Multi-modal management of aggressive vertebral hemangioma: A single center experience

Article information

J Cerebrovasc Endovasc Neurosurg. 2025;27(1):40-49

Publication date (electronic) : 2025 March 13

doi : https://doi.org/10.7461/jcen.2025.E2024.12.003

Mohamed Farouk, Mohamed Ali Kassem, Ashraf Ezzeldein, Mohamed Mohsen Ameen, Ali Hassan Elmokadem, Mohamed M Elsherbini

Department of Neurosurgery, Mansoura University, Mansoura, Egypt

Correspondence to Mohamed Farouk Department of Neurosurgery, Mansoura University, Elgomhouria St., Mansoura City, Egypt 35516 Egypt Tel +20 (50) 2383781 E-mail ns.m.farouk@gmail.com

Received 2024 December 7; Revised 2024 December 24; Accepted 2025 February 13.

Abstract

INTRODUCTION

Most cases of vertebral hemangioma (VH), the most common spinal lesion, are accidentally discovered and can be managed conservatively. When patients present with constant pain, the possibility of minimally invasive approaches such as vertebroplasty should be considered, as the high vascularity of the lesion itself makes surgical intervention relatively challenging, with high risk of blood loss. Therefore, surgical interventions are typically reserved only for cases with neural compression and neurological deficit.

Aggressive hemangiomas are symptomatic due to their pathological behavior, including bone expansion, disturbance of blood flow, compression fracture, and extraosseous extension either into spinal canal or neural foramen. Aggressive hemangiomas can be presented by local, radicular, or myelopathic symptoms, due to compression by body collapse, extradural hemorrhage, or extraosseous soft tissue extension. This rare course makes it difficult to suspect clinically, its differential diagnosis includes aneurysmal bone cysts, solitary myeloma, metastatic tumors, and Paget disease [10].

Trans-arterial embolization (TAE) can be introduced as a sole line of management, or in conjunction with other lines, either as a curative line of management or as a pre-operative step to reduce intra-operative blood loss. However, despite the availability of numerous lines of management, there is no fixed algorithm for treatment options, which makes decision making more complicated and potentially inconsistent.

METHODS

RESULTS

The study included nine patients, comprising six females and three males, with a mean age of 29.3 years (ranging from 14 to 46). Most lesions (5/9) were dorsal cases, followed by lumbar (n=3) cases, and a single cervical case. Six patients underwent TAE, of whom five underwent further surgical procedures, while one patient found TAE to be sufficient as a stand-alone management technique. Eight patients underwent surgical management, five of whom were pre-operatively embolized. The surgical procedures included hemilaminectomy, laminectomy, fixation, and vertebroplasty, with various combinations of selected procedures tailored and fashioned according to patients’ presentation and radiology (Table 1).

Table 1.

Clinical and epidemiological data

Six patients presented with neurological motor deficit with various degrees, with or without long standing history of spinal axial pain, as shown in Table 1, while three patients presented with intractable axial spinal pain and radiculopathy. Three patients of those who presented with neurological deficit (3/6 patients, 50 %) showed early post-operative improvement of motor power, while two patients’ neurological status remained unchanged, and only one patient (17%) suffered post-operative deterioration of motor power after surgical procedures (laminectomy, vertebroplasty, and fixation without pre-operative embolization).

The follow-up period ranged from 94 to 531 days, with mean follow-up period of 234.4 days. During follow-up, seven patients showed intact motor power on various intervals, while one patient’s motor power remained stationary, and the other one suffered post-operative decline of motor power to grade 2, and subsequently returned to basal pre-operative neurological status (grade 4).

Histopathological analysis confirmed pathological entities for patients who underwent open surgical procedures, and pathology was confirmed only radiologically for patients who underwent only vertebroplasty. No patients underwent pre-operative CT guided biopsy.

ILLUSTRATIVE CASES

Case 1 (Fig. 1)

Fig. 1.

(A) (sagittal) and (B) (axial) post-contrast MRI showed aggressive D6 hemangioma with leak into the spinal canal causing cord compression. (C-F) Conventional catheter angiography showed feeders from right D6, right D7, left D6, and left D7 intercostal arteries (respectively). (G-J) After embolization of all feeders using particles, marked reduction of vasculature is noted. (K) Post-operative CT scan showing the fenestration bone window for hematoma evacuation, cement vertebroplasty is noticed as well allover vertebral body. MRI, magnetic resonance imaging; CT, computed tomography

A 14-year-old female presented with progressive paraparesis for two months (LL motor power grade II to III). Post-contrast MRI revealed an aggressive D6 hemangioma with leak into the spinal canal causing cord compression. Conventional catheter angiography showed feeders from right D6, right D7, left D6, and left D7 intercostal arteries. Preoperative embolization of all feeders using particles to minimize intraoperative hemorrhage risk. Paraparesis showed mild improvement after embolization (G III to IV -ve). Surgical decompression of the hematoma through fenestration with cement vertebroplasty was performed with minimal blood loss. The patient recovered more rapidly after surgery and was discharged home on the seventh postoperative day with motor power grade IV +ve.

Case 2 (Fig. 2)

Fig. 2.

(A) Sagittal T2FS MRI showed D12 leaking hemangioma compressing the conus. (B) intraoperative image of surgical procedure which included D12 laminectomy and long segment fixation using transpedicular screws. (C) and (D) antroposterior and lateral radiograph shows results after vertebroplasty, decompression and posterior fixation. MRI, magnetic resonance imaging

A 34-year-old female presented with mid-dorsal back pain, paraparesis, and urine incontinence. MRI dorsal spine showed D12 leaking hemangioma compressing the conus. Catheter angiography showed D12 aggressive hemangioma fed from D10-12 radicular arteries bilaterally (not shown). There was no definitive large feeder suitable for catheter maneuvering for TAE. Patient underwent surgical decompression with intra-operative cement injection through the pedicle. After vertebroplasty completion, transpedicular screws were inserted at D10-D11-L1-L2. Laminectomy and decompression were performed, with minimal blood loss observed by the effect of vertebroplasty. Favorable postoperative course was noticed, the patient recovered full motor power one week after surgery.

DISCUSSION

VH is the most common benign lesion of the spine, characterized by proliferation of blood capillary and expansion of blood sinus. Autopsies and radiographic studies indicate up to 10-12% incidence among population [15]. VH is usually discovered accidentally, as it is asymptomatic, without aggressive behavior. When symptomizing, these lesions tend to be insidious in course, causing neurological defects over long intervals, and rarely become rapidly progressive [14].

VH is basically a lesion of the vertebral body, with less common involvement of pedicle and posterior elements. It is typically located in the dorsal and lumbar lesions of the spine, with approximately 33% of multiplicity [11]. Clinically, VH’s are classified to three types, according to the Enneking staging system: stage I is latent with no symptoms (i.e., accidentally discovered); stage II is associated with active bony destruction leading to pain; and stage 3 is aggressive VH, whereby patients present with neurological deficit due to epidural hematoma and/or soft tissue involvement [13].

Although there is no evidence-based algorithm for management of aggressive VH due to rarity of this incidence, it is not indicated to perform surgical decompression unless the patient is neurologically compromised, which is the chief distinction between grade II and grade III. In this study, three patients of grade II lesions underwent only minimally invasive approach either vertebroplasty or TAE; on the other hand, grade III lesions underwent surgical decompression. Although Hekster et al. [18] described the decompressive effect of embolization, in this series any case presented with neurological manifestations underwent surgical decompression with or without embolization.

Although the vertebral body is the most affected part by VH, only posterior surgical procedures were adopted in our practice, anterior approach for corpectomy and fixation was performed in previous reports [3,27,28]. The aim of anterior approaches is to achieve spine stability rather than radical excision which is not sought in these cases due to the benign nature of the pathology, the choice of surgical procedure in this study is concomitant with previous reports [8,17,22] which confirmed the long term efficiency of vertebroplasty for anterior column augmentation in cases of VH, these results out weights the surgical risk for anterior approaches, especially for thoracic and lumbar spine; the most common sites.

Pre-operative embolization is generally safe, with very rare adverse events reported in the literature. The most serious complication is unintended release of emboli causing vascular blockage, potentially leading to spinal cord ischemia, heart attack, stroke, or occlusion of the peripheral arteries. Spinal cord ischemia is particularly concerning because it can result in paralysis, loss of bowel and bladder control, sexual dysfunction, and sensory deficits. This risk arises when emboli inadvertently enter radiculomedullary arteries not clearly visible on pre-embolization angiograms, due to contrast preferentially flowing into the tumor’s high-flow vessels. Another risk factor is embolization material refluxing through unrecognized intersegmental artery anastomosis, which can also cause spinal ischemia [1].

Pre-operative thorough analysis of the spinal angiogram is essential to identify radiculomedullary and spinal arteries, as well as potential intersegmental anastomosis, a step that is essential to minimize the mentioned complications. Embolization is generally avoided if a radiculomedullary artery is close to tumor-feeding vessels, or if these vessels supply both the Adamkiewicz artery and hemangioma [25].

Relative contraindications include identification of radiculomedullary arteries through segmental connections, arteriovenous shunting within the tumor, renal failure, or severe coagulopathy. Alternative methods, such as transpedicular n-Butyl Cyanoacrylic Acid (nBCA) embolization, can be adopted when vasculature of the hemangioma is not ideal for TAE [12]. Additional potential complications include allergic reactions to embolizing agents, and bleeding from early revascularization, due to the rapid breakdown of the embolization materials.

Currently, there is no agreement on the most effective embolic agent for treating VHs. Various agents are commonly used, including Gelfoam, liquid embolic agents like Onyx, particulate agents such as polyvinyl alcohol particles, mechanical occlusion devices like coils, NBCA, and sclerosing agents such as ethanol. Some studies suggest favorable outcomes with ethanol embolization, while others highlight higher complication rates, such as spinal cord injury, Brown-Sequard Syndrome, osteonecrosis, asystole, vertebral collapse, transient neurological deterioration, and hemodynamic instability. Transient paraparesis is a reported potential complication after Gelfoam embolization, while paralysis has been observed following polyvinyl alcohol particles [23]. Alcohol is reported to be safe, with a preference over onyx due to economic factor [27]. In this study, pre-operative embolization was performed using particles in all cases who underwent the procedure.

Combining preoperative embolization with vertebroplasty has been successfully used to reduce intraoperative bleeding. This approach is advocated because arterial embolization blocks feeding vessels without eliminating the tumor’s capillary or cavernous channels. Vertebroplasty can address the residual vascular bed post-embolization, particularly in challenging cases where embolization is technically difficult, such as when the artery of Adamkiewicz originates from an intercostal artery or in the presence of tortuous vessels. Furthermore, vertebroplasty helps strengthen the anterior column, thereby lowering the risk of vertebral collapse in vertebrae weakened by hemangiomas. Complications associated with vertebroplasty are uncommon, but can include the formation of a dense cast around the tumor’s epidural component, and cement leakage into the spinal canal [2].

Radiotherapy alone as a line of management of acute viral hepatitis (AVH) is reported in small case series and single case reports [4,5,6,7]. Although radiotherapy is usually safe and effective for aggressive VH, it has some inherent drawbacks. For instance, neurological recovery is slower for radiotherapy when compared to surgery where recovery is nearly 100%. A major multi-center study of seven German institutes reported success rate of approximately 90.5% of patients with symptomatic VH who were treated with radiotherapy solely after a median of 68 months follow-up [19]. The study also included patients with neurological deficits, but neurological recovery was only recorded among 79.2% of such patients. Another study of 137 patients reported symptoms relief within 18 months after radiotherapy, while neurological deficit was not discussed clearly in this study [21].

Stereotactic radiosurgery was studied as well by Zhang et al in a small case series of five patients, improvement was achieved for four patients over 12-month period [30].

Radiation myelitis is a feared complication of radiotherapy, especially for patients with spinal cord compression, as proven by reports on neurological deterioration after radiotherapy for patients with more than 40% spinal canal encroachment [29].

In our experience, radiotherapy was not one of the lines of management due to slow course of improvement and the availability of pre-operative embolization which reduces the risk of surgery. The choice of the appropriate line of management is decided regarding lesion location and aggressiveness. Typically, VH involves the whole body of vertebrae with or without posterior element involvement, thus jeopardizing the integrity of the stability of the spine. Surgical decompression is indicated for cases with neuronal element compression, while fixation is indicated for instability and decided for each case solely.

The high vascularity of VH’s makes surgical intervention highly susceptible to significant blood loss, as discussed previously, therefore pre-operative embolization is an important concept for management, as it decreases blood loss [16,24]. Pre-operative embolization is a means of blood loss reduction and also identification of feeder vessels, to prevent iatrogenic injury of cord blood supply [9].

Our results and adopted scheme of management are in line with a recently published meta-analysis since its conclusion confirmed that the decision to decompress the spinal canal or not, with or without adjuvant line, is strictly based on neurological status of patient at presentation, as non-surgical lines of management can be used solely only for patients who are neurologically intact at presentation [26].

CONCLUSIONS

Aggressive vertebral hemangioma is a rare pathology with no clear algorithm for management, however, surgical decompression is indicated for cases with neural canal encroachment. Management of each case is individually tailored according to clinical and radiological presentation.

Pre-operative embolization and/or vertebroplasty are useful tools to decrease intra-operative bleeding of such a vascular pathology in cases undergoing open surgical procedures. Vertebroplasty as a single line of management is useful for intractable pain cases with no neural compression.

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