Keywords: Aggressive hemangioma, o-arm navigation, transoral vertebroplasty
Since the first vertebroplasty was performed in 1984, the indications for performing this method have been expanding and currently include severe vertebral compression fractures, fractures associated with traumatic ruptures, and fractures due to metastatic lesions.,,,, Vertebroplasty is the most common option for vertebral osteoporotic fractures, vertebral metastatic lesions, and vertebral hemangiomas. Vertebral hemangioma is a vascular malformation of disembryogenetic origin primarily affecting vertebral bodies and is characterized by abundant vascularization, atrophy of the surrounding bone, and a long-term asymptomatic state., Hemangiomas most frequently occur in middle-aged women. Several vertebral levels are affected in 30% of cases. Vertebral hemangiomas are extremely rarely diagnosed in children under ten years of age, with most cases accounting for the age group of 30 to 60 years of age. Hemangiomas are encountered in all vertebral regions, thoracic ones being the most common., The clinical presentation includes local pains matching the affected vertebral level in 54–94% of cases., Examination algorithm for patients with vertebral hemangiomas includes mandatory spinal computed tomography (CT) and magnetic resonance imaging (MRI) scans.
In 1989, Nguyen et al. defined five topographic types: 1) hemangioma affects the entire vertebra and can have paravertebral and intracanal extensions; 2) the lesion is limited to a vertebral body; 3) isolated lesions of the posterior semi-ring; 4) involvement of a vertebral body with partial involvement of the posterior semi-ring; 5) only epidural location of the tumor.
In 1986, Laredo et al. introduced a definition for aggressive hemangioma. The authors suggested the tumor is to be qualified as aggressive in presence of three or more out of the seven radiographic signs such as localization at the level of ThIII-ThlX vertebrae; totally affected vertebral body; involved vertebral arch pedicle; osseous expansion leading to cortical layer bulging with unclear margins; hemangioma's heterogeneous structure; the presence of either epidural or paravertebral component; combined low T1 and high T2 signals in weighted MRI images; contrast accumulation while CT scanning.
To date, there is no clear answer to the question of which vertebral hemangiomas should be considered aggressive. “Aggressiveness” does not mean a morphological diagnosis.
The term “aggressive hemangioma” reflects a set of radiological symptoms indicating a connection with the clinical picture and indirectly indicating an unfavorable prognosis of the spontaneous course of the disease.,
Our case should be considered as aggressive hemangioma with the appropriate classification of Nguyen and taking into account the data of MRI, CT, and patient complaints.
The methods to manage vertebral hemangiomas are as follows: alcohol ablation, endovascular embolization, radiation therapy, vertebroplasty,, kyphoplasty, surgical decompression, or various combinations of these methods., Each method ensures an improvement of symptoms to a variable degree.
Today, percutaneous vertebroplasty is the most common method to manage aggressive hemangiomas., The method was pioneered in 1984 in France by Galibert et al., who applied an anterolateral approach to treat a C2 hemangioma., A selection of small case series, where vertebroplasty was used to manage C2 aggressive hemangiomas is available in the literature., In clinical practice, a painful aggressive vertebral hemangioma resisting conservative treatment is the most common indication for vertebroplasty, in which case its volume should exceed 50% of the vertebral body volume.,, While extensive experience has been gained in managing thoracic and lumbar lesions, transoral vertebroplasty still remains underrepresented in the literature.,
Patient A., 28 y. o. was admitted on February 27, 2019, with complaints of pain in the cervical spine radiating to the occiput (visual analog scale (VAS) score up to 5, increased during verticalization). For 3 months, the patient was treated with non-steroidal anti-inflammatory drugs (NSAIDs), pain relief did not occur. Conservative treatment turned out ineffective. In terms of neurological status, no conduction impairments were discovered. Neck Disability Index (NDI) questionnaire showed a self-rated disability of 35.
MRI and CT scans of the cervical spine displayed hemangioma of the C2 vertebral body and odontoid process base [Figure 1], [Figure 2] classified as type 2 aggressive C2 hemangioma, according to Nguyen.
Surgical intervention in the form of C2 vertebroplasty was performed on February 27, 2019. Endotracheal anesthesia was applied. The patient was placed in a supine position with the head stabilized in a Mayfield frame [Figure 3]a. The oropharyngeal cavity was prepared using a chlorhexidine water solution. A Crockard retractor was used for the transoral approach [Figure 3]b. Treatment of the oropharyngeal cavity was done with a solution of aqueous chlorhexidine. A 12-gauge needle was directed under O-ARM guidance through the anterior C2 vertebral body to the anterior-middle junction [Figure 3]c. Spinal venography with iodine contrast dye (2 ml) was performed under fluoroscopic control. No contrast leakage to paravertebral epidural veins was observed. The C2 vertebral body was filled by injecting 2 ml of bone cement under fluoroscopic control. Within 3–5 min after removing the needle, manual compression was applied to the puncture sites to minimize the formation of hematomas. The needle was removed. O-ARM control scanning was performed.
Manual compression was applied to entry points for 3–5 min after needle removal to minimize hematoma formation. Chlorhexidine oral rinsing was prescribed for 5 days after the surgery.
Post-operative pain management was successful. No post-operative complications were observed. CT control after surgery [Figure 3]d. No signs of inflammation in the surgical site were found [Figure 4]. The patient was discharged for outpatient treatment in satisfactory condition. Follow-up examinations in 3, 6, and 12 months showed VAS scores not exceeding 2 and a functional adaptation score of 10, according to the NDI questionnaire.
Vertebroplasty is becoming an increasingly common approach to the management of the severe refractory pain associated with vertebral compression fractures, aggressive hemangiomas, osteoporosis, and vertebral metastatic lesions.,,, The method's popularity is due to immediate pain syndrome relief and low incidence of complications.
The reinforcing effect of injected bone cement ensures spine stability and prevents further collapse. However, the accurate mechanism of polymethyl's analgesic effect is not fully understood. It is possible that the exothermic effects of cement polymerization directly damage interosseous and/or periosteal nerve endings, which leads to the local death of nerve cells.
For the time being, the issue of injected cement amounts to ensure appropriate treatment remains debatable. Inexperienced surgeons often ask about the amount of cement to be injected for adequate treatment. The goal of vertebroplasty is not to fill the vertebral body completely. Actually, the cement should be injected into the anterior 2/3 or 3/4 of the vertebral body. Post-operative CT control is recommended in all cases to identify possible cement migration.
There is both anterior and posterior access to the C2 vertebra. Depending on the clinical situation, posterior or transoral approaches are used. The anterolateral approach comes with a risk of damaging neurovascular bundles because the needle is guided near such structures as the vagus nerve, lingual, hypoglossal, and laryngeal nerves, jugular veins, vertebral and carotid arteries, submandibular glands, and pharynx.
Cohen et al. performed vertebroplasty of C2 with metastatic lesions and aggressive hemangiomas in 15 patients using the same approach as in transdental C2 fixation. The authors claim this approach is associated with minimal risks of cement migration and local infections, while the transoral approach is associated with a risk of local infections from the oral microbiome.
The posterolateral approach without CT control is not as safe due to the small size of the C2 isthmus and the close proximity of vertebral arteries.
Frank Floeth et al. performed surgery on seven patients with severe neck pain associated with C2 vertebral body tumors. Low-pressure filling of the resection cavity with cement followed tumor removal with an open posterior approach. The method was chosen due to major bone defects of C2, an accompanying high risk of cement migration, and the necessity of sampling for diagnosis verification.
Vertebroplasty has proved to be effective in terms of pain management and spine stability in cases of C2 metastatic lesions. Open or percutaneous surgery may be performed using either a transoral or anterolateral approach.,,, From a technical standpoint, any approach in the upper cervical spine to allow for direct access to C2 is much more challenging, than transpedicular percutaneous vertebroplasty in the thoracic or lumbar spine.
Cement migration to the vertebral canal with clinical manifestations is a potentially serious complication. However, it can be avoided under continuous lateral radiographic control. Other complications are associated with the introduction of the needle (e.g., pedicle fractures and dura mater lesions). These complications are often asymptomatic.,,
Mont'Alverne et al. reported cement migration in 7 out of 12 patients with metastatic C2 lesions after percutaneous anterolateral vertebroplasty under fluoroscopic control. In one case, cement migration after the posterolateral approach was accompanied by occipital neuralgia, and in another by cerebellar and occipital ischemic stroke, while the remaining cases were asymptomatic.
Another paper showed that asymptomatic cement migration after C1, C2, and C3 vertebroplasty was observed in 33% of cases. The authors used the anterolateral approach under CT control. Cement migration remains a common problem even for experienced surgeons. In most cases, cement migration can only be evidenced as a radiological finding without any respective clinical manifestations. However, in the upper cervical spine, this complication may pose a significant threat and cause clinical symptoms and even fatal outcomes due to the tight intertwining of nervous and vascular structures adjacent to C2.,
Brage et al. reported an unusual case of an aneurysmal bone cyst of C2 affecting the whole vertebral body successfully treated by transoral vertebroplasty. The surgery was performed under general anesthesia with continuous neurophysiological control. The patient received systemic and local oral antibiotic prophylaxis. In our case report, there was no need for neurophysiological control, since hemangioma did not cause cerebrospinal compression and no conduction impairments were observed. Brage et al. stressed that the transpedicular approach to allow for direct access to the C2 vertebral body is unsafe due to the proximity of vertebral arteries and small pedicle size.
The transoral approach is the most straightforward one to provide the shortest access to the C2 vertebral body and make it possible to avoid injuries to vital vascular structures., The method's downside is an inability to ensure aseptic conditions during the surgery. According to the literature, the incidence of infections as a result of open transoral surgery varies from 0% to 50% depending on the preoperative treatment of a surgical site, use of antibiotic prophylaxis, surgery duration, and amount of tissue trauma. No infectious complications were observed in our case. Systemic and local antibiotic prophylaxis was ensured, surgery was performed quickly, and the size of the oropharyngeal puncture was minimized. The risk of infectious complications was reduced by injecting a single dose of 2 g of cefazolin intraoperative and post-operative and by chlorhexidine oral rinsing for 5 days after the surgery. Daily pharynx examinations had shown no signs of infection by the time of discharge.
Percutaneous vertebroplasty is a safe and effective minimally invasive method to manage type 1, 2, and 4 aggressive hemangiomas according to Nguyen.,, The main objective of vertebroplasty is to restore the supporting capability of the affected vertebra and achieve an analgesic effect.,, The complex anatomy of neurovascular bundles requires precise preoperative planning and adequate intraoperative control of cement injection.
Limitations of this approach are pathological fracture of the C2 vertebra and resorption of posterior bone elements.
C2 aggressive hemangioma is a rare malformation. Treatment of this pathology is a difficult task due to anatomical features, the location of neural structures, and significant vessels.
The use of the transoral approach for C2 vertebroplasty in cases of aggressive hemangiomas ensures good functional outcomes, pain relief, improved quality of life, and functional activity.
Transoral vertebroplasty is a safe and effective therapy for C2 aggressive hemangiomas, but it requires thorough preoperative planning and adequate surgical equipment.
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Conflicts of interest
There are no conflicts of interest.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]