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ORIGINAL ARTICLE
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Single-center outcomes of image-guided radiotherapy in the management of vertebral hemangioma with daily kilovoltage cone-beam computerized tomography


 Department of Radiation Oncology, Gulhane Training and Research Hospital, University of Health Sciences, Kecioren, Ankara, Turkey

Date of Submission22-Feb-2019
Date of Decision08-Jan-2020
Date of Acceptance12-Apr-2020
Date of Web Publication28-Oct-2020

Correspondence Address:
Bora Uysal,
Department of Radiation Oncology, Gulhane Training and Research Hospital, University of Health Sciences, Etlik, Kecioren, Ankara
Turkey
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcrt.JCRT_133_19

 > Abstract 


Background: Vertebral hemangiomas are defined as benign proliferation of blood vessels. Vertebral hemangiomas are generally found incidentally by computerized tomography or magnetic resonance imaging; however, they may also cause pain and quality-of-life impairment in some circumstances with reference to their location and association with the spinal cord. In this study, we assessed the utility of image-guided radiation therapy (IGRT) in the management of patients with painful vertebral hemangioma.
Materials and Methods: Patients receiving IGRT for the management of painful vertebral hemangioma were evaluated. The total dose was 24 Gy delivered in 12 daily fractions. The verbal numeric scale (VNS) was used for the assessment of pain relief. The median follow-up duration was 13 months (range: 6–24 months).
Results: Median preradiotherapy VNS score was 8 (range: 6–10) and median postradiotherapy VNS score was 1 (range: 0–2) for the total 135 patients treated with IGRT at our department for painful vertebral hemangioma. Reduction in VNS scores after IGRT was statistically significant (P < 0.05).
Conclusion: Our single-center study revealed that IGRT resulted in substantial relief of pain from vertebral hemangioma. Randomized prospective multicenter trials are needed to shed light on the optimal management of patients suffering from pain due to vertebral hemangioma.

Keywords: Hemangioma, image-guided radiation therapy, vertebra



How to cite this URL:
Uysal B, Gamsiz H, Dincoglan F, Sager O, Demiral S, Dirican B, Beyzadeoglu M. Single-center outcomes of image-guided radiotherapy in the management of vertebral hemangioma with daily kilovoltage cone-beam computerized tomography. J Can Res Ther [Epub ahead of print] [cited 2020 Dec 3]. Available from: https://www.cancerjournal.net/preprintarticle.asp?id=299457




 > Introduction Top


Vertebral hemangiomas are typically found incidentally by computerized tomography (CT) or magnetic resonance imaging (MRI); however, they may also cause pain and quality of life impairment in some circumstances with reference to their location and association with the spinal cord. Vertebral hemangiomas are defined as benign proliferation of blood vessels. While presentation with severe symptoms is not typical, patients suffering from pain due to vertebral hemangioma need prompt management. Therapeutic options include resection, vertebroplasty, intravertebral injection, and radiation therapy (RT). RT can be delivered postoperatively or as an upfront treatment option for symptomatic palliation of pain due to the vertebral hemangioma.[1]


 > Materials and Methods Top


One hundred and thirty-five patients receiving image-guided RT (IGRT) for painful vertebral hemangioma at our department between January 2008 and January 2017 were assessed retrospectively. All treated patients suffering from pain due to vertebral hemangioma had an eastern cooperative oncology group performance status of 0–1, and no patients had previous RT history.

Informed consent of all patients were obtained before the treatment, and the study was performed in compliance with the Declaration of Helsinki principles. A verbal numeric scale (VNS) was used for the assessment of pain relief. VNS scoring was performed before and after RT for all patients. Patients were asked to score the magnitude of pain from 0 to 10 where score 0 corresponds to no pain and score 10 corresponds to severe pain.

Planning CT images were acquired to delineate the critical structures and gross tumor volume, clinical target volume (CTV), and planning target volume for all patients. IGRT plans were generated for RT with image guidance. Supine positioning was used for treatment. All 135 patients were immobilized in the supine position with the arms above the head and planning CT images with 5-mm slice thickness were acquired. The planning CT images were sent to the contouring station through network. Advantage Sim MD simulation and localization software (Advantage Sim MD, GE, UK) were used for the delineation of the organs at risk and treatment volumes.

All RT plans were generated using PrecisePLAN (Elekta, UK) treatment planning system. CTV coverage was maintained between 95% and 107% for all RT plans. Dose-volume histograms were thoroughly assessed by the treating radiation oncologist and radiation physicist before treatment.

All patients received a total dose of 24 Gy delivered with conventional fractionation in 12 daily fractions of 2 Gy each. RT was delivered by synergy (Elekta, UK) linear accelerator with the capability of daily online set-up verification under image guidance with kilovoltage cone-beam CT (kv-CBCT) and (X-ray Volumetric Imaging [XVI], Elekta, UK). All patients were treated after online verification of setup by the use of kV-CBCT and XVI.

Statistical analysis

Statistical Package for the Social Sciences, version 16.0 (SPSS, Inc., Chicago, IL) software was used for the data analysis with the level of significance set at P < 0.05. Age, gender, location, number of hemangiomas, RT dose, follow-up time, median VNS scores before, and after RT were comparatively assessed.


 > Results Top


Patient characteristics are shown in [Table 1]. Of the total 135 patients, 81 patients (60%) were female and 54 patients (40%) were male. The median age was 46 years (range: 37–72 years). The median follow-up duration was 13 months (range: 6–24 months). Vertebral hemangiomas were located at the cervical vertebrae in 18 patients (13.3%), at the thoracic vertebrae in 72 patients (53.3%), and at the lumbar vertebrae in 45 patients (33.4%). While 63 patients (46.7%) had vertebral hemangioma in a single location, 72 patients (53.3%) had vertebral hemangiomas at two different locations.
Table 1: Patient characteristics

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Median preradiotherapy VNS score was 8 (range: 6–10), and median postradiotherapy VNS score was 1 (range: 0–2) for the total 135 patients treated with IGRT at our department for painful vertebral hemangioma. Reduction in VNS scores after IGRT was statistically significant (P < 0.05).


 > Discussion Top


Vertebral hemangiomas are defined as benign proliferation of blood vessels. Unnecessary biopsy should be avoided since these lesions may have a considerable risk for hemorrhage. Vertebral hemangiomas present with the typical honeycomb and striped appearance on direct graphy. They may be visualized as iso/hyperintense in T1-weighted and hyperintense on T2-weighted MRI sequences. Vertebral hemangiomas are more frequently located in thoracic or upper lumbar spine compared to the cervical or lower lumbar spine. Cold vertebrae appearance may be visualized on 18-FDG-PET/CT, which is typical for hemangioma; however, it may also be seen in the setting of metastasis or patients with previous RT history.[2] The incidence of vertebral hemangioma is relatively high among the benign spinal tumors; however, the presentation with severe pain is not usual, with most patients being diagnosed incidentally on CT or MRI.

Management options for hemangioma include surgical interventions, RT, and embolization. An intralesional injection may also be used in selected patients.[3] CT-guided percutaneous transpedicular injection of alcohol has also been suggested as an alternative manat modality by Murugan et al. with encouraging treatment outcomes.[4] Nevertheless, optimal treatment for patients with vertebral hemangioma remains to be an area of the active investigation despite the predominant utility of RT and surgical interventions along with other alternative therapeutic options. The surgery is typically the primary mode of management in the presence of neurological deficits due to vertebral hemangiomas. Cauda equina is an unusual location for vertebral hemangiomas, but lesions in this region may behave aggressively, requiring prompt management.[5] Percutaneous vertebroplasty may be used for the selected patients with vertebral hemangiomas. RT may be suggested for the management of nonaggressive vertebral hemangiomas.[6] Surgical intervention with total en bloc spondylectomy may be used as an effective and tolerable treatment modality, particularly in the presence of neurological deficits.[7] Surgical management should be primarily considered for the management of larger vertebral hemangiomas and in the setting of the spinal canal invasion, however, RT may be used postoperatively in selected patients.[8] RT has also been suggested for the management of symptoms resulting from the spinal cord compression due to vertebral hemangiomas.[9]

Multilevel thoracic hemangiomas in the pediatric population are exceedingly rare and may present with epidural extension resulting in progressive neurological deficits. RT has been used for the adjuvant management of vertebral hemangiomas following decompressive laminectomies.[10] Neurological deficits have to be examined carefully for the differential diagnosis.[11] The utility of medication by the use of beta-blocker drugs has also been investigated for the pediatric population with promising results.[3] In the study by Uzunaslan et al., the use of propranolol as a beta-blocker drug has achieved pain relief in children.

Radiosurgery is another highly conformal RT option comprising short course and dedicated treatment with the primary advantage of Improved dose homogeneity and normal tissue sparing compared to three-dimensional conformal RT. CyberKnife technology has been utilized for the management of vertebral hemangiomas with encouraging treatment results and negligible toxicity in terms.[12],[13] In the study, including a total of 137 patients, 123 patients were treated with daily conformal fractionation, and 14 patients were treated with single-dose radiosurgery. The total dose range was 8–30 Gy delivered with daily conventional fractionation (2 Gy/each daily fraction) in 111 patients. High fractional and total dose resulted in improved pain relief.[14] Postoperative RT was used for the management of a vertebral hemangioma located at the T9 thoracic vertebra. RT was delivered after the embolization of the afferent vessels. Heyd et al. concluded that the total dose over 34 Gy was highly effective for the relief of pain and local control in vertebral hemangioma management.[15],[16]

CyberKnife technology is capable of improved normal tissue sparing and dose homogenization compared to conventional conformal therapies. Aksu et al. reported a 58-year-old patient with the epidural extension of vertebral hemangioma. The patient received RT, and symptomatic improvement was achieved.[17]

Researchers from the University of California San Francisco reported their experience with stereotactic body RT (SBRT) for the management of vertebral hemangiomas. The patients were treated with SBRT as the primary treatment or as boost therapy following external RT or as salvage management. Local control outcomes were acceptable, and treatment was well tolerated by the patients.[18] Dose-effect relationship was analyzed by Rades et al. and Beyzadeoglu et al. These studies suggested that total delivered doses in the range of 20–34 Gy achieved comparable rates of pain relief with the 36–40 Gy dose range.[19],[20]

The risk of secondary carcinogenesis was assessed in the literature.[21],[22] Heyd et al. suggested that total delivered doses should not exceed 30 Gy for the minimization of acute and late toxicity and the risk of radiation-induced secondary neoplasms.[23] Sparing of the spinal cord may be improved by the use of image guidance and contemporary treatment techniques such as SBRT.[24],[25] [Table 2] demonstrates selected studies evaluating treatment outcomes with RT for the management of vertebral hemangiomas. The results of our study are consistent with the literature [Table 2].
Table 2: Selected series showing response rates after radiation therapy in the management of vertebral hemangioma

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Analgesia, palliation, and local control of vertebral hemangiomas are the main goals of radiotherapeutic management. IGRT with conventional fractionation has been utilized for safe and effective management of benign and nonaggressive vertebral hemangiomas in our study. SBRT may serve as an effective mode of vertebral hemangioma management, particularly for lesions smaller than 30–40 mm.

In our study, patients with vertebral hemangioma treated with IGRT were analyzed and discussed. This series of 135 patients include a larger sample size compared with other studies. Furthermore, daily image guidance with kv-CBCT has been performed for patient management. Clearly, randomized prospective multicenter trials are needed to shed light on the optimal management of patients suffering from pain due to the vertebral hemangioma.


 > Conclusion Top


IGRT may be an effective and alternative treatment modality for the vertebral hemangioma management despite the need for further supporting evidence.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
Pinto DS, Hoisala VR, Gupta P, Sarkar P. Aggressive vertebral body hemangioma causing compressive myelopathy-Two case reports. J Orthop Case Rep 2017;7:7-10.  Back to cited text no. 1
    
2.
Ogawa R, Hikata T, Mikami S, Fujita N, Iwanami A, Watanabe K, et al. Total en bloc spondylectomy for locally aggressive vertebral hemangioma causing neurological deficits. Case Reports in Orthopedics 2015;1-7.  Back to cited text no. 2
    
3.
Kravtsov MN, Manukovskiĭ VA, Zharinov GM, Kandyba DV, Tsibirov AA, Savello AV, et al. Aggressive vertebral hemangiomas: Optimization of management tactics. Zh Vopr Neirokhir Im N N Burdenko 2012;76:23-31.  Back to cited text no. 3
    
4.
Murugan L, Samson RS, Chandy MJ. Management of symptomatic vertebral hemangiomas: Review of 13 patients. Neurol India 2002;50:300-5.  Back to cited text no. 4
[PUBMED]  [Full text]  
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Xiang H, Shen N, Chen B, Ma X, Xin G. A rare invasive hemangioma in cauda equina. Eur Spine J 2017;26:192-6.  Back to cited text no. 5
    
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Miszczyk L, Tukiendorf A. Radiotherapy of painful vertebral hemangiomas: The single center retrospective analysis of 137 cases. Int J Radiat Oncol Biol Phys 2012;82:e173-80.  Back to cited text no. 6
    
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Tarantino R, Donnarumma P, Nigro L, Delfini R. Surgery in extensive vertebral hemangioma: Case report, literature review and a new algorithm proposal. Neurosurg Rev 2015;38:585-92.  Back to cited text no. 7
    
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Cherian J, Sayama CM, Adesina AM, Lam SK, Luerssen TG, Jea A. Multilevel thoracic hemangioma with spinal cord compression in a pediatric patient: Case report and review of the literature. Childs Nerv Syst 2014;30:1571-6.  Back to cited text no. 8
    
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Jaimini A, D'Souza MM, Seniaray N, Sharma H, Arbind A, Sharma R, et al. Characterization of 'cold' vertebrae on 18F-FDG PET/CT. Nucl Med Commun 2016;37:30-42.  Back to cited text no. 9
    
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Jiang L, Liu XG, Yuan HS, Yang SM, Li J, Wei F, et al. Diagnosis and treatment of vertebral hemangiomas with neurologic deficit: A report of 29 cases and literature review. Spine J 2014;14:944-54.  Back to cited text no. 10
    
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Uzunaslan D, Saygin C, Gungor S, Hasiloglu Z, Ozdemir N, Celkan T. Novel use of propranolol for management of pain in children with vertebral hemangioma: Report of two cases. Childs Nerv Syst 2013;29:855-60.  Back to cited text no. 11
    
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Harkati R, Baré M, Gulyban A, Dechambre D, Berkovic P, Coucke PA. Multiple vertebral hemangiomas treated simultaneously with CyberKnife ® Technology. Rev Med Liege 2017;72:349-53.  Back to cited text no. 12
    
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Zhang M, Chen YR, Chang SD, Veeravagu A. CyberKnife stereotactic radiosurgery for the treatment of symptomatic vertebral hemangiomas: A single-institution experience. Neurosurg Focus 2017;42:E13.  Back to cited text no. 13
    
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Bellomia D, Viglianesi A, Messina M, Caltabiano GA, Chiaramonte R, Pero G, et al. Vertebral aggressive hemangioma. A case report and literature review. Neuroradiol J 2010;23:629-32.  Back to cited text no. 14
    
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Aich RK, Deb AR, Banerjee A, Karim R, Gupta P. Symptomatic vertebral hemangioma: Treatment with radiotherapy. J Cancer Res Ther 2010;6:199-203.  Back to cited text no. 15
    
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Heyd R, Seegenschmiedt MH, Rades D, Winkler C, Eich HT, Bruns F, et al. The significance of radiation therapy for symptomatic vertebral hemangiomas (SVH). Strahlenther Onkol 2010;186:430-5.  Back to cited text no. 16
    
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Aksu G, Fayda M, Saynak M, Karadeniz A. Spinal cord compression due to vertebral hemangioma. Orthopedics 2008;31:169.  Back to cited text no. 17
    
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Rawat S, Nangia S, Ezhilalan RB, Bansal AK, Ghosh D. Variance in the treatment of vertebral hemangiomas. J Indian Med Assoc 2007;105:42-8.  Back to cited text no. 18
    
19.
Rades D, Bajrovic A, Alberti W, Rudat V. Is there a dose-effect relationship for the treatment of symptomatic vertebral hemangioma? Int J Radiat Oncol Biol Phys 2003;55:178-81.  Back to cited text no. 19
    
20.
Beyzadeoglu M, Dirican B, Oysul K, Surenkok S, Pak Y. Evaluation of radiation carcinogenesis risk in vertebral hemangioma treated by radiotherapy. Neoplasma 2002;49:338-41.  Back to cited text no. 20
    
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Mazonakis M, Tzedakis A, Lyraraki E, Damilakis J. Radiation dose and cancer risk to out-of-field ad partially in-field organs from radiotherapy for symptomatic vertebral hemangiomas. Med Phys 2016;43:1841.  Back to cited text no. 21
    
22.
Sewell MD, Dbeis R, Bliss P, Watkinson T, Hutton M. Radiotherapy for acute, high-grade spinal cord compression caused by vertebral hemangioma. Spine J 2016;16:e195-6.  Back to cited text no. 22
    
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Heyd R, Strassmann G, Filipowicz I, Borowsky K, Martin T, Zamboglou N. Radiotherapy in vertebral hemangioma. Rontgenpraxis 2001;53:208-20.  Back to cited text no. 23
    
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Castelli J, Thariat J, Benezery K, Courdi A, Chanalet S, Paquis P, et al. Spinals and paraspinals tumors treated by CyberKnife: Feasibility and efficacy. Cancer Radiother 2010;14:5-10.  Back to cited text no. 24
    
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Saghal A, Chou D, Ames C, Ma L, Lamborn K, Huang K, et al. Image-guided robotic stereotactic body radiotherapy for benign spinal tumors: The University of California San Francisco preliminary experience. Technol Cancer Res Treat 2007;6:595-604.  Back to cited text no. 25
    



 
 
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