Abstract
En bloc spondylectomy is the gold standard for surgical resection of sacral chordomas (CHO), but the effect of extent of resection on recurrence and survival in patients with CHO of the cervical spine remains elusive. MEDLINE, Embase, Scopus, and Cochrane were systematically reviewed. Patients with cervical CHO treated at three tertiary-care academic institutions were reviewed for inclusion. We performed an individual participant data meta-analysis to assess the overall survival (OS) and progression free survival (PFS) after en bloc-gross total resection (GTR) and intralesional-GTR compared to subtotal resection (STR). We then performed an intention-to-treat analysis including all patients with attempted en bloc resection in the en bloc group, regardless of the surgical margins. There was a total of 13 series including 161 patients with cervical CHO, including our current series of 22 patients. GTR (en bloc-GTR + intralesional-GTR) was associated with a significant decrease in the risk of local progression (pooled hazard ratio (PHR) = 0.22; 95% CI 0.08-0.59; p = 0.003) and risk of death (PHR 0.31; 95%; CI 0.12-0.83; p = 0.020). A meta-regression analyses determined that intralesional-GTR improved PFS (PHR 0.35; 95% CI 0.16-0.76; p = 0.009) as well as OS (PHR 0.25; 95% CI 0.08-0.79; p = 0.019) when compared to STR. En bloc-GTR was associated with a significant reduction in the risk of local progression (PHR 0.06; 95% CI 0.01-0.77; p = 0.030), but not a decreased OS (PHR 0.50; 95% CI 0.19-1.27; p = 0.145). Our intention-to-treat analyses revealed a near significant improvement in OS for the en bloc group (PHR: 0.15; 95% CI 0.02-1.22; p = 0.054), and nearly identical improvement in PFS. Radiation data was not available for the studies included in the meta-analysis. This is the first and only meta-analysis of patients with cervical CHO. We found that both en bloc-GTR and intralesional-GTR resulted in improved local tumor control when compared to STR.
Highlights
Chordomas (CHO) are the most common primary malignant spinal bone tumor with an age-adjusted incidence of 0.088 per 100,000 persons per year. [1][2] CHOs of the cervical spine are especially troublesome, as close association or even juxtaposition of critical structures such as the vertebral arteries, esophagus, cervical nerve roots, or the spinal cord itself make surgery a daunting task
A meta-regression analyses determined that intralesional-Gross total resection (GTR) improved progression free survival (PFS) (PHR = 0.35; 95%confidence intervals (CI) = 0.16–0.76; p = 0.009) as well as overall survival (OS) (PHR = 0.25; 95%CI = 0.08–0.79; p = 0.019) when compared to subtotal resection (STR)
En bloc-GTR was associated with a significant reduction in the risk of local progression (PHR = 0.06; 95%CI = 0.01–0.77; p = 0.030), but not a decreased OS (PHR = 0.50; 95% CI = 0.19–1.27; p = 0.145)
Summary
Chordomas (CHO) are the most common primary malignant spinal bone tumor with an age-adjusted incidence of 0.088 per 100,000 persons per year. [1][2] CHOs of the cervical spine are especially troublesome, as close association or even juxtaposition of critical structures such as the vertebral arteries, esophagus, cervical nerve roots, or the spinal cord itself make surgery a daunting task. Gross total resection (GTR) can be either intralesional, with piecemeal removal of the tumor, or en bloc, there is ample evidence that en bloc resection results in improved local control for patients with sacral CHOs [4, 5][6] The sin qua non of chordoma management is en bloc resection of the tumor, as CHO will nearly inevitably return unless it is resected with clear margins This can typically be accomplished with tedious pre-surgical planning, using the preoperative imaging as a guide. Because any single case series would be unlikely to result in any useful data, a meta-analysis is necessary for this rare but debilitating tumor In this manuscript, we review our series of patients with cervical CHO at three academic tertiary care institutions, and perform a comprehensive systematic review and meta-analysis of previously published reports to form the highest level of evidence to assess the influence of extent of resection on progression free survival (PFS) and overall survival (OS) for patients with cervical CHO. En bloc spondylectomy is the gold standard for surgical resection of sacral chordomas (CHO), but the effect of extent of resection on recurrence and survival in patients with CHO of the cervical spine remains elusive
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