Thoracic spinal osteochondroma: A rare presentation of spinal cord compression

Thoracic spinal osteochondroma: A rare presentation of spinal cord compression

Accepted Manuscript Thoracic spinal osteochondroma: A rare presentation of spinal cord compression David R. Hansberry, Raghav Gupta, Arpan V. Prabhu,...

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Accepted Manuscript Thoracic spinal osteochondroma: A rare presentation of spinal cord compression

David R. Hansberry, Raghav Gupta, Arpan V. Prabhu, Nitin Agarwal, Mougnyan Cox, Upasana Joneja, Mark T. Curtis, James S. Harrop, Adam E. Flanders PII: DOI: Reference:

S0899-7071(17)30093-1 doi: 10.1016/j.clinimag.2017.05.014 JCT 8252

To appear in: Received date: Revised date: Accepted date:

26 February 2017 12 May 2017 15 May 2017

Please cite this article as: David R. Hansberry, Raghav Gupta, Arpan V. Prabhu, Nitin Agarwal, Mougnyan Cox, Upasana Joneja, Mark T. Curtis, James S. Harrop, Adam E. Flanders , Thoracic spinal osteochondroma: A rare presentation of spinal cord compression, (2017), doi: 10.1016/j.clinimag.2017.05.014

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ACCEPTED MANUSCRIPT Thoracic Spinal Osteochondroma: A Rare Presentation of Spinal Cord Compression David R. Hansberry, M.D., Ph.D., 1 Raghav Gupta, B.S.,2 Arpan V. Prabhu, B.S.3, Nitin Agarwal, M.D. 4, Mougnyan Cox, M.D.1, Upasana Joneja, M.D.5, Mark T. Curtis, M.D., Ph.D. 5, James S. Harrop, M.D.6, Adam E. Flanders, M.D.1

Department of Radiology, Thomas Jefferson University Hospital, Philadelphia, Pennsylvania,

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U.S.A.

New Jersey Medical School, Rutgers University, Newark, New Jersey, U.S.A.

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Department of Radiation Oncology, University of Pittsburgh Cancer Institute, Pittsburgh,

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Pennsylvania, U.S.A.

Department of Neurological Surgery, University of Pittsburgh Medical Center, Pittsburgh,

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Pennsylvania, U.S.A.

Department of Pathology, Anatomy, & Cell Biology, Thomas Jefferson University Hospital,

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Philadelphia, U.S.A.

Department of Neurological Surgery, Thomas Jefferson University Hospital, Philadelphia,

Osteochondroma; spine; hereditary multiple exostoses; myelopathy

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KEY WORDS:

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Pennsylvania, U.S.A.

resonance imaging (MRI); computed tomography (CT)

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ABBREVIATIONS: Magnetic

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Corresponding Author

David R. Hansberry, M.D., Ph.D. Department of Radiology Thomas Jefferson University Hospitals 132 South 10th Street Philadelphia, Pennsylvania 19107 Phone: (856) 577-2088 Email: [email protected]

ACCEPTED MANUSCRIPT ABSTRACT

Osteochondromas are the most common benign bone tumor typically seen in the appendicular skeleton and are rarely found in the spine. We present a case of an osteochondroma of the spine

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presenting with spinal cord compression.

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27-year-old male presented with lower extremity weakness and paresthesia, decreased lower extremity sensation, and decreased proprioception. MRI showed a heterogeneous mass with

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minimal peripheral enhancement and without restricted diffusion. CT demonstrated a calcified

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mass extending from the left facet joint of T11-T12 with medial extension, resulting in severe central canal stenosis and cord compression. The patient underwent surgical resection with

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pathology demonstrating an osteochondroma.

ACCEPTED MANUSCRIPT INTRODUCTION

Osteochondromas comprise more than one-third of benign bone tumors. The condition is a result of progressive ossification of abnormal cartilaginous tissue during development, resulting in a bony outgrowth which is covered by cartilage.[1] The etiology of osteochondromas includes

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congenital, trauma, and post radiation therapy.[2] They have been attributed to significant radiation

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exposure in 12-15% of osteochondroma cases.[3] These lesions primarily develop in adolescents before they reach skeletal maturity, though the condition is usually diagnosed in adulthood as an

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incidental finding on imaging.[4]

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Males are two and a half times more likely to develop osteochondromas than females.[5] Although

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most of these lesions occur within the appendicular skeleton, 3% of cases can occur within the

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vertebral column, rarely resulting in neurological deficits. Half of spinal osteochondromas are found within the cervical spine although the thoracic spine is also commonly affected.[4-6] The

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lesions assume one of two forms: solitary or hereditary. Hereditary spinal osteochondromas are an autosomal dominant condition called ‘multiple exostoses’ that result in the formation of multiple

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lesions and are up to three times more likely to affect the spinal cord, resulting in compression.

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Solitary spinal osteochondromas are associated with singular, sporadic lesion-formations. Approximately 3% of osteochondromas can undergo malignant transformations.[5] Affected patients typically present with sciatica, back pain, radiculopathy, or associated myelopathies, although a majority of patients are likely asymptomatic.[4] Here, we present a case in which a patient with a thoracic osteochondroma underwent surgical resection.

ACCEPTED MANUSCRIPT CASE REPORT

A 27-year-old male presented with progressive left greater than right, lower extremity numbness, mild lower extremity weakness, and decreased proprioception. Physical exam found decreased

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lower extremity sensation of the feet and posterior legs extending from the heels to buttock as well

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as minimal decreased strength with left foot dorsiflexion and toe extension. Magnetic resonance

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imaging (MRI) of the lumbar spine with and without contrast found a mass measuring 3.1 x 1.5 x 2.7 cm at the T11-T12 level with extension into the T12 neural foramina (Figure 1). There was a

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heterogeneous signal with areas of increased T1 and T2 signal. Post contrast enhanced images

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showed minimal peripheral enhancement. There was no abnormal diffusion signal. Subsequent computed tomography (CT) imaging of the thoracic spine without contrast demonstrated an

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ossified mass from the left facet joint at T11-T12 projecting medially into the spinal canal with

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moderate to severe central canal stenosis (Figure 2).

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The patient underwent a partial vertebrectomy through a left sided costotransversectomy/

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extracavitary approach to remove the T11-T12 mass. Laminectomies from T10 to T12 with foraminotomies were performed. Additionally, there was fusion from T9 to L1 with pedicular

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fixation and local bone graft. Surgical pathology demonstrated a 3.3 cm lesion with attached bone. Histologically, the lesion comprised of fibrous perichondrium, linear columns of maturing cartilage and trabecular bone with bone marrow elements at the base (Figure 3). The histologic findings were consistent with an osteochondroma. Postoperatively he was discharged home with 5/5 strength in all muscle groups. At three-year follow-up, he had only minimal weakness in his

ACCEPTED MANUSCRIPT left anterior tibialis and extensor hallucis longus. An MRI at this time indicated no local recurrence

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or residual.

ACCEPTED MANUSCRIPT DISCUSSION

Solitary spinal osteochondromas are a rare occurrence and account for less than 1% of all tumors within the spine. Although benign, a small subset of these exostoses can become malignant, though this is far more frequent (10-25%) in patients harboring the congenital form of the condition.[5]

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Amongst solitary lesions, only 2.7% are believed to undergo a malignant conversion.[7] These

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tumors exclusively occur in bones that undergo endochondral ossification and are most commonly found on the posterior spine, including the transverse and spinous processes; these tumors are also

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more frequently diagnosed in males.[4, 8, 9] They often occur in the second or third decades of life.[10] The condition may be symptomatic, depending on whether the in-growth encroaches on

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the spinal canal.

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They most commonly affect the cervical and thoracic regions of the spinal cord, though rare cases have reported finding the lesions in the sacral and lumbar regions.[4, 6, 7, 11-39] Two different

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hypotheses have been proposed to help explain the anatomical distribution of these lesions within the spine. Albrecht et al suggests that the predominance of cervical osteochondromas is a function

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of the increased mobility and flexibility of these vertebrae, resulting in micro trauma to the

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underlying cartilaginous tissue and enabling aberrant growth.[1] Fiumara and colleagues contend that mobility is likely not an issue, given the paucity of cases documented within the amply mobile lumbar spine. They propose that the rate of ossification processes within the secondary centers influences the distribution of the lesions.[17]

CT, MRI, and radiographs can each be used to characterize osteochondromas. Radiographs of the lesions typically depict sessile osseous projections emanating from the posterior elements of the

ACCEPTED MANUSCRIPT spine.[1, 6] The cortex and spongiosa of the tumors are continuous with the original bone, serving as imaging identification markers. CT is the preferred imaging modality given the complex anatomy of the vertebrae. It can detect intervertebral foramen intrusion as well as the lesion’s site of origin.[6, 29] Used in conjunction with MRI, which can detect the size of the cartilaginous cap

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and delineate the surrounding soft tissue, the two imaging modalities provide a robust picture of

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the tumor’s immediate microenvironment.

Neurological symptoms are uncommon given the infrequency with which these lesions present

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with cord compression.[33] These lesions typically affect the posterior portion of the spinal

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column away from the neural elements.[29] However, the incidence of spinal cord compression in patients with multiple hereditary exostosis is greater.[40] Good clinical outcomes following

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surgical removal of the lesions have been reported with low rates of morbidity.[1, 4, 6, 17]

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Removal of the entire cartilaginous sheath surrounding the lesion is necessary to prevent

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recurrence of the lesion.[33, 41]

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In our case report, this patient had a facet joint osteochondroma extending from the bottom of the thoracic spine (T11-T12) into the spinal canal. The occurrence of such lesions affecting the facet

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joint is exceedingly low, but has been reported upon in the literature.[5] A posterolateral approach was used to resect the lesion and decompress the neural foramen and spinal canal from a single vantage point in an attempt to minimize postoperative complications seen in posterior approaches.[42]

ACCEPTED MANUSCRIPT CONCLUSION

Osteochondromas are benign bone tumors that are primarily found within the appendicular skeleton. They comprise an acutely small subset of all tumors in the spine. We present a case in which a patient with a thoracic facet joint osteochondroma was treated via a costotransversectomy,

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laminectomy with foraminotomy and fusion, and pedicular fixation. Post-operative resolution of

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symptoms with no local recurrence of these lesions was noted at his three-year follow up. This

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case supports the efficacy of surgical resection for the treatment of spinal osteochondromas.

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ACCEPTED MANUSCRIPT ACKNOWLEDGMENTS AND FUNDING SOURCES

Funding Statement This research did not receive any specific grant from funding agencies in the public, commercial,

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or not-for-profit sectors.

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Conflict of Interest Statement

The authors have no personal, financial, or institutional interest with regards to the authorship

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and/or publication of this manuscript.

ACCEPTED MANUSCRIPT FIGURE LEGENDS

Figure 1: Sagittal STIR imaging (left) demonstrates a mass measuring 3.1 x 1.5 x 2.7 cm centered at the T11-T12 level. Post contrast sagittal T1 weighted imaging (right) demonstrates heterogeneous hyperintense signal with minimal peripheral enhancement.

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Figure 2: Sagittal CT (left) and axial CT demonstrates an ossified mass from the left facet joint at

Figure 3: (A) Fibrous perichondrium (

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T11-T12 projecting medially into the spinal canal with moderate to severe central canal stenosis.

) overlies the cartilaginous cap of an osteochondroma (

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), and it mimics the zone of hypertrophy of a growth plate. The base of the cap is being sealed by bone, indicating cessation of growth (

). (B) The chondrocytes in the cartilaginous cap are

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arranged in columns and as they approach the base, they mature and acquire more cytoplasm.

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Figure 1

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Figure 2

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Figure 3