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International Journal of Surgery Case Reports logoLink to International Journal of Surgery Case Reports
. 2023 Mar 22;105:108027. doi: 10.1016/j.ijscr.2023.108027

A less aggressive approach to the treatment of aggressive vertebral hemangioma of the thoracic spine: A case report and literature review

Seyed Reza Mousavi a,b, Somayeh Akbari c, Alireza Rasekhi d, Ali Kazeminezhad e, Mohammadhadi Amir Shahpari Motlagh a, Sanaz Taherpour a,⁎
PMCID: PMC10073888  PMID: 36965444

Abstract

Background

Different treatments are available for aggressive vertebral hemangioma [AVH], but a consensus is yet to be reached about the best therapeutic approach.

Purpose

To explore the possibility that selecting a less aggressive therapeutic approach for AVH decreases the intraoperative and postoperative complications while providing similar clinical, radiographic, and disease-free survival results to more aggressive therapeutic methods.

Study

We report the case of an AVH of the thoracic spine at the T5 level, treated via perioperative selective arterial embolization plus surgical decompression via laminectomy and reconstruction with polymethylmethacrylate (PMMA) vertebroplasty.

Patient

The patient was a 17-year-old male referred to our center with the chief complaint of low back pain from two months earlier, without any response to analgesics, and with neurologic manifestations as paraparesis (one month) and progressive sphincter problems (one week). Upon imaging, the impression was an aggressive spinal tumor with cord compression.

Outcome measures

After the operation, the patient's pain immediately improved, and his neurologic manifestations progressively improved.

Results

The patient started walking with help about three weeks after the operation. Roughly six months later, he achieved a complete neurological recovery. At present, about two years following the operation, he has a normal life without any neurological problems.

Conclusion

Based on our experience with AVH, the selection of less aggressive therapeutic approaches such as perioperative diagnostic angiography and selective embolization decrease the intraoperative and postoperative complications like intraoperative bleeding and neurological injury, while achieving similar clinical, radiographic, and disease-free survival outcomes to more aggressive therapeutic methods.

Keywords: Aggressive vertebral hemangiomas, Vertebroplasty, Embolization

Highlights

  • •

    Vertebral hemangioma [VH] is a benign vascular spine tumor.

  • •

    Explore a less aggressive therapeutic approach for AVH

  • •

    Decreases the intraoperative and postoperative complications

1. Introduction

1.1. Natural history

Vertebral hemangioma [VH] is a benign vascular spine tumor derived from the vascular endothelium of capillaries and veins, occurring in 10 to 20 % of the general population. VH is incidentally diagnosed in most cases and commonly involves the thoracolumbar region [1], [2].

1.2. Symptoms

Symptomatic VH occurs in 0.9–1.2 % of cases. Nonspecific symptoms include localized pain, radicular pain, and myelopathy [2]. VH is classified into three types based on imaging, histology, and clinical behavior [3]:

  • ‐

    Typical hemangioma: The computerized tomography (CT) scan reveals a thick vertical trabecula, while magnetic resonance imaging (MRI) reveals a hyperintense lesion on both the T1W and T2W modalities. These asymptomatic lesions are incidentally diagnosed in most cases [3].

  • ‐

    Atypical hemangioma: This type is also asymptomatic, though the MRI reveals variable signals because of the presence of fat, vessels, and interstitial edema.

  • ‐

    Aggressive hemangioma: This type features pain and neurological manifestations of cord or nerve root compression secondary to the rapid growth and extension of VH beyond the vertebral body [3].

1.3. Histopathology

Vertebral hemangioma is a benign tumor composed of different blood vessels from capillary to cavernous size. Macroscopically, VH is a soft mass with a well-demarcated border and dark red color. Microscopically, it is composed of blood-filled vessels with a thin single-layer endothelial wall in an edematous stroma. These vessels permeate the bone marrow and trabeculae and create a secondary reactive phenomenon like adipose or fibrous involution [3].

1.4. Diagnostic methods

The best diagnostic modality in VH is the CT scan, which demonstrates the polka dot sign. However, MRI is the diagnostic modality of choice for clarifying tumor invasion to epidural space and soft tissues [3]. Angiography is a diagnostic and therapeutic modality in AVH. Angiography can help diagnosis and preoperative planning; it also reduces intraoperative bleeding if preoperative embolization is done [3].

Like most spinal tumors, the standard diagnosis of VH depends on the biopsy result [2], [3]. However, in 2019, Shehata Elhelf and colleagues, in a case report of a giant aggressive vertebral hemangioma (AVH), used the Tc-99 m red blood cell scan as a new diagnostic modality for differentiating between hemangiomas and potentially malignant spinal tumors [4].

1.5. Differential diagnoses

Diagnosis of an atypical VH is complex and challenging because it can mimic the characteristic features of a malignant primary or metastatic tumor of the spine [3]. Some differential diagnoses of typical VH include focal fatty replacement of bone marrow, Modic type II changes, and prior radiotherapy [3]. In atypical VH, metastatic lesions and focal bone marrow infiltration due to multiple myeloma are included in the differential diagnoses list [3]. In AVH, because of the variable radiographic features, there is a more heterogeneous list of differential diagnoses: solitary bone plasmacytoma, metastasis, chordoma, lymphoma, and epithelioid hemangioendothelioma [3].

1.6. Treatment

Different therapeutic methods for AVH include transarterial embolization, direct embolization with ethanol, radiotherapy, vertebroplasty, and decompression and/or spondylectomy with or without preoperative embolization [3], [4], [5]. Based on a systematic review study in 2015, preoperative embolization in AVH decreases intraoperative bleeding and is highly recommended [6].

Treatment of VH depends on its symptoms. The common treatment of choice in AVH with refractory pain without neurologic manifestations is vertebroplasty and kyphoplasty, with good pain control results [2], [3]. In cases with neurological compromise, decompression of the cord and nerve roots with tumor resection is obligatory [2], [3].

Radiotherapy is frequently a postoperative adjuvant treatment for the prevention of tumor recurrence, especially in cases with subtotal resection of AVH [3]. Wang et al. showed that isolated radiotherapy was effective and safe in cases without cord compression and neurological compromise [7].

1.7. Outcome

In a 2021 systematic review of 535 patients with VH (diagnosed based on Odom's criteria), the most common surgical methods were stand-alone decompression (48.4 %), instrumented fusion (41.5 %), and spondylectomy (10.1 %). The study reported excellent treatment results in 81.7 % of cases, though those who presented with a focal neurological deficit or myelopathy had less favorable outcomes. Also, embolization did not have any effect on the outcome [1].

Basically, this tumor rarely presents with aggressive behavior and rarely compresses the cord and root. Because of the rarity of AVH, treatment approaches for this type of VH are complex and challenging without any consensus on the optimal treatment method. Clinical trials that compare different treatment methods are yet to be conducted.

2. Case presentation

2.1. Clinical presentation

The patient was a 17-year-old male without any underlying disorders, presenting with refractory low back pain since two months earlier, inability to walk since one month earlier, and sphincter problems since one week earlier. The family history was not significant. On imaging, there was an aggressive spinal tumor with cord compression at the level of T5 with the polka dot sign and thick bony trabeculae on the axial and sagittal CT scans, respectively (Fig. 1). On MRI with gadolinium injection in the vicinity of the involved vertebra, there was an extension of the aggressive bony lesion to the epidural space and in the rostrocaudal direction, indicating the high probability of a vascular tumor such as AVH (Fig. 2). The bony lesion was hypointense on T1W-MRI and hyperintense on T2W-MRI (Fig. 2), with positive enhancement with gadolinium (Fig. 3). There was no posterior vertebral arch involvement, vertebral body fracture, spinal instability, and sagittal and/or coronal malalignment. The patient was referred to our neurospine surgery referral center with the impression of a vascular tumor of the thoracic spine.

Fig. 1.

Fig. 1

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Computer Tomography (CT) image of the thoracic spine before surgery. The lesion in the T5 vertebral body is characterized by thick bony trabeculae in the sagittal (A) and coronal (B) views and the polka dot sign in the axial (C) view.

Fig. 2.

Fig. 2

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Magnetic Resonance Imaging (MRI) of the thoracic spine before surgery. A T5 vertebral body lesion is seen with a low signal in T1 (A) and a high signal in T2 (sagittal B and axial C), expanding to the epidural space from T1 to T7.

Fig. 3.

Fig. 3

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Magnetic Resonance Imaging (MRI) of the thoracic spine with gadolinium injection before surgery. The tumor lesion and its expansion to the epidural space are enhanced. Axial (A) and sagittal (B) views are shown.

On physical examination, there was paraparesis as 3/5, sensory level from T7 below, a sphincter problem as urinary retention, a positive Babinski test, and hyporeflexia.

Considering the male gender of the patient, and consequently, the absence of the risk of disease eruption due to hormonal causes and the absence of pregnancy risk factors, as well as the stability of the spine and the absence of involvement of the posterior vertebral arch, our treatment plan to treat the tumor and reduce its recurrence included spinal cord decompression, epidural pathology resection, and vertebroplasty. Due to the normal preoperative alignment of the spine and the spinal stability during the surgery, the instrumentation and fixation of the spine were waived. Radiotherapy was not recommended because of the risk of radiation-induced myelopathy, especially in young adults, and the low risk of recurrence in this patient due to intraoperative vertebroplasty.

The treatment steps were as follows:

A selective diagnostic-therapeutic spinal angiography was emergently performed. After preparing and draping, under local anesthesia a Judkins catheter was passed to the left T7 intercostal artery via the femoral approach. Injection revealed a large vascular lesion extending from T5 to T7. The vessel was super-selectively catheterized using a Rebar microcatheter after the passage of a hydrophilic microwire. This area was embolized by polyvinyl alcohol (PVA) and then completely occluded using a 1:1 mixture of glue (cyanoacrylate) and lipiodol. The picture after embolization revealed complete occlusion of the main trunk and side branches (Fig. 4). Then, the patient became a candidate for urgent operation.

Fig. 4.

Fig. 4

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Spinal angiography and super-selective embolization of the main arterial feeder of the vertebral body vascular lesion: pre-embolization (A), during the procedure (B), and post-embolization (C).

2.2. Operative procedure

After thoroughly explaining the patient's condition along with our therapeutic plan and its alternatives to the patient and his parents, written consent to perform a high-risk operation was obtained.

In the operation room, under general anesthesia, preparing and draping were done in the prone position with consideration of all precautions about positioning. The operation site was marked under fluoroscopic guidance as T4 to T6. After incision of the skin, subcutaneous tissue, and fascia, a subperiosteal dissection of paravertebral muscles was done. For decompression of cord, bilateral laminectomy from T4 to T6 was performed, and the epidural lesion was removed. A biopsy was taken and sent for a frozen section, revealing an AVH. The postoperative pathology result was the same (Fig. 5). At this stage, we selected a less aggressive intraoperative trans-pedicular vertebroplasty with the injection of polymethylmethacrylate (PMM) in the body of T5. In the end, the site of operation was closed with meticulous hemostasis. The operation did not have any complications.

Fig. 5.

Fig. 5

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Histopathology of the lesion (A and B). Sections show a vascular tumor located between normal bone trabeculae (*). The lesion consists of many small and intermediate-size spaces lined with endothelial cells and filled with red blood cells (arrowheads) (×100, ×400, Hematoxylin and Eosin stain). (For interpretation of the references to color in this figure legend, the reader is referred to the web version of this article.)

2.3. Postoperative course

After sterile dressing of the operation site and changing position to a supine state, the patient was transferred to the recovery ward. The patient's pain was immediately improved after operation, and the postoperative imaging study showed adequate cord decompression and vertebroplasty spine reconstruction (Fig. 6). About three weeks post-operation, the patient's neurological problems progressively improved. After six months, complete neurological recovery was acquired without any motor, sensory, or sphincter problem. Based on the pathology result that showed a benign lesion and for prevention of radiotherapy complications, radiotherapy was withheld.

Fig. 6.

Fig. 6

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Plain radiography and Computed Tomography (CT) of the thoracic spine after surgery. Successful posterior decompression with laminectomy and vertebroplasty of the T5 vertebral body is evident in the anterior-posterior (A), lateral (B), sagittal (C), coronal (D), and axial (E) views.

At present, after two years of regular follow-up and imaging, there is no recurrence of the tumor, and the patient is pain-free with complete neurological recovery (Fig. 7).

Fig. 7.

Fig. 7

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Magnetic resonance imaging (MRI) of the thoracic spine after surgery. After two years of follow-up, no evidence of local tumor recurrence was observed. T1 phase (A); T2 phase (sagittal B and axial C).

We obtained informed consent from patient and his parents for the publication of this study.

3. Discussion

Chopra and colleagues, in a recent case report of a cervical AVH in association with a systematic review of the literature, described the surgical outcomes for VH as predominantly favorable, though aggressive VHs had the potential to cause significant residual postoperative neurological morbidities [1]. Pinheiro Santos, in another literature review, described four cases of AVH treated conservatively. During follow up, two developed acute neurological problems and required emergent operations. One patient did not have any progression in tumor size and manifestations for six years, and the final patient was under treatment with propranolol and did not need an operation for 18 months [5].

Patel and colleagues, in a cohort study in 2021, investigated the treatment method of choice and overall survival in 102 patients with AVH. This study showed that surgical resection and adjuvant radiotherapy led to best overall survival results within an average follow-up time of 1.94 years [8].

There are various surgical methods for treating AVH. Here, we discuss some of the studied methods.

3.1. Total spondylectomy with spinal stabilization approach

Tutar and colleagues in 2016 selected the total spondylectomy in association with anterior and posterior spinal fixation approach for thoracic AVH [9], and Ogawa and colleagues in 2015 accepted this approach as an acceptable and decisive treatment without adjuvant radiotherapy [10]. Brunette-Clément and colleagues reported a pediatric case of thoracic AVH that was treated via en bloc resection with total spondylectomy. According to this study, radical operation in pediatric cases was preferred because of total resection of the lesion, lack of requiring postoperative radiotherapy, 360 degrees cord decompression, and better cord blood supply in the pediatric group [11].

Handa and colleagues reported 15 cases of AVH treated between 1996 and 2018 and showed acceptable results with preoperative embolization and total resection of the tumor. However, subtotal tumor resection and decompression method was cited as a palliative operation, with reoperation and resection of the recurrent tumors sometimes required on the long-term follow-up [12].

On the other hand, Vasudeva et al., in a retrospective study of five patients with AVH, showed that total or subtotal tumor resection in association with vertebroplasty or adjuvant radiotherapy in AVH was adequate. En bloc resection had similar results with increased morbidity [13].

3.2. Decompression, stabilization and intraoperative vertebroplasty approach

Hu et al. performed a retrospective study on five patients with thoracic AVH. Patients were operated as decompressive laminectomy, transpedicular fixation, and vertebroplasty [14]. They attained similar results as a retrospective study by Wang et al. (2017) on 39 cases of AVH treated with decompression, stabilization, and intraoperative vertebroplasty. This latter treatment method effectively improved the patients' neurologic status because of cord decompression, with the vertebroplasty decreasing the intraoperative bleeding and tumor recurrence rate [15].

Huang et al., in a retrospective study of 17 patients with thoracic AVH, showed that in patients with thoracic AVH in Enneking stage 3, the best treatment method was preoperative embolization, vertebroplasty, decompression, and internal fixation [16].

Chen et al. reported that the combined treatment approach of posterior decompression, vertebroplasty, and posterior fixation was an acceptable method, and they added that adjuvant radiotherapy decreased tumor recurrence rate [17]. Rai and colleagues also acquired the same results [18]. Finally, Pratheep et al., in a retrospective study of 21 patients with AVH, showed that the best treatment method in AVH is decompression and posterior fixation with vertebroplasty and intralesional tumor resection, and concluded that this treatment method was not only a less invasive method compared to total resection but also provided good clinical and neurologic results with a lower tumor recurrence rate [19].

Girardo et al. performed a retrospective study about less invasive treatment methods like posterior short segment stabilization, polyvinyl alcohol microparticles embolization, and poly methyl methacrylate. The researchers demonstrated less complications and bleeding, good clinical results, and improvement of neurological manifestations [20].

3.3. Minimally invasive approaches

Srinivasan et al., in 2021, performed a retrospective study about transpedicular embolization with ethanol as a less invasive treatment modality in AVH. They concluded that transarterial embolization prior to direct transpedicular embolization with ethanol could prevent untoward effects of the latter method like cord ischemia [21].

Cianfoni and colleagues, in 2014, described a 78-year-old patient with an AVH of the T8 and T9 levels, revealing that the percutaneous injection of ethanol into the vertebral body was a successful method. Three months later, the epidural component of the lesion had shrunk, and the neurologic symptoms had improved [22].

Pinto and colleagues, in 2017, reported two cases of AVH with onyx transpedicular injection and showed this method was better than surgery and adjuvant radiotherapy [23].

Papadokos and colleagues, in 2020, reported a case of thoracic AVH with paraparesis and refractory pain that was treated with the less invasive method of tubular laminotomy and percutaneous vertebroplasty. Six months later, the patient did not have any neurologic problems or disease recurrence [2]. Shamhoot et al., in a retrospective study of 9 cases of AVH, showed that surgical decompression with simultaneous vertebroplasty was an effective and safe treatment method [24].

Sedeno et al. published a case report study about a new therapeutic approach in AVH as intraoperative radiotherapy in association with vertebroplasty. They reported that this approach was safe and effective [25].

Kaoudi et al. reported a case of sacral AVH with canal compromise that was treated with robot-assisted radiofrequency ablation [2016] and demonstrated the usefulness of this method as an alternative treatment for a case of AVH that could not be treated via the usual manner [26].

In 2020, Xuxu and colleagues retrospectively studied 13 geriatric cases of AVH. They showed that a multimodality approach involving intraoperative absorbable gelatin sponge injection was safe and effective [27].

Canbay et al., in a case report, introduced a new treatment method for AVH that included radiofrequency ablation, injection of a hemostatic agent (FLOSEAL, Baxter, USA), and bone autograft placement in the affected vertebral body. This treatment was effective for decreasing intraoperative bleeding and strengthening the vertebral body [28].

3.4. Preoperative embolization

Because of the vascular nature of AVH, some precautions must be considered during the use of any surgical modality. In 2021, a retrospective study by Xu and colleagues of three groups of AVH patients [two groups included 16 patients and one group included 19 patients] with different therapeutic approaches showed that preoperative embolization and vertebroplasty in association with any surgical approach was effective and decreased intraoperative bleeding [29].

In a 2021 case report, Nagashima and colleagues introduced a new and less invasive therapeutic approach for AVH as preoperative embolization and gross total resection of the tumor with a real-time navigation-guided drilling technique. In the one-year follow-up, the patient did not have any neurological deficits or tumor recurrence [30].

Corniola et al., conducted a retrospective study of five patients with AVH and examined the demographic characteristics, manifestations on arrival, tumor location, use of preoperative embolization, duration of operation, amount of intraoperative bleeding, and duration of follow-up. They showed that one-stage holistic treatment was a safe, effective method for treating AVH, and preoperative sclerotherapy and/or intraoperative sodium tetradecyl sulfate usage as a sclerosing agent was effective and safe [31].

3.5. Treatment approaches in pregnancy

Pregnancy is a known etiology for transforming a typical hemangioma to an AVH, frequently during the third trimester [32]. Unlike the common location of AVH in non-pregnant individuals, this tumor tends to involve the upper thoracic spine in pregnant individuals [33], [34]. Rarely, progressive myelopathy and a pathological fracture of the involved vertebra can occur at any time during pregnancy, delivery, or in the postpartum period. However, early diagnosis and timely treatment and decompression can prevent morbidity [33], [34].

The approach to treating AVH in pregnancy is based on paying attention to both maternal health (including assessment of pain, signs of neurological deficits, and radiographic evidence of cord and/or nerve root compression) and fetal health. These two issues should be taken into consideration throughout conservative or surgical treatment. In particular, radiotherapy is contraindicated in these patients, and embolization is also unsafe due to the possibility of vascular damage and exposing the fetus to radiation [34].

Various surgical methods can be performed in this group, but due to the risks of intraoperative bleeding and its side effects for the mother and fetus, the choice of surgical approach should be made individually for each patient [33], [34]. The timing of surgical intervention in pregnancy is still controversial because many patients' symptoms improve spontaneously after delivery [34].

Two key aspects in choosing the time of surgery are the week of pregnancy and the severity of neurological symptoms. Based on the therapeutic algorithm of Chi et al., if the patient is at 36 weeks of gestation or above, conservative treatment is recommended until delivery, and if the neurological symptoms are progressive, labor induction and then tumor treatment is recommended. Between weeks 32 and 36, conservative treatment is recommended for mild to moderate neurological symptoms, while surgical treatment is only indicated in case of severe neurological symptoms or progression of symptoms. Before 32 weeks of gestation, emergency decompression treatment is performed only for cases with severe neurological symptoms (e.g., paraplegia). In all cases of interventional treatment, continuous monitoring of the fetus is necessary [34].

3.6. Which treatment method to choose?

Delabar and colleagues reported the results of treatment of AVH in 27 cases and showed that multimodality treatment could be effective and safe [35].

Based on our experience and retrospective studies, minimally invasive techniques are effective in asymptomatic AVHs without neurological deficits. In symptomatic AVH, a multimodality one-stage approach like preoperative embolization, posterior decompression, subtotal resection, and intraoperative vertebroplasty is an effective method because it decreases intraoperative bleeding, reduces operative complications, is less invasive, and can decrease tumor recurrence rate with vertebroplasty usage, produce excellent neurological and clinical results, and decrease the morbidity rate.

4. Conclusion

Because of the rarity of AVH, clinical trials are yet to be available for comparing treatment methods, and all data are from retrospective studies and personal experiences. There is still no consensus about the ideal treatment approach for AVH, so further studies are needed.

Consent

All identifying information is hidden and undisclosed. Consent was obtained from the patient for publication.

Ethical approval

All procedures compliance with ethical standards.

Funding

This work was not source of funding.

Author contribution

SRM involved in writing of the manuscript, study design and data analysis. SA involved in data collection, data analysis, data interpretation, literature search andgeneration of figs. AR involved in study design, data collection, data analysis, data interpretation and literature search. AK involved in writing of the manuscript, study design, data collection and data analysis. MASM involved in study design, writing of the manuscript, data analysis, data interpretation and literature search. ST involved in writing of the manuscript, study design, data collection and data analysis.

Guarantor

Seyed Reza Mousavi & Sanaz Taherpour.

Registration of research studies

Not applicable.

Declaration of competing interest

No conflict of interest.

Acknowledgement

The authors would like to thank Dr. Amir Reza Dehghanian for improving the manuscript and for his technical assistance.

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