Abstract
Purpose
Sacral chordomas (SC) are rare, locally invasive, malignant neoplasms. Despite surgical resection and adjuvant therapies, local recurrence (LR) is common and overall survival (OS) is poor. The objective of this study was to identify prognostic factors that have an impact on the local recurrence-free survival (LRFS) and OS of patients with SC.
Methods
Utilizing the AOSpine Knowledge Forum Tumor multicenter ambispective database, surgically treated SC cases were identified. Cox regression modeling was used to assess the effect of several clinically relevant variables on OS and LRFS.
Results
A total of 167 patients with surgically treated SC were identified. The male/female ratio was 98/69 with a mean age of 57 ± 15 years at the time of surgery. The LR was 35 % (n = 57), death occurred in 30 % of patients (n = 50) during the study period. The median OS was 6 years post-surgery and LRFS was 4 years. In the univariate analysis, previous tumor surgery at the same site (P = 0.002), intralesional resection (P < 0.001), and larger tumor volume (P = 0.030) were significantly associated with LR. Increasing age (P < 0.001) and a preoperative motor deficit of C or D (P = 0.003) were significantly associated with poor OS, and nerve root sacrifice showed a trend towards significance (P = 0.088). In the multivariate models, previous surgery and intralesional resection were significantly related to LR, while increasing age and motor deficit of C or D were associated with poor OS.
Conclusions
This study identified two predictive variables for LRFS (previous tumor surgery and type of surgical resection) and two for OS (age and impaired motor function) in surgically treated SC patients. Our results indicate that en bloc resection reduces LR but does not influence OS. However, this was likely due to short follow-up (3.2 years).
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References
Walcott BP, Nahed BV, Mohyeldin A, Coumans JV, Kahle KT, Ferreira MJ (2012) Chordoma: current concepts, management, and future directions. Lancet Oncol 13(2):e69–e76. doi:10.1016/S1470-2045(11)70337-0
Ropper AE, Cahill KS, Hanna JW, McCarthy EF, Gokaslan ZL, Chi JH (2012) Primary vertebral tumors: a review of epidemiologic, histological and imaging findings, part II: locally aggressive and malignant tumors. Neurosurgery 70(1):211–219. doi:10.1227/NEU.0b013e31822d5f17 (discussion 219)
Fourney DR, Gokaslan ZL (2003) Current management of sacral chordoma. Neurosurg Focus 15(2):E9
Fourney DR, Rhines LD, Hentschel SJ, Skibber JM, Wolinsky JP, Weber KL, Suki D, Gallia GL, Garonzik I, Gokaslan ZL (2005) En bloc resection of primary sacral tumors: classification of surgical approaches and outcome. J Neurosurg Spine 3(2):111–122. doi:10.3171/spi.2005.3.2.0111
Hsu W, Kosztowski TA, Zaidi HA, Dorsi M, Gokaslan ZL, Wolinsky JP (2009) Multidisciplinary management of primary tumors of the vertebral column. Curr Treat Options Oncol 10(1–2):107–125. doi:10.1007/s11864-009-0102-8
Varga PP, Szövérfi Z, Lazary A (2014) Surgical treatment of primary malignant tumors of the sacrum. Neurol Res 36(6):577–587. doi:10.1179/1743132814Y.0000000366
Ahmed AR (2009) Safety margins in resection of sacral chordoma: analysis of 18 patients. Arch Orthop Trauma Surg 129(4):483–487. doi:10.1007/s00402-008-0674-y
Hanna SA, Aston WJ, Briggs TW, Cannon SR, Saifuddin A (2008) Sacral chordoma: can local recurrence after sacrectomy be predicted? Clin Orthop Relat Res 466(9):2217–2223. doi:10.1007/s11999-008-0356-7
York JE, Kaczaraj A, Abi-Said D, Fuller GN, Skibber JM, Janjan NA, Gokaslan ZL (1999) Sacral chordoma: 40-year experience at a major cancer center. Neurosurgery 44(1):74–79 (discussion 79–80)
Fuchs B, Dickey ID, Yaszemski MJ, Inwards CY, Sim FH (2005) Operative management of sacral chordoma. J Bone Joint Surg Am 87(10):2211–2216. doi:10.2106/JBJS.D.02693
Hulen CA, Temple HT, Fox WP, Sama AA, Green BA, Eismont FJ (2006) Oncologic and functional outcome following sacrectomy for sacral chordoma. J Bone Joint Surg Am 88(7):1532–1539. doi:10.2106/JBJS.D.02533
Atalar H, Selek H, Yildiz Y, Saglik Y (2006) Management of sacrococcygeal chordomas. Int Orthop 30(6):514–518. doi:10.1007/s00264-006-0095-x
Baratti D, Gronchi A, Pennacchioli E, Lozza L, Colecchia M, Fiore M, Santinami M (2003) Chordoma: natural history and results in 28 patients treated at a single institution. Ann Surg Oncol 10(3):291–296
Kayani B, Hanna SA, Sewell MD, Saifuddin A, Molloy S, Briggs TW (2014) A review of the surgical management of sacral chordoma. Eur J Surg Oncol. doi:10.1016/j.ejso.2014.04.008
Ruggieri P, Angelini A, Ussia G, Montalti M, Mercuri M (2010) Surgical margins and local control in resection of sacral chordomas. Clin Orthop Relat Res 468(11):2939–2947. doi:10.1007/s11999-010-1472-8
McGirt MJ, Gokaslan ZL, Chaichana KL (2011) Preoperative grading scale to predict survival in patients undergoing resection of malignant primary osseous spinal neoplasms. Spine J 11(3):190–196. doi:10.1016/j.spinee.2011.01.013
Samson IR, Springfield DS, Suit HD, Mankin HJ (1993) Operative treatment of sacrococcygeal chordoma. A review of twenty-one cases. J Bone Joint Surg Am 75(10):1476–1484
Bergh P, Kindblom LG, Gunterberg B, Remotti F, Ryd W, Meis-Kindblom JM (2000) Prognostic factors in chordoma of the sacrum and mobile spine: a study of 39 patients. Cancer 88(9):2122–2134
Schwab JH, Healey JH, Rose P, Casas-Ganem J, Boland PJ (2009) The surgical management of sacral chordomas. Spine 34(24):2700–2704. doi:10.1097/BRS.0b013e3181bad11d
Cheng EY, Ozerdemoglu RA, Transfeldt EE, Thompson RC Jr (1999) Lumbosacral chordoma. Prognostic factors and treatment. Spine 24(16):1639–1645
Szövérfi Z, Lazary A, Bozsódi Á, Klemencsics I, Éltes PE, Varga PP (2014) Primary spinal tumor mortality score (PSTMS): a novel scoring system for predicting poor survival. Spine J 14(11):2691–2700. doi:10.1016/j.spinee.2014.03.009
Boriani S, Saravanja D, Yamada Y, Varga PP, Biagini R, Fisher CG (2009) Challenges of local recurrence and cure in low grade malignant tumors of the spine. Spine 34(22 Suppl):S48–S57. doi:10.1097/BRS.0b013e3181b969ac
Fisher CG, Goldschlager T, Boriani S, Varga PP, Fehlings MG, Bilsky MH, Dekutoski MB, Luzzati A, Williams RP, Berven S, Chou D, Reynolds JJ, Quraishi NA, Rhines LD, Bettegowda C, Gokaslan ZL (2013) A novel scientific model for rare and often neglected neoplastic conditions. Evid-Based Spine-Care J 4(2):160–162. doi:10.1055/s-0033-1357365
von Elm E, Altman DG, Egger M, Pocock SJ, Gotzsche PC, Vandenbroucke JP (2007) The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies. Lancet 370(9596):1453–1457. doi:10.1016/S0140-6736(07)61602-X
Harris PA, Taylor R, Thielke R, Payne J, Gonzalez N, Conde JG (2009) Research electronic data capture (REDCap)–a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform 42(2):377–381. doi:10.1016/j.jbi.2008.08.010
Enneking WF, Spanier SS, Goodman MA (1980) A system for the surgical staging of musculoskeletal sarcoma. Clin Orthop Relat Res 153:106–120
Fisher CG, Saravanja DD, Dvorak MF, Rampersaud YR, Clarkson PW, Hurlbert J, Fox R, Zhang H, Lewis S, Riaz S, Ferguson PC, Boyd MC (2011) Surgical management of primary bone tumors of the spine: validation of an approach to enhance cure and reduce local recurrence. Spine 36(10):830–836. doi:10.1097/BRS.0b013e3181e502e5
Ruggieri P, Angelini A, Pala E, Mercuri M (2012) Infections in surgery of primary tumors of the sacrum. Spine 37(5):420–428. doi:10.1097/BRS.0b013e3182213a44
Tokuhashi Y, Matsuzaki H, Toriyama S, Kawano H, Ohsaka S (1990) Scoring system for the preoperative evaluation of metastatic spine tumor prognosis. Spine 15(11):1110–1113
Treatment of spinal metastases and MSCC (2008) In: NICE Clinical Guidelines, No. 75. Metastatic spinal cord compression: diagnosis and management of patients at risk of or with metastatic spinal cord compression National Institute for Health and Clinical Excellence: Guidance, Cardiff, p 29
Peduzzi P, Concato J, Feinstein AR, Holford TR (1995) Importance of events per independent variable in proportional hazards regression analysis. II. Accuracy and precision of regression estimates. J Clin Epidemiol 48(12):1503–1510
Smoll NR, Gautschi OP, Radovanovic I, Schaller K, Weber DC (2013) Incidence and relative survival of chordomas: the standardized mortality ratio and the impact of chordomas on a population. Cancer 119(11):2029–2037. doi:10.1002/cncr.28032
Acknowledgments
We are grateful to the collaborating centers’ local clinical research personnel and support staff for their active participation. This study was organized and funded by AOSpine International, through the AOSpine Knowledge Forum Tumor, a pathology-focused working group of up to ten international spine experts acting on behalf of AOSpine in the domain of scientific expertise. Study support was provided directly through AOSpine’s Research department and AO’s Clinical Investigation and Documentation unit.
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The authors have no financial conflicts of interest.
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P. P. Varga and Z. Szövérfi are co-first authors.
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Varga, P.P., Szövérfi, Z., Fisher, C.G. et al. Surgical treatment of sacral chordoma: prognostic variables for local recurrence and overall survival. Eur Spine J 24, 1092–1101 (2015). https://doi.org/10.1007/s00586-014-3728-6
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DOI: https://doi.org/10.1007/s00586-014-3728-6