Clinically, the majority of vertebral hemangiomas are asymptomatic and discovered by chance, and usually no treatment is required. However, aggressive vertebral hemangioma could destroy the vertebral structure, and cause pain, local malformations, and even spinal cord compression[15]. In 1927, Makrykostas reported a case of spinal stenosis due to aggressive vertebral hemangioma that led to neurological symptoms[16]. Therefore, active treatment is usually required for patients with significant spinal instability and spinal nerve compression. At present, there are many treatment methods for aggressive vertebral hemangioma, including total vertebral resection and decompression surgery, partial vertebral resection, preoperative radiotherapy, vascular embolization, and intraoperative vertebroplasty with bone cement[17]. Most of those methods achieve certain effects, but selection of the optimal treatment strategy is still controversial. For aggressive vertebral hemangiomas, the aim of surgery is to relieve compression and restore spinal stability to improve symptoms while minimizing recurrence. Although total vertebral resection can effectively reduce tumor recurrence, it is limited by its complications, such as massive intraoperative blood loss, high risk of nerve injury, and other complications. Chen[8] reported a case series of patients with compressive vertebral hemangioma, and showed that blood loss for the en bloc procedure was around 1,200 mL and the surgical duration was 2.3 hours. He stated that for aggressive vertebral hemangioma with incomplete paralysis, total en bloc spondylectomy should be considered. However, his article included only one patient who had undergone en bloc spondylectomy. Ryo Ogawa[18] described a patient with aggressive vertebral hemangioma with spinal cord compression; the patient received total en bloc spondylectomy, preceded by arterial embolization, and lost 2,232 g of blood after 413 minutes of surgery. In a multicenter study, Goldstein[3] explored the results of surgical treatment for spinal hemangiomas. By analyzing 68 patients who had undergone surgical treatment, he concluded that it is unnecessary to perform total en bloc resection to achieve a wide surgical range, as total resection would lead to more intraoperative bleeding and higher surgical morbidity. Xinran Ji[4] reported 23 patients with vertebral hemangiomas treated with total en bloc spondylectomy. The mean operation time of patients with preoperative embolization was 426.6 ± 104.3 min and the mean blood loss was 1,883.3 ± 932.1 mL. Neurological deficits improved in all cases, without recurrences. As surgery alone can lead to significant blood loss and mortality, it can be combined with various preoperative or intraoperative measures. With the improvement of surgical techniques, it would be possible to relieve neurological function and restore spinal stabilization.
Aggressive vertebral hemangioma is rich in blood supply and often accompanied by massive bleeding during surgical resection. Endovascular embolization is one of the effective non-surgical methods for treatment of aggressive vertebral hemangioma. Antonino Raco[19] reported five vertebral hemangioma patients with neurological symptoms. At first, embolization was attempted for them, and if the symptoms persisted, decompressive laminectomy would be performed. Two of them received embolization as the only treatment, and after 18 and 36 months of follow-up, they achieved remission of symptoms without recurrence. Hurley[13] reported on Onyx embolization of two aggressive vertebral hemangiomas, wherein the post-operative CT confirmed the desired results of Onyx distribution through the body with good visibility, good control, and shorter injection times. However, there were also some negative experiences, such as lower effective rate and higher frequency of relapse. In Smith’s[20] research, two patients who received embolization as a sole treatment did not improve clinically. Yilmaz Kiroglu[21] reported a case of a pregnant woman suffering from T4 vertebral hemangioma and used endovascular embolization as sole treatment. Her symptoms and neurologic deficits improved quickly after the embolization, but restarted two years later, so that she had to receive intraoperative vertebroplasty and internal fixation at last. Therefore, endovascular embolism is not only an independent treatment but also an efficacious adjunctive procedure to reduce bleeding and improve surgical safety.
Just as endovascular embolization, vertebroplasty with bone cement is an independent treatment of aggressive vertebral hemangioma, as well as an aid to surgical resection and decompression. It can further embolize small vessels and destroy the tumor tissue while enhancing the strength of the vertebral body. At the same time, vertebroplasty is also an effective method to reduce the risk of recurrence and relieve local pain[22]. Spinal cord compression caused by leakage or expansion of bone cement is the most common complication of this procedure[23]. Moreover, vertebroplasty makes the spine segment stiffer and could cause adjacent segment fracture. This risk was more frequent with older generations of bone cement. Majed Issa[24] reported two cases of aggressive vertebral hemangiomas treated with vertebroplasty. Under biplanar fluoroscopic guidance, acrylic cement was injected in the vertebral body without leakage, and the clinical efficacy and safety were satisfactory at two-year follow-up. Ben Wang[12] retrospectively reviewed 39 patients diagnosed with aggressive (Enneking stage 3, S3) vertebral hemangiomas, including 17 patients who had undergone decompression alone and another 22 patients who had received decompression along with intraoperative vertebroplasty. The average estimated blood loss of decompression group was higher than of decompression plus vertebroplasty group (1,764.7 mL vs. 1,068.2 mL), and vertebroplasty helped in minimizing recurrence after decompression.
Here, we presented different surgical managements of aggressive vertebral hemangioma, which involved preoperative embolization, vertebroplasty, decompression, and internal fixation. We demonstrated that the combination of embolization and vertebroplasty can be carried out with less blood loss and improvement in pain and neurological function. Only one patient recurred after the treatment. Considering high local infiltration and rapid growth characteristics of aggressive vertebral hemangioma, this recurrence rate is acceptable, which proves the effectiveness and durability of the treatment strategy in this study. However, it was surprising that the only recurrence case was in group C, which showed that even the comprehensive treatment including canal decompression, endovascular embolization, vertebroplasty, and internal fixation does not mean absolute cure.