Neurosurg Rev DOI 10.1007/s10143-015-0616-4

CASE REPORT

Surgery in extensive vertebral hemangioma: case report, literature review and a new algorithm proposal Roberto Tarantino & Pasquale Donnarumma & Lorenzo Nigro & Roberto Delfini

Received: 23 January 2014 / Revised: 26 April 2014 / Accepted: 16 November 2014 # Springer-Verlag Berlin Heidelberg 2015

Abstract Hemangiomas are benign dysplasias or vascular tumors consisting of vascular spaces lined with endothelium. Nowadays, radiotherapy for vertebral hemangiomas (VHs) is widely accepted as primary treatment for painful lesions. Nevertheless, the role of surgery is still unclear. The purpose of this study is to propose a novel algorithm of treatment about VHs. This is a case report of an extensive VH and a review of the literature. A case of vertebral fracture during radiotherapy at a total dose of 30 Gy given in 10 fractions (treatment time 2 weeks) using a linear accelerator at 15 MV high-energy photons for extensive VH is reported. Using PubMed database, a review of the literature is done. The authors have no study funding sources. The authors have no conflicting financial interests. In the literature, good results in terms of pain and neurological deficits are reported. No cases of vertebral fractures are described. However, there is no consensus regarding the treatment for VHs. Radiotherapy is widely utilized in VHs determining pain. Surgery for VHs determining neurological deficit is also widely accepted. Perhaps, regarding the width of the lesion, no indications are given. We consider it important to make an evaluation before initiating the treatment for the risk of pathologic vertebral fracture, since in radiotherapy, there is no convention regarding structural changes determined in VHs. We propose a new algorithm of treatment. We recommend radiotherapy only for small lesions in which vertebral stability is not concerned. Kyphoplasty can be proposed for asymptomatic patients in which VHs are small and in patients affected by VHs determining pain without spinal canal invasion in which the VH is small. In patients affected

R. Tarantino : P. Donnarumma : L. Nigro (*) : R. Delfini Department of Neurology and Psychiatry, Division of Neurosurgery, University of Rome BSapienza^, Viale del Policlinico 155, 00161 Rome, Italy e-mail: [email protected]

by pain without spinal canal invasion but in which the VH is wide or presented with spinal canal invasion and in patients affected by neurological deficits, we propose surgery. Keywords Vertebral hemangiomas . Radiotherapy . Neurological deficits . Pain

Introduction Hemangiomas are benign dysplasias or vascular tumors consisting of vascular spaces lined with endothelium [1]. Vertebral hemangiomas (VHs) are typically located in one of three anatomical locations: on the periosteal surface, within the cortex, or within the medullary canal. According to large autopsy and radiography studies [2], VH is a relatively common tumor, with an estimated incidence of 10–12 % in the general population. They are usually asymptomatic and often found incidentally by radiographs, CT, and/or MRI [1, 3, 4]. Only 3.7 % of VHs became active and symptomatic, and 1 % may invade the spinal canal and/or paravertebral space [2]. There are a few spinal lesions that have such a broad array of treatment options (observation, radiation therapy, radiosurgery, embolization, alcohol injection, vertebroplasty, kyphoplasty, laminectomy, intralesion resection, total en bloc spondylectomy, and combinationx of these treatments) with no consensus in the literature regarding which is most appropriate and in which clinical circumstances [5]. Roentgen therapy, introduced in 1930, was largely initiated as the result of the dismal early surgical experience. Nowadays, radiotherapy for VHs is widely accepted as primary treatment for painful lesions. In a few studies in the last 30 years [6–12], no details about location and width of the lesion are considered. Nevertheless, the role of surgery is still unclear. In this manuscript,

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we present the case of a 64-year-old man affected by thoracic back pain who developed a T6–T7 vertebral fracture during radiotherapy for an extensive vertebral hemangioma. Patient was abruptly referred to our institute and underwent a T6–T7 corporectomy with a titanium expandable cage implant via antero-lateral trans-thoracic approach. A review of the literature about vertebral hemangioma primarily treated with radiotherapy is done.

Case report Sixty-four-year-old man referred to another hospital with a 2month history of intermittent thoracic back pain. Neurological examination was normal. The MRI scan revealed an extensive lesion involving T6 and T7 vertebral body, extending through the intervertebral disc with spinal canal invasion and slight compression of the spinal cord. It was hypointense on T1weighted images and hyperintense on T2-weighted images (Fig. 1a, b). Basing on these radiological features and the honeycomb-like appearance, the lesion was interpreted as an aggressive hemangioma and treated with radiotherapy at a total dose of 30 Gy given in 10 fractions (treatment time 2 weeks) using a linear accelerator at 15 MV high-energy photons. At the 5th fraction, the patient reported severe thoracic back pain, weakness in dorsiflexion of his right foot and flexion of the thigh on the pelvis associated with generalized hyperreflexia and numbness at his right lower limb. The CT

scan showed a T7 pathologic fracture. The patient underwent surgery of posterior decompression and fusion with sublaminar hooks. No clinical improvements were documented. The postoperative CT scan showed thoracic kyphotic deformity (Fig. 2a–c). Abruptly, the patient was referred to our hospital. After executing thoracic MRI on T1- and T2weighted images (Fig. 3a, b), we performed a T6–T7 corporectomy with a titanium expandable cage implant via antero-lateral trans-thoracic approach. The patient was positioned in the left lateral decubitus. PESS and PEM were continuously monitored during surgery. A posterior-lateral right side thoracotomy was executed. After pleural opening, T6–T7 corporectomy was performed and pathologic tissue was taken for histological examination. Then the X-MESH® Expandable Cage (DePuy) was inserted between T5 and T8 soma and gently expanded (Fig. 4a). A lateral rod was applied to complete the fixation (Fig. 4b). No complications occurred in the post-surgical period. The postoperative CT scan showed the correct positioning of the cage and of the rod and screws and reduction of thoracic kyphosis (Fig. 5a–c). The histological exam showed bone and fibrotic elements; no presence of vascular elements was evident. One week later, the patient was transferred to a neuromotor rehabilitation institute. At 3 months follow-up, the MRI scan showed no recurrence of pathology; no further neurological deficits were evident; a partial recovery of the motor deficit with a reduction of the numbness at the lower limb was evident.

Discussion

Fig. 1 a, b On T2-weighted MRI, the lesion was heterogeneously hyperintense involving T6 and T7 vertebral body extending through the intervertebral disc with spinal canal invasion and slight compression of the spinal cord

The first case of vertebral hemangioma has been reported by Virchow in 1863. He described a patient who developed paraplegia during a lengthy discourse on Bangiomata.^ On autopsy, a tumor composed of dilated blood channels in the center of the 10th thoracic and 3rd lumbar vertebrae had been shown [13]. In 1963, Reeves presented the case of a 70-year-old woman treated with radiotherapy for a vertebral hemangioma at T4 presenting with pain and neurological deficits. Radiotherapy was chosen because of the patient’s age and general conditions. After the treatment, the patient showed improvement of the symptoms [14]. Patients with VHs may remain asymptomatic for years. Asymptomatic VHs are generally intraosseous well-delimited lesions. Painful VHs of the thoracic spine involving posterior elements, with cortical blistering or soft tissue extension, must be considered growing lesions and should result in a spinal cord compression. Spinal cord compression may be due to direct neural compression by epidural tumor tissue as an extension from the vertebral body or posterior elements, by expanded vascular bone, by epidural hematoma, by compression fracture of the hemangiomatous vertebra, or by anomalous vessels draining or feeding the lesion. In aggressive VHs the clinical onset of spinal cord

Neurosurg Rev Fig. 2 a–c Post-operative CT scan shows T7 pathologic fracture, posterior decompression, and fusion with sublaminar hooks. A thoracic kyphotic deformity was also evident

compression is often progressive over many months but may be sudden. Neck or back pain often precedes the neurological symptoms. Thoracic myelopathy is the most common presenting syndrome [2]. VHs usually do not impair the stability of vertebral bodies, but pathologic fractures may occur in cases with extensive vertebral bone involvement [15]. In typical cases, hemangiomas are homogeneous and can be diagnosed using CT and MRI [3]. In latent VHs, MRI shows increased signal intensity on both T1- and T2weighted images with altered hypointense areas. The

hyperintense areas represent fat, while the hypointense areas represent flow voids [3]. It has been illustrated that aggressive VHs usually produce a low signal on T1-weighted images and a high signal on T2 [2, 3, 16]. Low signal intensity on T1weighted MR imaging indicates a hypervascular lesion with the potential to compress the spinal cord [2]. The vertebral end plates are usually preserved, but extension into disc spaces and into neighboring ribs has been described. Basing on the patient’s lesion and symptoms, VHs could be classified into four categories: type I, latent (Enneking S1), mild bony destruction with no symptom; type II, active (S2),

Fig. 3 a, b T1- and T2-weighted MRI shows T7 vertebral fracture with pathologic tissue posteriorly dislocated and compressing the spinal cord. The lesion appears heterogeneously hypointense

Fig. 4 a The picture shows the expandable titanium mesh inserted between T5 and T8 soma and gently expanded. b The picture shows the lateral rod applied to complete the fixation

Neurosurg Rev Fig. 5 a–c The postoperative CT scan showed the correct positioning of the cage and of the rod and screws and reduction of thoracic kyphosis

bony destruction with pain; type III aggressive (S3), asymptomatic lesion with epidural and/or soft tissue extension; type IV aggressive (S3), neurological deficit with epidural and/or soft tissue extension [3, 17]. In the literature, there is no consensus regarding the treatment for VHs. Fox et al. propose only observation for type I VHs [2]. Percutaneous vertebroplasty, usually with polymethyl methacrylate (PMMA), is a relatively new, palliative technique of treatment of symptomatic hemangiomas. Jankowski et al. assert that patients with pathologic fractures of the vertebral body and persistent pain are qualified for percutaneous vertebroplasty or kyphoplasty, even if vertebroplasty is a contraindication in patients with hemangioma expansion into the spinal canal [18]. Sudden onset of neurological deficits qualifies the patient for imminent surgery. Doppman et al. for type III VHs emphasize the importance of early diagnosis and treatment [19]. On the other hand, Boriani et al. affirm that VHs are not really aggressive even with extracompartmental extension; hence, preventive treatments are not mandatory [17]. Treatment protocol for type IV VHs is more controversial. Reported treatments include radiotherapy [20], vertebroplasty, alcohol injection [19], embolization of the feeding arteries, surgery, and a combination of these treatments. Having VHs a benign histology even if CT and MRI features (osteolysis, vertebral body collapse, epidural extension, etc.) mimic those of primary and secondary malignant tumors, there are no evidences that total removal has a better prognosis [4]. Fourney et al. propose an intraoperative vertebroplasty after laminectomy to achieve direct visualization [5]. Dang et al. also affirm a decompression is

recommended only in cases with rapid progressive or severe myelopathy and recommend embolization of feeding vessel as a presurgical adjuvant treatment for reducing intraoperative hemorrhage [3]. The aim of surgery includes bony decompression and excision of the soft tissue components of the tumor that compress the neural elements. Different surgical techniques have been reported as laminectomy [2], corporectomy, spondylectomy and total en-bloc spondylectomy [1]. Multiple studies have demonstrated safe and effective results from preoperative embolization of vertebral hemangiomas. Smith et al. demonstrated the safety and effectiveness of transarterial embolization of symptomatic vertebral hemangiomas in eight cases using polyvinyl alcohol. In 2013, Yao et al. described four cases of transpedicular n-butyl cyanoacrylate (NBCA) direct-puncture embolization of spinal hemangiomas as a preoperative adjunct, facilitating resection of these high vascular tumors. Hurley et al. were the first to document the use of Onyx, transarterially, in the treatment of two cases of aggressive vertebral hemangiomas. Additional studies report use of Onyx via an arterial route and by direct puncture in the treatment of hypervascular head, neck, and spinal tumors. Advantages of Onyx embolization include a more controlled injection due to gradual precipitation in a centripetal fashion, allowing for slower and more accurate injections as well as penetration of very small caliber vessels. Careful attention must be paid to the initial diagnostic angiographic run prior to embolization to ensure opacification of a spinal pial artery such as the artery of Adamkiewicz which is not seen [21]. Radiotherapy has been suggested for patients with slight and slow progressive neurological deficit because the effects of

2 Pts

9 Pts

25 Cases (21 pt with 11 Males, 12 females 19 to 64 years single lesion, 2 pts with multiple lesions)

137 Cases (101 pts, of which 22 with multiple hemangiomas)

1 Pt

10 Pts

Faria [9]

Yang [7]

Miszczyk [12]

Reeves [14]

Jiang [6]

35 to 63 years

28 and 75 years

4 Females, 6 males

Female

34 to 72 years

70 years

Duration of the symptoms

Symptoms

2 Cervical, 14 thoracic, 9 lumbar

4 Dorsal, 5 lumbar

D4 and D4

2 Cervical, 7 thoracic, 1 lumbar

T3

4 months in average

14 months

Less than 6 months in 8 patients, 6 to 12 months in 2, 1 to 2 years in 9, 2 to 5 years in 4 patients 2 to 369 months (mean, 41.5)

From 2 months to 12 years

6 months

Not specified

4000 cGy in 4 weeks

Pain, weakness at lower limbs, loss of control of the bladder Mild neurological deficit

Not specified

2500 r

Back pain alone in 8; From 3000 to 4000 Pain plus numbness cGy in 4 to 6 weeks in limb in 5; pain, sensory impairment and paresis in 3; paraplegia in 7 All pain, 2 numbness, 2 From 8 to 30 Gy weakness of the limbs (mean, 22.9 Gy)

Good results in 8, in 2 weakness worsening

Recovery of neurologi al deficit, pain relief

Mean of percentage relief at 1, 6, 12 and 18 months were 60.5, 65.4, 68.3, 78.4 and 60.4 % respectively

5 of 7 paraplegic pts able to walk, pain in the back and numbness in the limbs relieved in 80 and 88 %, respectively

7 Pts, no pain; 1 pt, partial response, paraplegic pt recovered completely

Good improvement of the symptoms

Pain became tolerable from the third week of radiation, analgesics were no more required in any of them; muscle power gradually improved but recovery of sensory loss took a rather longer period

Dose of radiotherapy Outcome

8 Back pain, 1 paraplegia 3000 to 4000 cGy

Neurological deficit

5 Thoracic, 1 thoracolumbar, Average 10.6 months All pain, 1 paraplegia, 1 lumbar 5 paraparesis

Location

66 Females, 35 males From 19 to 76 years 11 Cervical, 33 upper thoracic, 40 lower thoracic, 51 lumbar, 2 sacral

7 Females, 2 males

1 Female, 1 male

Mean 36 years

McAllister [10]

5 Females, 2 males

7 Pts

Age

Aich K [8]

Sex

Patients

Author

Table 1 The table shows cases of VHs treated with radiotherapy reported in literature. Author, number of patients, sex, age, location, duration of the symptoms, symptoms, dose of radiotherapy, and outcome are exposed

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radiotherapy are not as prompt as desired for patients with progressive neurological deficits caused by cord compression [3]. Dang et al. affirm radiotherapy represents the first of choice of treatment for type II VHs [3]. On the other hand, some authors support the role of radiotherapy for the treatment of VHs. Rades et al. consider radiotherapy as the most common treatment for painful lesions [11]. Heyd et al. affirm that radiotherapy is easy and effective, and surgery is recommended if radiotherapy misses in 3 months time. They reported neurological symptoms are completely resolved in 79 % of their cases after radiotherapy [20]. In another retrospective multicenter study of about 24 cases presenting neurological symptoms, 20 had surgical intervention [3]. In radiotherapy for VHs optimal dose, fractionation protocol and time period necessary for a therapeutic effect are controversial. The problem of the dose–response relationship for painful vertebral hemangiomas is widely discussed in literature. Previously, the commonly used total dose in this situation was 20– 24 Gy delivered in 2 Gy fractions. In recent publications, the need of total dose increasing is frequently highlighted. A meta-analysis of 117 patients treated with radiotherapy in 19 centers clearly showed that the total dose of 36–44 Gy is more efficient than a total dose of 20–34 Gy [11]. A subsequent publication presenting similar dependency was an article by Heyd after analyzing 84 patients [20]; similarly in Miszczyk’s study, the superiority of total dose >34 Gy over the total dose than 3 cm has been proposed, and a novel algorithm of treatment that can guide the decision-making process is exposed (Fig. 6). We differentiate asymptomatic patients from symptomatic ones. For asymptomatic patients, it is important to evaluate the width of the lesion. For small lesions, clinical and radiological follow-up are suggested; for wide lesions, we consider appropriate kyphoplasty or radiotherapy as treatment of these lesions before becoming symptomatic. For symptomatic lesions, we distinguish lesions determining pain from those determining neurological deficits. The first ones are separated into two further groups: those without invasion of spinal canal and those with spinal canal invasion. Those without spinal canal invasion can be divided into two other different groups. Small painful VHs without invasion of the canal can be treated with radiotherapy or kyphoplasty plus follow-up, while for wide VHs, surgery is proposed. For lesions with spinal canal invasion, surgery is proposed. For symptomatic patients presenting with neurological deficits, surgery is mandatory with the purpose to decompress spinal cord and to stabilize the spine.

Conclusions Radiotherapy as primary treatment for painful vertebral hemangioma is widely utilized based on the radiologic features without taking a histological examination of the lesion. Studies demonstrate good results in terms of pain relief and neurological outcome, but no information are given about the width and the location of the lesion. Studies about reossification or fatty conversion of hemangioma after the radiotherapic treatment are in debate. For these reasons, we recommend radiotherapy only for small lesions in which vertebral stability is not concerned. There are some controversies regarding the use of radiotherapy and kyphoplasty; however, these treatments can be proposed for symptomatic patients in

which VHs are small and in patients affected by VHs determining pain without spinal canal invasion in which the VH is small. In patients affected by pain without spinal canal invasion but in which the VH is wide or presented with spinal canal invasion and in patients affected by neurological deficits, we propose surgery.

References 1. Acosta FL Jr, Sanai N, Cloyd J, Deviren V, Chou D, Ames CP (2011) Treatment of Enneking stage 3 aggressive vertebral hemangiomas with intralesional spondylectomy: report of 10 cases and review of the literature. J Spinal Disord Tech 24(4):268–275 2. Fox MW, Onofrio BM (1993) The natural history and management of symptomatic and asymptomatic vertebral hemangiomas. J Neurosurg 78(1):36–45 3. Dang L, Liu C, Yang SM, Jiang L, Liu ZJ, Liu XG, Yuan HS, Wei F, Yu M (2012) Aggressive vertebral hemangioma of the thoracic spine without typical radiological appearance. Eur Spine J 2(10): 1994–1999 4. Alexander J, Meir A, Vrodos N, Yau YH (2010) Vertebral hemangioma: an important differential in the evaluation of locally aggressive spinal lesions. Spine 35(18):E917–E920 5. Fourney DR (2012) Expert’s comment concerning grand rounds case entitled BAggressive vertebral hemangioma of the thoracic spine without typical radiological appearance^ (Lei Dang, Chen Liu, Shao Min Yang, Liang Jiang, Zhong Jun Liu, Xiao Guang Liu, Hui Shu Yuan, Feng Wei, Miao Yu). Eur Spine J 21(10):2000–2002 6. Jiang L, Liu XG, Yuan HS, Yang SM, Li J, Wei F, Liu C, Dang L, Liu ZJ (2013) Diagnosis and treatment of vertebral hemangiomas with neurologic deficit: a report of 29 cases and literature review. Spine J 7. Yang ZY, Zhang LJ, Chen ZX, Hu HY (1985) Hemangioma of the vertebral column. A report on twenty-three patients with special reference to functional recovery after radiation therapy. Acta Radiol Oncol 24(2):129–132 8. Aich RK, Deb AR, Banerjee A, Karim R, Gupta P (2010) Symptomatic vertebral hemangioma: treatment with radiotherapy. J Cancer Res Ther 6(2):199–203 9. Faria SL, Schlupp WR, Chiminazzo H Jr (1985) Radiotherapy in the treatment of vertebral hemangiomas. Int J Radiat Oncol Biol Phys 11(2):387–390 10. McAllister VL, Kendall BE, Bull JW (1975) Symptomatic vertebral haemangiomas. Brain 98(1):71–80 11. Rades D, Bajrovic A, Alberti W, Rudat V (2003) Is there a dose-effect relationship for the treatment of symptomatic vertebral hemangioma? Int J Radiat Oncol Biol Phys 55(1):178–181 12. Miszczyk L, Tukiendorf A (2012) Radiotherapy of painful vertebral hemangiomas: the single center retrospective analysis of 137 cases. Int J Radiat Oncol Biol Phys 82(2):e173–e180 13. Virchow R. Die krankhaften Gerschwulste. A. Hirschwald; 1863– 1867, 3, p 306–496 14. Reeves DL (1964) Vertebral hemangioma with compression of the spinal cord. J Neurosurg 21:710–712 15. Nguyen JP, Djindjian M, Pavlovitch JM, Badiane S (1989) Vertebral hemangioma with neurologic signs. Therapeutic results. Survey of the French Society of Neurosurgery. Neurochirurgie 35(5):299–303, 305–8. French 16. Urrutia J, Postigo R, Larrondo R, Martin AS (2011) Clinical and imaging findings in patients with aggressive spinal hemangioma requiring surgical treatment. J Clin Neurosci 18(2):209–212

Neurosurg Rev 17. Boriani S, Weinstein JN, Biagini R (1997) Primary bone tumors of the spine. Terminology and surgical staging. Spine 22:1036–1044 18. Jankowski R, Nowak S, Zukiel R, Szymaś J, Sokół B (2011) Surgical treatment of symptomatic vertebral haemangiomas. Neurol Neurochir Pol 45(6):577–582 19. Doppman JL, Oldfield EH, Heiss JD (2000) Symptomatic vertebral hemangiomas: treatment by means of direct intralesional injection of ethanol. Radiology 214(2):341–348 20. Heyd R, Seegenschmiedt MH, Rades D et al (2010) Radiotherapy for symptomatic vertebral hemangiomas: results of a multicenter study and literature review. Int J Radiat Oncol Biol Phys 77:217–225 21. Sedora-Roman NI, Gross BA, Reddy AS, Ogilvy CS, Thomas AJ (2013) Intra-arterial onyx embolization of vertebral body lesions. J Cerebrovasc Endovasc Neurosurg 15(4):320–325 22. Sakata K, Hareyama M, Oouchi A, Sido M, Nagakura H, Tamakawa M et al (1997) Radiotherapy of vertebral hemangiomas. Acta Oncol 36(7):719–724 23. Boschi V, Pogorelić Z, Gulan G, Perko Z, Grandić L, Radonić V (2011) Management of cement vertebroplasty in the treatment of vertebral hemangioma. Scand J Surg 100(2):120–124

Comments Siamak Asgari, Ingolstadt, Germany The authors present a case report about a patient with two-level midthoracic vertebral body lesions suspected as vertebral hemangiomas. In a foreign institution, percutaneous radiation therapy was performed. During this form of therapy, pathological fracture of one of the vertebral bodies occurred inducing severe back pain and neurological signs. Therefore, a dorsal ineffective stabilization with insertion of a sublaminar hook and rod system was performed. The authors themselves performed resection of the pathological vertebral bodies with antero-lateral vertebral reconstruction with a titanium lift device and plating. The paper discusses the role of radiation therapy, kyphoplasty, and open surgery in vertebral hemangioma disease. The authors gave information about the role of classical and percutaneous endovascular embolization procedures. Interestingly, the pathologist was not able to diagnose hemangioma in the reported case. The authors had to argue about their certainty about the diagnosis. In how many cases of vertebral hemangioma resections the histological diagnosis would be accurate? Their proposed management algorithm based on the experience of the largest series with observation of the natural course, radiation therapy, kyphoplasty, endovascular treatment, and surgeries in vertebral hemangiomas reported in the literature. Julio Urrutia, Santiago, Chile Vertebral hemangiomas (VHs) are dysplastic tissues or vascular tumors that are frequently found in routine imaging studies. They are histologically benign, but a small group can become clinically aggressive, producing bone destruction, pain, and compression of neural elements [1, 2]. No consensus has yet been obtained in the medical literature regarding the optimal management of aggressive VH; the treatment options include observation, radiotherapy, vertebral augmentation procedures (vertebroplasty/kyphoplasty), direct ethanol injection, embolization, and surgery (including laminectomy, intralesional resection, and total en bloc vertebral resection), as well as combinations of these treatments [3]. Tarantino et al. present us a case report of an interesting case of aggressive VH and a literature review on this topic. Although randomized trials, systematic reviews and meta-analyses represent the best clinical

evidence, sometimes that high-level evidence is not attainable, with case reports and case series representing the best available evidence. Since aggressive VH is a very unusual condition, this kind of reports and literature reviews provide a good teaching opportunity. For most VH (Enneking stages 1 and 2) magnetic resonance imaging (MRI) studies present increased signal intensity on both T1- and T2weighted images, with alternating hypointense areas; hyperintense areas represent fat and degenerated marrow, while hypointense areas represent thickened trabeculae. Conversely, as shown by Tarantino et al., aggressive VH usually exhibit less fat tissue, showing low signal intensity on T1-weighted images and high signal intensity on T2-weighted images [1, 2]. Although the importance of pathological diagnosis cannot be underestimated, it is not unusual to find only blood and necrotic tissue in vertebral biopsies patients with aggressive VH, and characteristic imaging may be the way to diagnose them, as happened with this patient. In this case report, the patient underwent radiotherapy as first treatment; the rationale for this treatment is supported on the vascular necrosis produced by radiation therapy; this allows bone to heal and can reduce pain. However, the patient developed a pathological fracture with cord compression and neurological deficit; the patient did not show neurological improvement after posterior decompression and stabilization; in such circumstances, a corpectomy and fusion (as the authors performed) should be warranted to recover the neurological function. This treatment allowed partial recovery of the motor deficit at last follow-up. In their article, Tarantino et al. also provided a thorough review of this unusual condition, and they proposed a new algorithm of treatment for aggressive VH. We must thank the authors for providing this simple and useful algorithm that should help to decide the best treatment in these unusual cases. However, in this algorithm, the role of embolization was not considered; embolization has not only been routinely used to reduce the intraoperative bleeding [1, 4] but it could also reduce the epidural soft tissue extension of the VH after interruption of the blood flow through the main feeding vessel, and it has been shown that embolization on its own can be successful at reducing neurological symptoms [2, 5]. In view of the scarcity of high-level evidence available on the treatment options for aggressive VH, future multicenter studies that could provide management recommendations based on better evidence should be warranted. References 1. Jiang L, Liu XG, Yuan HS, Yang SM, Li J, Wei F, Liu C, Dang L, Liu ZJ (2014) Diagnosis and treatment of vertebral hemangiomas with neurologic deficit: a report of 29 cases and literature review. Spine J 14:944–954. doi: 10.1016/j.spinee.2013.07.450 2. Urrutia J, Postigo R, Larrondo R, Martin AS (2011) Clinical and imaging findings in patients with aggressive spinal hemangioma requiring surgical treatment. J Clin Neurosci 18:209–212. doi: 10.1016/j.jocn. 2010.05.022 3. Dang L, Liu C, Yang SM, Jiang L, Liu ZJ, Liu XG, Yuan HS, Wei F, Yu M (2012) Aggressive vertebral hemangioma of the thoracic spine without typical radiological appearance. Eur Spine J 21:1994–1999. doi: 10.1007/s00586-012-2349-1 4. Acosta FL, Jr., Sanai N, Cloyd J, Deviren V, Chou D, Ames CP (2011) Treatment of Enneking stage 3 aggressive vertebral hemangiomas with intralesional spondylectomy: report of 10 cases and review of the literature. J Spinal Disord Tech 24:268–275. doi: 10.1097/BSD. 0b013e3181efe0a4 5. Hekster RE, Luyendijk W, Tan TI (1972) Spinal-cord compression caused by vertebral haemangioma relieved by percutaneous catheter embolisation. Neuroradiology 3:160–164

Surgery in extensive vertebral hemangioma: case report, literature review and a new algorithm proposal.

Hemangiomas are benign dysplasias or vascular tumors consisting of vascular spaces lined with endothelium. Nowadays, radiotherapy for vertebral hemang...
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