterior percutaneous C2 vertebroplasty was performed, which resulted in long-standing pain control and maintenance of normal motions, as well as C1-C2 alignment.
2. Case Presentation
A 52-year-old woman, who presented with severe axial neck pain over the last 5 months, was referred to our neurospine clinic. Her physical examination revealed upper cervical tenderness without any neurologic deficits. The pain was aggravated by cervical movement, especially in anterior flexion. A cervical CT scan showed a well-defined osteolytic lesion in the odontoid process with extension into the body of the axis. After discussing the potential risks of the procedure with the patient and her family, written informed consent was obtained for operation. All the primary lab data, including hematologic profile and urine analysis and metastasis workup, were within the normal range, excluding the possibility of a primary tumor or metastasis. On the day of the first visit, her Visual Analog Scale (VAS) score was 10 and resistant to all conservative measures (Figure 1).

 

Following general anesthesia and intubation, the patient was positioned supine, the neck was extended, and a roll was placed under the patient’s shoulder between the scapulae. The right aspect of anterior cervical was then prepped with a povidone-iodine solution. Under biplanar fluoroscopic guidance, a 22-gauge needle was inserted percutaneously from the anterior-inferior aspect into the C2 body which was further replaced by a 6-inch 13 gauge Jamshidi needle. After a needle biopsy, with the aid of continuous fluoroscopic guidance, approximately 3 cc of PMMA was slowly injected to fill the lesion and after 5 minutes, the needle was gently retracted. Fluoroscopic images confirmed the successful filling of the defect without leakage of the cement. In the post-operative course, a rigid cervical collar was fixed for the patient (Figure 2). The VAS score decreased from 10 prior to operation to 2 immediately after recovery from general anesthesia. Early post-operative flexion and extension cervical X-rays showed acceptable C1-C2 alignment. Samples of pathologic study revealed C2 solitary plasmacytoma. A PET (positron emission tomography) scan excluded the accompanying bony lesions (Figure 3). 
 

3. Discussion
Solitary plasmacytoma may present with pain, pathologic fractures, and devastating neurological compromise. Vertebroplasty is an effective measure for the treatment of painful solitary plasmacytoma involving the spinal column in mid to lower cervical and thoracolumbar area [2]. Injection of PMMA not only results in pain relief but also has shown to possess oncolytic properties and may enhance the cervical spine biomechanics with improving stiffness and axial load-bearing characteristics (Figure 4).

 

Vertebroplasty in the upper cervical spine is technically challenging through anterolateral and transpedicular approaches, with potential complications of endangering the carotid sheath and vertebral arteries, respectively. As shown in this case, fluoroscopically guided anterior percutaneous C2 vertebroplasty can be safely practiced in experienced hands as an alternative to open surgery with the aim of pain relief and restoration of regional alignment; however further studies and biomechanical investigations are needed to support this hypothesis (Figure 5).
 

A few instances of this technique are available in the literature. Farrokhi et al. [2] reported trans-oral vertebroplasty in patients with C2 metastasis for pain palliation. They showed in their case report the efficacy of this treatment with significant improvement in VAS scores. They suggested that female patients and those who suffered metastasis from breast cancer may have more chances of becoming pain-free after vertebroplasty (VAS=0) (Figure 6).
 

In some other studies, [8, 9] the transoral vertebroplasty of C2 metastatic lesion was practiced, followed by posterior fusion. It is believed that vertebroplasty not only alleviates pain but also enhances the biomechanical properties of the axis to withstand axial load and in the absence of pre-operative overt instability, posterior fusion seems unnecessary [9]. Besides, posterior fusion limits cervical motion and thus may negatively affect the quality of life.
We presented our experience with anterior percutaneous vertebroplasty of a painful C2 pathologic fracture, with an emphasis on the preservation of normal cervical motion through avoiding an unnecessary posterior fusion procedure, with the durability of pain palliation after 36 months and no recurrences. We agree that this procedure has a high risk of vascular injury, and bleeding, as of other minimally-invasive procedures. Careful case selection, a meticulous technique, and acceptable experience with anterior percutaneous procedures are essentials before proceeding with this approach.
4. Conclusions
Percutaneous C2 vertebroplasty can be practiced as an effective treatment for highly selected C2 osteolytic lesions without a clear indication for open surgery. The results may be long-term pain palliation, restoration of cervical motion, and improved quality of life. Nevertheless, the safety of the approach and the efficacy of improving the regional biomechanics of the spine are to be assessed in future studies. 
Ethical Considerations
Compliance with ethical guidelines

All ethical principles are considered in this article. The participants were informed about the purpose of the research and its implementation stages; they were also assured about the confidentiality of their information; moreover, they were free to leave the study whenever they wished, and if desired, the research results would be available to them.
Funding
This research did not receive any grant from funding agencies in the public, commercial, or non-profit sectors. 
Authors' contributions
Conceptualization, Data analysis, and Supervision: Majid Reza Farrokhi, Amir Tarokh; Data collection: Amir Tarokh; Drafting the article: Amir Tarokh; Critically revising: Majid Reza Farrokhi, Amir Tarokh, Javad Safaei; Reviewing submitted version of manuscript: Amir Tarokh, Javad Safaei; 
Conflict of interest
The authors declared no conflict of interest.


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Type of Study: Case report | Subject: Spine

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