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Original Article

The Clinical Outcomes of Cervical Spine Chordoma: A Nationwide Multicenter Retrospective Study

Hangeul Park1orcid, Yunhee Choi2orcid, Sungjoon Lee3orcid, Sun-Ho Lee3orcid, Eun-Sang Kim3orcid, Sun Woo Jang4orcid, Jin Hoon Park4orcid, Yunseong Cho5orcid, Giwuk Jang5orcid, Yoon Ha5orcid, Yun-Sik Dho6orcid, Heon Yoo6orcid, Sung Uk Lee7orcid, Seung-Ho Seo8orcid, Ki-Jeong Kim8orcid, Seil Sohn9orcid, Chun Kee Chung10orcid
Neurospine 2024;21(3):942-953.
Published online: September 30, 2024

1Department of Neurosurgery, Seoul National University Hospital, Seoul, Korea

2Division of Medical Statistics, Medical Research Collaborating Center, Seoul National University Hospital, Seoul, Korea

3Department of Neurosurgery, Samsung Medical Center, Seoul, Korea

4Department of Neurosurgery, Asan Medical Center, Seoul, Korea

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8Department of Neurosurgery, Seoul National University Bundang Hospital, Seongnam, Korea

9Department of Neurosurgery, CHA Bundang Medical Center, Seongnam, Korea

10Neuroscience Research Institute, Seoul National University College of Medicine, Seoul, Korea

Corresponding Author Chun Kee Chung Neuroscience Research Institute, Seoul National University College of Medicine, 103 Daehak-ro, Jongno-gu, Seoul 03080, Korea Email: chungc@snu.ac.kr
• Received: May 1, 2024   • Revised: June 26, 2024   • Accepted: July 2, 2024

Copyright © 2024 by the Korean Spinal Neurosurgery Society

This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

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The Clinical Outcomes of Cervical Spine Chordoma: A Nationwide Multicenter Retrospective Study
Neurospine. 2024;21(3):942-953.   Published online September 30, 2024
DOI: https://doi.org/10.14245/ns.2448448.224
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The Clinical Outcomes of Cervical Spine Chordoma: A Nationwide Multicenter Retrospective Study
Neurospine. 2024;21(3):942-953.   Published online September 30, 2024
DOI: https://doi.org/10.14245/ns.2448448.224
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The Clinical Outcomes of Cervical Spine Chordoma: A Nationwide Multicenter Retrospective Study
Image Image Image
Fig. 1. The distribution of cervical spine chordomas. (A) The distribution of vertebral body involvement in cervical spine chordoma is as follows (per total number of vertebral involvement, 106): C2 involvement was the most common, observed in 34 patients (32.1%, 34 out of 106), followed by C3 involvement in 27 patients (25.5%, 27 out of 106). C1 involvement was noted in 15 patients (14.2%), while C4 involvement was seen in 12 patients (11.3%). Involvement of C5 was observed in 6 patients (5.7%). Both C6 and C7 showed involvement in 5 patients each (4.7%). Additionally, C0 and T1 involvement were each observed in 1 patient (0.9%). C0 and T1 level chordomas were cases where cervical spine chordoma extended to the C0 and T1 levels, respectively. (B) The distribution of epicenter for cervical spine chordomas is as follows (per total number of patients, 45): C2 was the most common, observed in 30 patients (66.7%), followed by C3 in 5 patients (11.1%). C4 and C5 were involved in 3 patients (6.7%) each, while C6 was involved in 2 patients (4.4%). C1 and C7 were the epicenter in 1 patient (2.2%) each.
Fig. 2. Schematic overview of the treatment and outcomes for 45 cervical spine chordomas. Among a total of 45 patients, 3 underwent biopsy, 2 of whom were lost to follow-up (FU), and the remaining one received proton therapy (PT). The other 42 patients underwent operation (OP). One patient received neoadjuvant therapy (Neoadj) and OP, but experienced a recurrence, leading to a secondary OP and adjuvant therapy (adj). Following another recurrence and the detection of distant metastasis, the patient underwent stereotactic radiosurgery (SRS) and targeted therapy (TT). Of the 11 patients who underwent OP alone, 2 were lost to FU, and recurrence occurred in 6 patients. One of the patients who experienced a recurrence was subsequently lost to FU. One patient experienced a recurrence after secondary OP and subsequently underwent tertiary OP. Four patients received secondary OP and adj; of these, 2 experienced further recurrences, 1 was lost to FU, and another underwent tertiary OP and adj. Thirty patients received OP and adj, with 14 experiencing recurrences; 3 of these were lost to FU. Two patients received radiation therapy (RT), 1 of whom experienced a recurrence and was subsequently lost to FU. Five patients underwent secondary OP; of these, one had a recurrence and received tertiary OP, while another, diagnosed with distant metastasis, underwent RT, PT, and chemotherapy (CT). Four patients underwent secondary OP and adj, all of whom experienced recurrences. One patient underwent tertiary OP following a recurrence and subsequently required quaternary OP. Another patient received RT and, after a recurrence, underwent tertiary OP. Recurrences and distant metastasis were confirmed in 2 patients: one received RT and SRS, and the other underwent tertiary OP and SRS, followed by recurrence and additional SRS. Bx, biopsy; SD, stable disease; OS, overall survival; PD, progressive disease.
Fig. 3. Cumulative incidence of local recurrence (LR) and overall survival of cervical spine chordomas. (A) The cumulative incidence of LR among 42 patients with cervical spine chordoma was reported as follows: the 5-year cumulative LR rate was 51.3% (95% confidence interval [CI], 32.8%–67.1%), and the 10-year cumulative LR rate was 60% (95% CI, 34.7%–78.2%). (B) The overall survival (OS) after initial surgery for 41 patients with cervical spine chordoma was as follows: the 5-year OS rate was 82% (95% CI, 64.1%–91.8%), and the 10-year OS rate was 53% (95% CI, 30.5%–71.3%). (C) Comparing OS after initial surgery between patients aged 46 years and older and those under 46 years old (p<0.005). The median survival for patients under 46 years was 81.6 months, while the median survival for those aged 46 years and older was 51.6 months. (D) Comparing OS after initial surgery in patients 20 years of age or younger and those 60 years of age or older (p=0.039). The median survival for patients 20 years or younger was 81.6 months, while the median survival for those 60 years or older was 46.8 months.

Fig. 1.

Fig. 2.

Fig. 3.

The Clinical Outcomes of Cervical Spine Chordoma: A Nationwide Multicenter Retrospective Study
Variable Value
Age (yr) 46.4 ± 22.0 (7–86)
Male sex 30 (66.7)
Clinical presenting symptom
 Pain 31/44 (70.5)
 Motor deficit 11/44 (25)
 Sensory deficit 8/44 (18.2)
 Local mass effects 6/44 (13.6)
 Incidental 2/44 (4.5)
Duration of illness (mo) (n = 39) 3.9 (2.0–8.0)
Radiological parameters
 Maximal tumor diameter (cm) (n = 41) 3.9 ± 1.7 (1.0–9.4)
 Number of involved vertebral bodies 2 (2–3)
 Radial distribution by WBB staging (n = 41) 6 (4–9)
 Concentric distribution by WBB staging (n = 41) 4 (3–4)
 Vertebral artery canal involvement (n = 41) 28 (68.3)
Variable Value
Biopsy only 3 (6.7)
Preoperative vertebral artery embolization 2/40 (5.0)
Neoadjuvant radiation 1 (2.2)
Surgical approach
 Anterior 10/42 (23.8)
 Posterior 16/42 (38.1)
 Staged 16/42 (38.1)
Adjuvant therapy
 Radiation 17/30 (56.7)
 Proton 11/30 (36.7)
 Combined radiation and proton 1/30 (3.3)
 Radiosurgery 1/30 (3.3)
Treatment after 1st local recurrence
 Surgery 6/21 (28.6)
 Surgery and adjuvant therapy 9/21 (42.9)
 Radiation 2/21 (9.5)
 Follow-up loss 4/21 (19.0)
Treatment after 2nd local recurrence
 Surgery 3/9 (33.3)
 Surgery and adjuvant therapy 1/9 (11.1)
 Radiation or radiosurgery 4/9 (44.4)
 Follow-up loss 1/9 (11.1)
Treatment after 3rd local recurrence
 Surgery 2/3 (66.7)
 Radiosurgery 1/3 (33.3)
Treatment for distant metastasis
 Surgery 1/4 (25.0)
 Radiation and radiosurgery 1/4 (25.0)
 Targeted therapy 1/4 (25.0)
 Radiation and proton, chemotherapy 1/4 (25.0)
Variable Value
Degree of resection
 En bloc resection 2/40 (5)
 Intralesional total resection 12/40 (30)
 STR 14/40 (35)
 PR 12/40 (30)
Improvement of preoperative symptoms
 Pain 22/30 (73.3)
 Motor deficit 8/10 (80.0)
 Sensory deficit 5/7 (71.4)
 Local mass effects 3/3 (100)
Complication
 Death 1/42 (2.4)
 Instrument failure 1/42 (2.4)
 CSF leakage 1/42 (2.4)
 Pontine infarction 1/42 (2.4)
 Meningitis 1/42 (2.4)
 Vocal cord palsy 1/42 (2.4)
Follow-up duration (mo) 52.0 (31.6–88.2)
Local recurrence 21 (46.7)
Distant metastasis 4 (8.9)
 The period from initial surgery to distant metastasis (mo) 27 (12.5–34.7)
Death 16 (35.6)
Last follow-up status
 SD 36 (80.0)
 PD 9 (20.0)
Variable Univariate
 p-value
sHR 95% CI
Age 1.00 0.98–1.02 0.838
Male sex 0.87 0.35–2.18 0.765
Duration of illness 0.98 0.93–1.03 0.349
Maximal tumor diameter 1.04 0.80–1.34 0.796
No. of involved vertebral bodies 1.08 0.68–1.73 0.744
Radial distribution by WBB staging 1.03 0.90–1.19 0.636
Concentric distribution by WBB staging 0.93 0.50–1.75 0.824
Surgery and perioperative adjuvant therapy 0.58 0.22–1.54 0.276
Total resection 0.80 0.32–2.00 0.634
Variable Univariate
 p-value Multivariate
 p-value
HR 95% CI HR 95% CI
Age 1.05 1.01–1.08 0.008 1.04 1.01–1.07 0.015
Male 0.91 0.31–2.68 0.869 - - -
Duration of illness 1.00 0.95–1.05 0.861 - - -
Maximal tumor diameter 1.27 0.94–1.72 0.118 1.44 0.97–2.14 0.073
Number of involved vertebral bodies 0.78 0.38–1.64 0.514 - - -
Radial distribution by WBB staging 0.91 0.75–1.11 0.346 - - -
Concentric distribution by WBB staging 1.26 0.66–2.41 0.484 - - -
Surgery and perioperative adjuvant therapy 0.87 0.28–2.73 0.808 - - -
Total resection 0.49 0.14–1.73 0.268 - - -
Table 1. Baseline characteristics (N=45)

Values are presented as mean±standard deviation (range), number (%), or median (interquartile range).

WBB, Weinstein-Boriani-Biagnini.

Table 2. Details of treatment protocol in cervical spine chordoma (N=45)

Values are presented as number (%).

Table 3. Oncologic outcomes of cervical spine chordoma (N=45)

Values are presented as number (%) or median (interquartile range).

STR, subtotal resection; PR, partial resection; CSF, cerebrospinal fluid; SD, stable disease; PD, progressive disease.

Table 4. Fine-Gray subdistribution hazard model for local recurrence

sHR, subdistribution hazard ratio; CI, confidence interval; WBB, Weinstein-Boriani-Biagnini.

Table 5. Cox proportional hazard model for death

HR, hazard ratio; CI, confidence interval; WBB, Weinstein-Boriani-Biagnini.

Table 1.

Table 2.

Table 3.

Table 4.

Table 5.

NS : Neurospine
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