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. 2017 Jun 1;8(4):323–339. doi: 10.1177/2192568217701096

Solitary Osteochondroma of the Spine—A Case Series: Review of Solitary Osteochondroma With Myelopathic Symptoms

Ramakanth Yakkanti 1, Ikemefuna Onyekwelu 1, Leah Y Carreon 2,✉, John R Dimar II 1,2
PMCID: PMC6022963  PMID: 29977716

Abstract

Study Design:

Case series and literature review.

Objective:

There is a growing body of literature supporting that osteochondroma of the spine may not be as rare as previously documented. The purpose of this study was to perform an updated review and present our experience with 4 cases of solitary osteochondroma of the spine, including surgical treatment and subsequent outcomes.

Methods:

A review of 4 cases and an updated literature review.

Results:

All 4 cases were diagnosed as solitary osteochondroma of the spine based on clinical and histopathologic findings. Majority of the lesions arose from the posterior column with one case showing extension into the middle column with clinical neurologic sequelae. Treatment strategies for all cases included complete marginal excision of the lesions using a posterior approach. All 4 cases showed no radiographic evidence of recurrence. The literature review yielded 132 cases of solitary osteochondroma and 17 case associated with multiple hereditary exostosis. Out of the 132 cases, 36 presented with myelopathic symptoms.

Conclusion:

Osteochondroma of the spine may not be as rare as previously reported. The best approach to treatment in almost all symptomatic cases include wide surgical excision of the tumor. This should include complete resection of the cartilaginous cap of the tumor in an effort to prevent recurrence. When excision is performed properly, the outcomes are excellent with very low recurrence of the tumor.

Keywords: osteochondroma, literature review, case series, exostosis, surgical excision

Introduction

Osteochondroma (exostosis) is the most common benign bone tumor, accounting for 36% of benign bone tumors.1 Most often found in long bones, reports suggest osteochondroma of the spine to be relatively rare, accounting for only 4% to 7% of primary benign spinal tumors1-3 and less than 3% of all osteochondromas.3,4 Osteochondroma can arise as a solitary lesion or as part of an inherited condition known as multiple hereditary exostosis (MHE).1 Several studies have reported that solitary osteochondromas are more common in the spine when compared with osteochondroma associated with MHE.2,4,5 There is a growing body of evidence suggesting osteochondroma of the spine may not be as rare as previously reported.6-8 In this article, we describe our experience with the diagnosis, treatment, and natural history of osteochondroma of the spine of 4 cases, and the most up-to-date literature review of this topic since 2003.

Case Series

The authors have obtained the patients’ informed written consent for print and electronic publication of the case report.

Case 1

A 6-year-old female was brought to the emergency department for nonradiating neck pain that was localized to her left posterior neck. The patient had no neurological signs or symptoms. Radiographs demonstrated an osseous neck mass arising from the posterior cervical elements (Figure 1). Advanced imaging (magnetic resonance imaging and computed tomography scans) demonstrated an osseous lesion with a medullary cavity contiguous with the left C6 lamina. No signs of cord or root compression were seen. The patient has no known significant medical or family history of similar lesions.

Figure 1.

Figure 1.

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A 6-year-old female referred for a nonpainful mass in her neck noticed by her family. (A) Lateral radiograph showing ossified mass involving the C6 vertebrae and (B) sagittal magnetic resonance imaging showing the extent of the soft tissue involvement and the mass arising from the posterior cervical elements. (C) Hematoxylin and eosin slide of the cervical mass demonstrating a benign cartilage cap with subchondral bone, findings typical of an osteochondroma.

Case 2

A healthy 35-year-old male complained of 2 weeks of persistent mid-back pain after riding a go-cart. The patient denied any neurological symptoms during this time and was neurologically intact on physical examination. Imaging studies showed an osseous lesion about the low thoracic and thoracolumbar junction (Figure 2) without neurological involvement. The patient denies any medical or family history of similar lesions.

Figure 2.

Figure 2.

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A 35-year-old male with mid-back pain following a minor injury. Plain radiographs demonstrated a mass arising from the posterior elements of T12. (A) Mid-Sagittal computed tomography scan of the thoracic spine demonstrating an osseous mass at the thoracolumbar junction. (B) Intraoperative clinical photograph of well-encapsulated thoracic mass.

Case 3

An 11-year-old male had progressive right posterior neck swelling for the past 10 months. A cervical computed tomography scan showed a mass at the C6 vertebrae with medullary continuity with the right lamina and spinous process. There was no evidence of cord or nerve root compression or vascular compromise. There was no significant medical or family history of similar lesions.

Case 4

A 36-year-old female had neck pain and progressive myelopathy (bowel and bladder dysfunction, gait abnormality, and progressive upper and lower extremity weakness). No history of antecedent trauma was reported. Plain radiographs showed an osseous lesion arising from the posterior column with significant canal compromise at the level of the C3 and C4 vertebrae (Figure 3). The patient had no medical or family history of similar lesions.

Figure 3.

Figure 3.

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(A) Lateral radiograph and (B) axial computed tomography scan showing osseous mass arising from the right C3 lamina and invading the spinal canal causing cord compression. Biopsy revealed an osteochondroma. (C) Postoperative lateral radiograph showing C3 vertebrectomy, anterior reconstruction with a titanium cage and plate, and a posterior instrumented fusion from C2 to C5 required to stabilize the spine following a wide resection of the osteochondroma.

Literature Review

Ovid MEDLINE and other nonindexed citations database search engines were used with the assistance of a medical librarian. The terms “osteochondroma” and “spine” and/or proxy descriptors were used to query PubMed. No limit in publication year, country, or language of publication was used. This yielded a list of all reported cases of osteochondroma of the spine since 1951. The list of articles was screened using the inclusion criterion—all reported cases from 2016 to 2004—and the following exclusion criteria: literature reviews, cases of primary tumor not arising from spine, non–case report accounts of cases, and nontumor processes (infection). Each case was reviewed for each parameter of clinical history and radiographic description whenever available in the case reports. Demographics, anatomic location of tumor, symptoms, treatment, and recurrence rates of tumor were almost always available and reported. A custom-built Excel database was used to organize and analyze the data. Descriptive statistics were used to summarize the results of the data.

Results

All 4 cases were diagnosed as solitary osteochondroma of the spine based on clinical findings and histopathologic features. All cases except for “Case 4” had no neurological symptoms—Case 4 was associated with cord compression and progressive myelopathy. Three of the 4 cases involved the cervical spine (includes case with cord compression) and 1 of 4 from the thoracolumbar region. All cases of osteochondroma in this series appeared to arise from the posterior column, with one case showing extension into the middle column and clinical neurologic sequelae. Treatment strategies for all cases included complete marginal excision of osteochondroma lesions using a posterior approach. Additionally, Case 4 (osteochondroma with cervical retrovertebral lesion and cord compression) required anterior corpectomy with placement of an interbody cage, followed by posterior decompression and instrumented fusion. All patients had complete symptomatic relief at their latest follow-up (up to 2 years) and showed no radiographic evidence of recurrence.

The review literature yielded a total of 223 articles,2-92 of which 110 were from the 2016 to 2004 period. Twenty-six articles were excluded, leaving 84 articles in the final analysis. The 84 articles yielded 149 reported cases. One hundred and thirty-two (88.6%) were solitary osteochondromas and 17 (11.4%) were associated with MHE.

Table 1 lists all 132 cases of solitary osteochondroma of the spine from the literature. Table 1 highlights the interesting data from each case of solitary osteochondroma. The location, treatment, and outcome of the cases are shown, along with the demographic data. For solitary osteochondromas (Table 2), there was a female-to-male ratio of 1:1.6 and an average age of 35.2 years (range = 2-77). The most common spinal level involved was cervical, with 63 (52.2%) of the cases, followed by lumbar 35 (26.5%), thoracic 24 (18.2%), sacrum 9 (6.8%), and coccyx 1 (0.76%). The most frequent spinal anatomic column involved was the posterior column, with 85 cases (64.3%), followed by unknown 28 (21.2%), anterior column 19 (14.3%), and 0 in the middle column.

Table 1.

Reviewed Cases in Literature: Interesting Dataa.

Author Journal Year Age Sex Tumor Level Location Presentation Radiculopathy Myelopathy Treatment Clinical Outcomes Recurrence
Ramzan et al Pediatric Neurosurgery 2016 8 Male C1 Posterior C1 arch Symptomatic No Yes Complete excision Complete resolution N/A
Bauer et al Skeletal Radiology 2015 19 Female C1 C1 posterior arch Symptomatic No Yes Complete excision Complete resolution No
Michael Journal of Pediatric Orthopaedics 2015 16 Female L5-S1 Facet joints Symptomatic No No Complete excision Complete resolution No
Haque et al European Spine Journal 2015 21 Male S3-S4 Sacrum L of midline Asymptomatic No No Complete Excision Complete resolution No
Sciubba et al Journal of Neurosurgery: Spine 2015 35 Female C7 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 48 Male S1 N/A N/A N/A N/A En bloc resection N/A Yes
Sciubba et al Journal of Neurosurgery: Spine 2015 46 Male C7 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 46 Female T9-T10 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 61 Male L2 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 65 Male C3-T2 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 48 Male S1 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 43 Female C6-C7 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 76 Female T11-T12 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 21 Male S1 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 49 Female C5-C7 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 17 Male S1 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 32 Male L4-L5 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 60 Male T12 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 68 Female L2 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 13 Female T1 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 19 Male L5 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 38 Male L4-L5 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 20 Male L5 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 36 Female T11-T12 N/A N/A N/A N/A En bloc resection N/A Yes
Sciubba et al Journal of Neurosurgery: Spine 2015 26 Male C4-C5 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 33 Female L1 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 21 Male T7 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 18 Male T1-T2 N/A N/A N/A N/A Unknown N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 13 Male L5-S3 N/A N/A N/A N/A En bloc resection N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 17 Female C1-C3 N/A N/A N/A N/A Intralesional excision N/A No
Sciubba et al Journal of Neurosurgery: Spine 2015 2 Female T8-T11 N/A N/A N/A N/A En bloc resection N/A No
Kang et al JAMA Otolaryngology—Head and Neck Surgery 2015 60 Female C2 N/A Symptomatic No No Complete excision N/A N/A
Eren et al The Spine Journal 2015 24 Female L4 Spinous process Symptomatic No No N/A N/A N/A
Neal et al Military Medicine 2015 40 Male L5 Right anteriosuperior endplate of L5 Symptomatic No No Complete excision Complete resolution No
Dormont et al Clinical Neuroradiology 2014 59 Male C4 Posterior C4 arch Symptomatic No No Complete excision Near complete resolution No
Mahore et al BMJ Case Reports 2014 28 Male D2-D3 Posterior arch Symptomatic No Yes Laminectomy Complete resolution No
Boucetta et al Pan African Medical Journal 2014 48 Male C6 Posterior arch Symptomatic No No Complete excision Complete resolution N/A
Kantarsi et al The Spine Journal 2014 24 Female C3 Lamina Symptomatic Yes No N/A N/A N/A
Fumiaki et al Neurologia Medico Chirucgica 2014 57 Male L4 Inferior articular process Symptomatic Yes No Laminectomy Complete resolution No
Fumiaki et al Neurologia Medico Chirucgica 2014 63 Female S1 Superior articular process of Symptomatic Yes No Hemilaminectomy Complete resolution No
Fumiaki et al Neurologia Medico Chirucgica 2014 48 Female L4 Inferior articular process Symptomatic Yes No Hemilaminectomy Near complete resolution No
Fumiaki et al Neurologia Medico Chirucgica 2014 32 Male L4 Inferior articular process Symptomatic Yes No Hemilaminectomy Complete resolution No
Fumiaki et al Neurologia Medico Chirucgica 2014 62 Male L4 Inferior articular process Symptomatic Yes No Hemilaminectomy Complete resolution No
Barbagallo et al European Review for Medical and Pharmacological Sciences 2014 68 Male C4-C5 Anterior arch Symptomatic No No Complete excision Complete resolution No
Parekh et al BMJ Case Reports 2014 20 Male C7-T1 Posterior arch Asymptomatic No No Complete excision N/A N/A
Hopper et al Journal of Belgian Society of Radiology 2014 68 Female T9-L3 Posterior arch Symptomatic No No Complete excision Complete resolution No
Mont et al Orthopedics (Healio) 2014 11 Male L2-L4 Inferior articular process Symptomatic Yes No En bloc resection Complete resolution No
Jameel et al Journal of Clinical and Diagnostic Research 2014 14 Female C5-C6 Transverse process Asymptomatic No No Total excision Complete resolution No
David et al Asian Spine Journal 2014 52 Male C2-C6 Transverse process Symptomatic Yes No Laminectomy Complete resolution No
Chow et al Pediatric Neurology 2013 9 Male C1-C2 Inner surface of C2 arch Symptomatic Yes No C2 hemilaminectomy, resection of posterior C1 arch Complete resolution N/A
Scuotto et al BMJ Case Reports 2013 56 Female L2 Lamina Symptomatic Yes No En bloc resection Complete resolution No
Garg et al Kulak Burun Bogaz Ihtis Derg 2013 22 Male C3-C4 Vertebrae and pedicles N/A N/A N/A N/A N/A N/A
Jianru et al Journal of Spinal Disorders Tech 2013 43 Male L4 Spinous process Symptomatic Yes No Complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 26 Male C1-C2 Lateral mass Symptomatic No Yes Complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 11 Male T1 Laminar mass Symptomatic Yes No Laminectomy, complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 60 Female C1 Lateral mass Symptomatic No Yes Complete excision Worsening of symptoms N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 34 Female C1-C2 Lateral mass Symptomatic No Yes Laminectomy, complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 17 Female C1 Transverse process Symptomatic Yes No Complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 63 Female C5-C7 Lamina Symptomatic Yes No Complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 17 Female T6 Pedicle Symptomatic Yes No Laminectomy, complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 49 Female C2-C3 Vertebral body Symptomatic No Yes Laminectomy, complete excision Worsening of symptoms N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 68 Female L2 Lamina Symptomatic No Yes Laminectomy, complete excision Complete resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 56 Female T5 Vertebral body Symptomatic Yes No Laminectomy, complete excision Partial functional resolution N/A
Jianru et al Journal of Spinal Disorders and Techniques 2013 57 Female C5 N/A Symptomatic Yes No Laminectomy, complete excision Complete resolution N/A
Madan et al South Asian Journal of Cancer 2013 9 Male T1 Vertebral body, posterior arch Symptomatic No Yes Partial resection Near complete resolution No
Rai et al Global Spine Journal 2013 65 Male C2 Vertebral body Symptomatic No No Complete excision Complete resolution N/A
Ghasemikhah et al Iranian Journal of Radiology 2013 19 Male T9 Posterior arch Symptomatic No Yes Laminectomy Complete resolution N/A
Temiz et al Acta Orthopaedica et Traumatologica Turcica 2012 62 Female L2 Inferior articular process Symptomatic No Yes Complete excision Complete resolution No
Chang et al Skeletal Radiology 2012 39 Male L4 N/A Symptomatic No No N/A N/A N/A
Hussain et al BMJ Case Reports 2012 16 Male C1 Posterior arch Symptomatic No Yes Laminectomy Near complete resolution No
Temiz et al Turkish Neurosurgery 2012 48 Male L3 Inferior articular process Symptomatic Yes No Hemilaminectomy, complete excision Near complete resolution No
Kettner et al Spine 2012 21 Female C5 Spinous process Symptomatic No No Laminectomy w/t en bloc resection Complete resolution N/A
Mamindla et al Asian Journal of Neurosurgery 2012 14 Male C3 Lamina Symptomatic No Yes Laminectomy w/t en bloc resection Complete resolution N/A
Shin et al Journal of Korean Neurosurgery 2012 32 Male C4-C5 Lamina and facet joint Symptomatic No Yes Hemilaminectomy Near complete resolution No
Kars et al Asian Spine Journal 2012 42 Female C1 Lamina Symptomatic No Yes Laminectomy Complete resolution No
Nakamura et al Skeletal Radiology 2011 69 Male C7-T1 N/A Symptomatic No Yes Laminectomy Complete resolution No
Rousseaux et al Orthopaedics & Traumatology: Surgery & Research 2011 23 Male C4 Posterior arch Symptomatic No Yes Laminectomy Complete resolution No
Kettner et al Journal of Manipulative and Physiological Therapeutics 2011 24 Male C4 Vertebral body Symptomatic No No Nonsurgical—Spinal manipulation Complete resolution No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 26 Male L1 Spinous process Symptomatic No No Complete excision Complete resolution No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 9 Male C3/C4-T1 Spinous process, posterior arch Asymptomatic No No Complete excision Asymptomatic No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 36 Female T11-L1 Lamina Symptomatic Yes No Laminectomy, complete excision Near complete resolution No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 65 Male C4 Vertebral body Symptomatic Yes No Anterior excision, followed by anterior cervical fusion Complete resolution No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 19 Male C5-C6 Spinous process Symptomatic No No Complete excision Complete resolution No
Saglik et al Archives of Orthopaedic and Trauma Surgery 2011 32 Female L3-L4 Lamina Asymptomatic No No Nonsurgical Asymptomatic No
Schneider et al Ethiopian Medical Journal 2010 7 Male Coccyx Coccyx Symptomatic No No En bloc excision Complete resolution N/A
Shimada et al Neurologia Medico Chirucgica 2010 58 Male C1-C2 Spinous process Symptomatic Yes Yes En bloc excision Complete resolution No
Meshkini et al Journal of Neurosurgery: Spine 2010 29 Male L4 Pedicle Symptomatic Yes No Laminectomy Complete resolution N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 58 Male L5 Vertebral body Symptomatic Yes Yes Laminectomy Complete resolution N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 60 Male C5 Lamina Symptomatic No Yes Hemilaminectomy Complete resolution N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 34 Male C5-C6 Lamina Symptomatic No Yes Laminectomy Near complete recovery N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 55 Male T9 Vertebral body Symptomatic No Yes Complete excision Complete resolution No
Meshkini et al Journal of Neurosurgery: Spine 2010 17 Male L3 Inferior facet Symptomatic Yes No Hemilaminectomy Complete resolution N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 34 Female C7 Pedicle Symptomatic No Yes Hemilaminectomy Near complete resolution N/A
Meshkini et al Journal of Neurosurgery: Spine 2010 31 Male T8 Superior facet Symptomatic No Yes Laminectomy N/A N/A
Kim et al The Spine Journal 2010 54 Female S1 Sacral ala—anterior surface Symptomatic No No Complete excision Complete resolution No
Cha et al Journal of Korean Neurosurgery 2010 57 Female L3 Lamina Symptomatic Yes No En bloc resection, laminectomy, facetectomy Complete resolution N/A
Horiuchi et al Journal of Neurosurgery: Spine 2009 77 Female C1 Posterior arch Symptomatic No Yes Hemilaminectomy w/t en bloc resection Complete resolution No
Horiuchi et al Journal of Neurosurgery: Spine 2009 72 Male L4 Inferior facet Symptomatic No No Marginal resection and facetectomy Complete resolution No
Horiuchi et al Journal of Neurosurgery: Spine 2009 69 Male L4-L5 Inferior facet Symptomatic No No Intraarticular injection, biopsy, and ablation of articular facet joint Complete resolution No
Tian et al Orthopedics 2009 38 Male L5 Lamina Symptomatic No Yes Laminectomy Complete resolution No
Jakheria et al Journal of Pediatric Orthopaedics B 2009 8 Female C2-C6 Spinous process Symptomatic No No En bloc resection Complete resolution No
Chou et al Case Reports/Journal of Clinical Neuroscience 2009 16 Female C1-C2 Vertebral Body Symptomatic No No Complete Excision Complete resolution No
Wenyuan et al SAS Journal 2009 28 Male T8 Transverse process Symptomatic No No Radial excision Complete resolution N/A
Hassankhani et al Cases Journal 2009 16 Female L3 Spinous process Asymptomatic No No En bloc resection N/A No
Srikantha et al Journal of Neurosurgery: Spine 2008 17 Male C3 Spinolaminar Junction Symptomatic No Yes En bloc resection Complete resolution No
Srikantha et al Journal of Neurosurgery: Spine 2008 23 Male C4-C5 Transverse processes, lamina, pedicles Symptomatic No Yes Partial resection, C4-C5 corpectomy, C3-C5 fusion Complete resolution No
Srikantha et al Journal of Neurosurgery: Spine 2008 40 Female C6 Superior articular facet Symptomatic Yes No Medial facetectomy Complete resolution No
Byung-June et al Joint Bone Spine 2007 23 Male L5-S1 Facet Symptomatic Yes No Partial laminectomy Complete resolution No
Song et al European Journal of Pediatric Surgery 2006 11 Male T4 Superior articular process Symptomatic No Yes Laminectomy (T2-T3) Complete resolution No
Zhao et al Spine 2007 23 Female C7 Transverse process Symptomatic No No En bloc resection Complete resolution No
Chatzidakis et al Acta Neurochirurgica 2007 22 Male C2 Dens of C2 Symptomatic No No N/A N/A No
Ozturk et al Acta Orthopaedica Belgica 2007 46 Male C1 Lamina Symptomatic Yes No Hemilaminectomy Complete resolution No
Maheshwari et al Orthopaedic Surgery 2006 20 Male C7 Pedicle Symptomatic No Yes Laminectomy Complete resolution No
Moon et al Pediatric Neurosurgery 2005 16 Male C5-C7 Spinous process Symptomatic Yes Yes Hemilaminectomy, complete excision of tumor Complete resolution No
Samartzis et al Spine 2006 11 Male S2 Lamina Symptomatic Yes No Laminectomy S1-S4 Complete resolution No
McCall et al Journal of Neurosurgery 2006 13 Female C3 Lamina Asymptomatic No No Complete excision N/A N/A
Yoshida et al Acta Oto-Laryngologica 2006 61 Female C1 Anterior arch Symptomatic No No Complete excision Complete resolution No
Grivas et al European Spine Journal 2005 46 Female C7 Pedicle Symptomatic Yes No Complete excision Complete resolution No
Brastianos et al Neurosurgery 2005 26 Female T12 Vertebral body Symptomatic No Yes Complete excision, T12 corpectomy Complete resolution No
Agrawal et al Pediatric Neurosurgery 2005 14 Male L5-S1 Illiac crest Symptomatic Yes No Laminectomy Complete resolution No
Faik et al Joint Bone Spine 2005 19 Male T4-T5 Costovertebral angle, T4-T5 foramina Symptomatic Yes No Laminectomy, complete excision Complete resolution No
Miyamoto et al Spinal Cord 2005 23 Male C2 Pedicle Symptomatic No Yes L hemilaminectomy, partial excision Partial functional recovery No
Kouwenhoven et al European Spine Journal 2004 42 Male C1-C2 Neural arches Symptomatic Yes No Laminectomy, en bloc resection Complete resolution No
Gu rkanlar et al Journal of Clinical Neuroscience 2004 35 Male L4 Lamina Symptomatic Yes No Complete Excision Complete resolution No
Schrot et al Journal of Neurosurgery 2004 15 Male C8 Dermatome Symptomatic Yes No Hemilaminectomy, pediculectomy w/t complete excision of tumor Complete resolution No
Kulkarni et al Neurologia Medico Chirucgica 2004 15 Male T10-T11 Facet Symptomatic No Yes Laminectomy Complete resolution No
Gille et al Spine 2004 18 Female C4 Transverse process Symptomatic Yes No Cervicotomy Complete resolution No
Gille et al Spine 2004 15 Male C5 Vertebral body Symptomatic No Yes Laminectomy and cervicotomy Complete resolution No
Gille et al Spine 2004 73 Male C2 Posterior arch Symptomatic No Yes Laminectomy Complete resolution No
Gille et al Spine 2004 18 Male T11 Pedicle Asymptomatic No No Laminectomy Complete resolution No
Gille et al Spine 2004 28 Female L4 Posterior arch Symptomatic Yes No Laminectomy Complete resolution No
Gille et al Spine 2004 45 Female S1 Vertebral body Symptomatic Yes No Lumbotomy Complete resolution No
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aAll 132 cases reviewed from literature are presented. Age, sex of the patient, location of lesion, type of surgery, symptoms, and recurrence are shown. If symptomatic w/o myelopathic or radiculopathic symptoms, symptomatic due to pain. N/A, data unavailable in the literature.

Table 2.

Demographic Data of Solitary Osteochondroma of the Spine, 132 Cases, Without a Known Hereditary Genetic Disorder.

Sex, males 61.3%
Age, years (mean, range) 35.2 (2-77)
Spinal level of tumor
 Cervical 65 (52%)
 Thoracic 24 (18%)
 Lumbar 35 (27%)
 Sacrum 9 (7%)
 Coccyx 1 (1%)
Involved spinal column
 Posterior 85 (64%)
 Anterior 19 (14%)
 Middle 0 (0%)
 Unknown 28 (21%)
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There were 36 (27.2%) cases that involved solitary osteochondroma with myelopathic symptoms (Table 3). This group had a female-to-male ratio of 1:2.6 and average age of 35.1 years (range = 8-77). The most common spinal level involved was cervical in 24 (66.6%) cases, followed by thoracic 8 (22.2%) and lumbar 4 (11.1%). The most frequent spinal anatomic column involved was posterior column, with 29 cases (80.5%), followed by anterior column 6 (16.6%), unknown 1 (2.7%), and middle column 0 (0%). The osteochondroma began in the posterior arch in 20 (55.5%) of the cases, followed by the lamina in 7 (19.4%), vertebral body in 5 (16.6%), spinous process in 2 (5.5%), and unknown location in 1 (2.7%) of the cases.

Table 3.

Demographic Data on Solitary Osteochondroma With Spinal Cord Compressiona.

Sex, males 72.2%
Age, years (mean, range) 35.1 (8-77)
Spinal level of tumor
 Cervical 24 (66.6%)
 Thoracic 12 (22.2%)
 Lumbar 5 (11.1%)
 Sacrum 0 (0%)
Involved spinal column
 Posterior 29 (81.5%)
 Anterior 6 (16.6%)
 Middle 0 (0%)
 Unknown 1 (2.7%)
Origin of tumor
 Pedicle 3 (8.3%)
 Laminae 7 (19.4%)
 Spinous process 2 (5.5%)
 Posterior arch other than pedicle, laminae, spinous  process 17 (47.2%)
 Vertebral body 6 (16.6%)
 Unknown location 1 (2.7%)
Treatment
 Anterior approach 3 (8.3%)
 Posterior approach 29 (80.5%)
 Combined anterior-posterior approach 2 (5.5%)
 Unknown approach 2 (5.5%)
 Patients requiring excision 36 (100%)
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aA case series of 27 patients with unknown locations of osteochondroma and unknown symptoms, which was part of the data, had to be excluded from the results due to lack of data.

All 36 patients underwent surgery, of whom 29 (80.5%) underwent a posterior approach, 3 (8.3%) underwent an anterior approach, 2 (5.5%) underwent a combined anterior-posterior approach, and 2 (5.5%) approaches were unknown. The clinical outcomes showed improvement of symptoms in 34 (94.4%) of the patients, with 28 people showing a complete recovery and 6 with a partial recovery. Two cases showed worsening symptoms after surgery. There were 2 recurrences among all cases recorded, and none among solitary lesions with myelopathic symptoms.

Discussion

The first solitary osteochondroma was reported in 1843 by Reid.64 Many reports in the literature show that solitary osteochondroma is more common than lesions associated with MHE. The prevalence of osteochondroma in the spine is likely higher than previously thought. There seems to be a rise in the amount of case reports of osteochondroma published in the recent years (2004 to 2016). When Albrecht et al2 reviewed the relevant English literature from 1843 to 1992, it yielded 96 cases of solitary spinal osteochondroma. When Gille et al8 updated the review, they identified 54 additional cases of solitary spinal osteochondroma from 1992 to 2003. Our study yielded 132 new cases reported from 2004 to 2016, representing a 2.4-fold increase since 2003.

This increase in the number of cases in a smaller period of time is likely due to a higher rate of case reports being published on the topic rather than an actual increase in the incidence of these tumors. Nevertheless, the higher number of reported cases in the past decade likely underestimates the true prevalence of osteochondroma, because a significant portion of these tumors/lesions remain asymptomatic and, thus, may not be seen by a health care provider and/or require surgical treatment.

The review and analysis of the reported cases corroborate some of the trends seen in the literature, such as cervical spine being the most common site for a solitary osteochondroma of the spine, complete surgical excision being the most common method of surgical treatment, and the good outcomes and low recurrence rates after excision.

Additionally, the review of literature indicated that 27.2% of the cases with solitary osteochondromas of the spine had myelopathic features. This is in concordance with previous reports of 30% by Albrecht et al.2 It is proposed that the myelopathic symptoms seen in osteochondroma are due to progressive compression of the spinal structures, but may include a potentiated effect as the tumor grows over several years; likewise, the onset of age-related degenerative changes seen with spinal stenosis may also contribute.8

Osteochondroma is a form of exostosis that can be seen in any age group. It is generally reported that the age range for symptomatic presentation for solitary osteochondroma is between 10 and 30 years for peripheral lesions, but it appears that spine patients develop symptoms at an average age of 32, distinctly different from the peripheral lesions seen in children. By definition, osteochondroma has a characteristic cartilage cap on histology and a medullary continuity with the host bone, and can be sessile or pedunculated. MHE involves many exostoses in a single patient, unlike in the case of solitary osteochondroma, which is more common. An incidence of 1.3% to 4.1% has been reported as the percentage of solitary osteochondromas that affect the spine; however, 9% of MHE lesions are found in the spine.2 In the current review, 11.4% of all the cases of osteochondromas of the spine reviewed were associated with MHE.

Malignant transformation is low in solitary osteochondroma (<3%), but can be as high as 10% when associated with inherited genetic mutations as seen with MHE. MHE has an autosomal dominant inheritance pattern and involves mutations in the EXT 1, 2, and 3 genes on chromosome 8, 11, and 19, respectively. Malignant degeneration leads to a low-grade peripheral chondrosarcoma, which is managed with complete surgical resection. Malignant transformation of solitary osteochondroma is most frequently reported in the pelvis and rarely occurs in the spine.

A treatment algorithm for these lesions should begin with a thorough history and physical examination, to evaluate for genetic inheritance of similar lesions and to rule out neurovascular compromise that will necessitate surgery. Moreover, the majority of these lesions remain benign and are painless. In benign cases, observation with radiographic surveillance (computed tomography and magnetic resonance imaging and other advanced imaging may be used as indicated to better characterize the lesion and its local effects). Osteochondromas do have a tendency to increase in size and, depending on its location, may be associated with neurologic sequelae. In cases where unrelenting pain and/or evidence of neurovascular compromise (radiculopathy, myelopathy, or vascular compression) exists, surgical management may be warranted. Surgical treatment may include in situ marginal or wide excision, via a posterior, anterior, or combined approach, with or without instrumentation. In some cases, that is, Case 4, a need for cord or nerve root decompression along with instrumented stabilization with or without fusion may be required. Tumor excision may sometimes require both an anterior approach and a posterior approach. Of paramount importance during surgical excision is complete resection of the characteristic cartilage cap seen with these tumors. Incomplete resection of the cartilage cap may increase the risk of recurrence, and the pediatric population is more susceptible to tumor recurrence given their higher growth potential/age at presentation.

The recurrence rate in the review of the literature was 1.3% for all cases, and 0% for solitary spinal osteochondromas with myelopathic symptoms. Nevertheless, the current review of literature demonstrates a lower recurrence rate than previously reported (4%).8 However, there may be a number of unreported recurrences, given that not all cases in the literature explicitly reported this parameter. There is also the impact of a better understanding of the biology of the tumor, advanced imaging, and surgical techniques allowing for more expedient treatment in the recent years.

Conclusion

Osteochondroma is a relatively common bone tumor, accounting for 36% of all benign bone tumors,1 but occurs infrequently in the spine accounting for less than 3% of all osteochondromas.3,4 The solitary lesions in the spine may cause neurologic symptoms including radiculopathy and myelopathy, 29.5% and 27%, respectively, as reported in this review. The best approach to treatment in almost all symptomatic cases is marginal excision of the tumor. Meticulous surgical excision, with complete resection of the cartilaginous cap of the tumor, is important in preventing recurrence. When tumor excision is performed adequately, the outcomes are excellent with very low recurrence rates.

Footnotes

Declaration of Conflicting Interests: The author(s) declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: RY and IO have no relationships to disclose. JRD is an employee of Norton Healthcare; board member, Scoliosis Research Society; receives consulting fees from Medtronic and DePuy; receives payments for lectures from Medtronic, DePuy, and Norton Healthcare; holds patents and receives royalties from Medtronic; is on the editorial review board of JBJS Highlights, Spine, Spine Deformity, JAAOS, and Global Spine. Nuvasive provided funds directly to database company. No funds are paid directly to individual or individual’s institution 06/2012 to 04/2015. LYC is an employee of Norton Healthcare; member, Editorial Advisory Board, Spine and Spine Journal; institutional review board member, University of Louisville Institutional Review Board; research committee member, Scoliosis Research Society; receives research funds from the Orthopedic Research and Educational Fund, 2013 to present; receives Scoliosis Research Society Research Funding, 2013 to present; received funds for travel for Study Planning Meetings from the Center for Spine Surgery and Research of the University of Southern Denmark; received funds for travel for annual required Continuing Education for Institutional Review Board Members, University of Louisville Institutional Review Board; Nuvasive provided funds directly to database company. No funds are paid directly to individual or individual’s institution 06/2012 to 4/2015.

Funding: The author(s) received no financial support for the research, authorship, and/or publication of this article.

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