Long-Term Outcomes of High-Dose Single-Fraction Radiosurgery for Chordomas of the Spine and Sacrum

Abstract INTRODUCTION To evaluate outcomes of patients with primary chordomas treated with spine stereotactic radiosurgery (SRS) alone or in combination with surgery, drawing from a single-institution database to elucidate treatment options associated with durable radiographic control of these conventionally radioresistant tumors. Chordomas result in significant morbidity, with a high rate of local recurrence and potential for metastases. SRS as a primary treatment could save patients from extensive surgery. Spine SRS outcomes support exploration of its role in the durable control of these conventionally radioresistant tumors. METHODS Clinical records were reviewed for outcomes of patients with primary chordoma of the mobile spine and sacrum who underwent single-fraction SRS between 2006 and 2017. Radiographic local recurrence-free survival (LRFS), overall survival (OS), symptom response, and toxicity were assessed in relation to extent of surgery. RESULTS In total, 35 patients with de novo chordoma of the mobile spine (49%) and sacrum (51%) received SRS with a median post-SRS follow-up of 38.8 mo (range: 2.0-122.9). The median PTV dose was 24Gy (range: 18–24Gy). Overall, 12 patients (33%) underwent definitive SRS and 23 patients (66%) underwent surgery followed by adjuvant SRS. Surgical strategies included separation surgery prior to SRS, curettage/intralesional resection, and en bloc resection in 7, 6, and 10 patients, respectively. The 3- and 5-yr LRFS rates were 86.2% and 80.5% respectively. Among 32 patients receiving 24 Gy (91%), the 3- and 5-yr LRFS were 96.3% and 89.9%. The 3- and 5-yr OS rates were 90.0% and 84.3%. The symptom response rate to treatment was 88% for pain and radiculopathy. Extent or type of surgery was not associated with LRFS, OS, or symptom response rates (P > .05), but en bloc resection was associated with higher CTCAE v. 5.0 surgical toxicity compared to epidural decompression and curettage/intralesional resection (P = .03). The long-term = grade 2 SRS toxicity rate was 31%, including 17% grade 3 tissue necrosis, recurrent laryngeal nerve palsy, fracture, and secondary malignancy. CONCLUSION High-dose spine SRS offers the chance of durable radiographic control and effective symptom relief with acceptable toxicity for primary chordomas as either definitive or adjuvant therapy.

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Long-term outcomes of high-dose single-fraction radiosurgery for chordomas of the spine and sacrum

Journal of Neurosurgery Spine ◽

10.3171/2019.7.spine19515 ◽

2020 ◽

Vol 32 (1) ◽

pp. 79-88 ◽

Cited By ~ 2

Author(s):

Chunzi Jenny Jin ◽

John Berry-Candelario ◽

Anne S. Reiner ◽

Ilya Laufer ◽

Daniel S. Higginson ◽

...

Keyword(s):

Local Recurrence ◽

En Bloc Resection ◽

Bloc Resection ◽

High Dose ◽

En Bloc ◽

Single Fraction ◽

Mobile Spine ◽

Symptom Response ◽

Clinical Records

OBJECTIVEThe current treatment of chordomas is associated with significant morbidity, high rates of local recurrence, and the potential for metastases. Stereotactic radiosurgery (SRS) as a primary treatment could reduce the need for en bloc resection to achieve wide or marginal margins. Spinal SRS outcomes support the exploration of SRS’s role in the durable control of these conventionally radioresistant tumors. The goal of the study was to evaluate outcomes of patients with primary chordomas treated with spinal SRS alone or in combination with surgery.METHODSClinical records were reviewed for outcomes of patients with primary chordomas of the mobile spine and sacrum who underwent single-fraction SRS between 2006 and 2017. Radiographic local recurrence-free survival (LRFS), overall survival (OS), symptom response, and toxicity were assessed in relation to the extent of surgery.RESULTSIn total, 35 patients with de novo chordomas of the mobile spine (n = 17) and sacrum (n = 18) received SRS and had a median post-SRS follow-up duration of 38.8 months (range 2.0–122.9 months). The median planning target volume dose was a 24-Gy single fraction (range 18–24 Gy). Overall, 12 patients (34%) underwent definitive SRS and 23 patients (66%) underwent surgery and either neoadjuvant or postoperative adjuvant SRS. Definitive SRS was selectively used to treat both sacral (n = 7) and mobile spine (n = 5) chordomas. Surgical strategies for the mobile spine were either intralesional, gross-total resection (n = 5) or separation surgery (n = 7) and for the sacrum en bloc sacrectomy (n = 11). The 3- and 5-year LRFS rates were 86.2% and 80.5%, respectively. Among 32 patients (91%) receiving 24-Gy radiation doses, the 3- and 5-year LRFS rates were 96.3% and 89.9%, respectively. The 3- and 5-year OS rates were 90.0% and 84.3%, respectively. The symptom response rate to treatment was 88% for pain and radiculopathy. The extent or type of surgery was not associated with LRFS, OS, or symptom response rates (p > 0.05), but en bloc resection was associated with higher surgical toxicity, as measured using the Common Terminology Criteria for Adverse Events (version 5.0) classification tool, than epidural decompression and curettage/intralesional resection (p = 0.03). The long-term rate of toxicity ≥ grade 2 was 31%, including 20% grade 3 tissue necrosis, recurrent laryngeal nerve palsy, myelopathy, fracture, and secondary malignancy.CONCLUSIONSHigh-dose spinal SRS offers the chance for durable radiological control and effective symptom relief with acceptable toxicity in patients with primary chordomas as either a definitive or adjuvant therapy.

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Mobile spine chordoma: results of 166 patients from the AOSpine Knowledge Forum Tumor database

Journal of Neurosurgery Spine ◽

10.3171/2015.7.spine15201 ◽

2016 ◽

Vol 24 (4) ◽

pp. 644-651 ◽

Cited By ~ 30

Author(s):

Ziya L. Gokaslan ◽

Patricia L. Zadnik ◽

Daniel M. Sciubba ◽

Niccole Germscheid ◽

C. Rory Goodwin ◽

...

Keyword(s):

Local Recurrence ◽

Survival Data ◽

Large Scale ◽

Surgical Techniques ◽

En Bloc Resection ◽

Bloc Resection ◽

En Bloc ◽

Increased Risk ◽

Significant Difference ◽

Mobile Spine

OBJECT A chordoma is an indolent primary spinal tumor that has devastating effects on the patient's life. These lesions are chemoresistant, resistant to conventional radiotherapy, and moderately sensitive to proton therapy; however, en bloc resection remains the preferred treatment for optimizing patient outcomes. While multiple small and largely retrospective studies have investigated the outcomes following en bloc resection of chordomas in the sacrum, there have been few large-scale studies on patients with chordomas of the mobile spine. The goal of this study was to review the outcomes of surgically treated patients with mobile spine chordomas at multiple international centers with respect to local recurrence and survival. This multiinstitutional retrospective study collected data between 1988 and 2012 about prognosis-predicting factors, including various clinical characteristics and surgical techniques for mobile spine chordoma. Tumors were classified according to the Enneking principles and analyzed in 2 treatment cohorts: Enneking-appropriate (EA) and Enneking-inappropriate (EI) cohorts. Patients were categorized as EA when the final pathological assessment of the margin matched the Enneking recommendation; otherwise, they were categorized as EI. METHODS Descriptive statistics were used to summarize the data (Student t-test, chi-square, and Fisher exact tests). Recurrence and survival data were analyzed using Kaplan-Meier survival curves, log-rank tests, and multivariate Cox proportional hazard modeling. RESULTS A total of 166 patients (55 female and 111 male patients) with mobile spine chordoma were included. The median patient follow-up was 2.6 years (range 1 day to 22.5 years). Fifty-eight (41%) patients were EA and 84 (59%) patients were EI. The type of biopsy (p < 0.001), spinal location (p = 0.018), and if the patient received adjuvant therapy (p < 0.001) were significantly different between the 2 cohorts. Overall, 58 (35%) patients developed local recurrence and 57 (34%) patients died. Median survival was 7.0 years postoperative: 8.4 years postoperative for EA patients and 6.4 years postoperative for EI patients (p = 0.023). The multivariate analysis showed that the EI cohort was significantly associated with an increased risk of local recurrence in comparison with the EA cohort (HR 7.02; 95% CI 2.96–16.6; p < 0.001), although no significant difference in survival was observed. CONCLUSIONS EA resection plays a major role in decreasing the risk for local recurrence in patients with chordoma of the mobile spine.

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The long term surgical outcome of spinal giant cell tumor.

Journal of Clinical Oncology ◽

10.1200/jco.2019.37.15_suppl.e22510 ◽

2019 ◽

Vol 37 (15_suppl) ◽

pp. e22510-e22510

Author(s):

Joonho Yoon ◽

Chun Kee Chung ◽

Sunho Lee ◽

Chiheon Kim

Keyword(s):

Local Recurrence ◽

En Bloc Resection ◽

Bloc Resection ◽

Rank Test ◽

Radical Excision ◽

Total Removal ◽

En Bloc ◽

Additional Surgery ◽

Significant Difference ◽

Mobile Spine

e22510 Background: GCT is a benign and locally aggressive tumor that occur infrequently in the mobile spine. The radical excision is generally agreed for treatment of GCTs. Methods: We performed retrospective analysis of 20 GCTs in the mobile spine from 2005 to 2013. Verteberal involvement was classified with Weinstein-Boriani-Biagini grading system. Surgery types were classified into 3 groups; partial excision, gross total removal (GTR), and en-bloc resection. We analysis by GTR versus non-GTR group, and enbloc versus non-enbloc group in GTR group. The log rank test was used for comparison of factors, and a P value of less than 0.05 was deemed significant. Results: After first surgery of the 20 patients, 17 additional surgery performed for 10 (50%) patients. There were 7 (35%) local recurrences and 3 (15%) distant metastasis. The time to recurrence or progression of tumor ranged from 5 months to 39 months. Two (10%) patients had additional surgery for instrument failure. One (5%) patient died from the complication of the surgery. The follow up duration ranged from 24 months to 141 months. The local recurrence was associated with partial resection (P < 0.01) and history of recurrence (P = 0.39). There was no significant difference between non-en bloc GTR and en bloc GTR in local recurrence (p = 0.101). Conclusions: Gross total resection should be considered for GCTs of the mobile spine. The choice of en bloc resection must be balanced with risks of the procedure. En bloc resection or radical excision could be challenging in many cases. In that cases, the gross total removal by intralesional resection is considerable for local control.

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En Bloc Resection in Combination with High Dose Radiation Improves Patient Survival in Mobile Spine Chordoma

The Spine Journal ◽

10.1016/j.spinee.2013.07.192 ◽

2013 ◽

Vol 13 (9) ◽

pp. S68

Author(s):

Polina Osler ◽

Kathryn A. Hess ◽

Thomas F. DeLaney ◽

Al Ferreira ◽

Francis J. Hornicek ◽

...

Keyword(s):

Patient Survival ◽

En Bloc Resection ◽

Bloc Resection ◽

High Dose ◽

En Bloc ◽

Mobile Spine ◽

Dose Radiation

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Long-term outcomes in primary spinal osteochondroma: a multicenter study of 27 patients

Journal of Neurosurgery Spine ◽

10.3171/2014.10.spine14501 ◽

2015 ◽

Vol 22 (6) ◽

pp. 582-588 ◽

Cited By ~ 15

Author(s):

Daniel M. Sciubba ◽

Mohamed Macki ◽

Mohamad Bydon ◽

Niccole M. Germscheid ◽

Jean-Paul Wolinsky ◽

...

Keyword(s):

En Bloc Resection ◽

Bloc Resection ◽

Benign Tumors ◽

En Bloc ◽

Tumor Stages ◽

Mobile Spine ◽

Spinal Osteochondroma ◽

Stage 1

OBJECT Clinical outcomes in patients with primary spinal osteochondromas are limited to small series and sporadic case reports. The authors present data on the first long-term investigation of spinal osteochondroma cases. METHODS An international, multicenter ambispective study on primary spinal osteochondroma was performed. Patients were included if they were diagnosed with an osteochondroma of the spine and received surgical treatment between October 1996 and June 2012 with at least 1 follow-up. Perioperative prognostic variables, including patient age, tumor size, spinal level, and resection, were analyzed in reference to long-term local recurrence and survival. Tumor resections were compared using Enneking appropriate (EA) or Enneking inappropriate surgical margins. RESULTS Osteochondromas were diagnosed in 27 patients at an average age of 37 years. Twenty-two lesions were found in the mobile spine (cervical, thoracic, or lumbar) and 5 in the fixed spine (sacrum). Twenty-three cases (88%) were benign tumors (Enneking tumor Stages 1–3), whereas 3 (12%) exhibited malignant changes (Enneking tumor Stages IA–IIB). Sixteen patients (62%) underwent en bloc treatment—that is, wide or marginal resection—and 10 (38%) underwent intralesional resection. Twenty-four operations (92%) followed EA margins. No one received adjuvant therapy. Two patients (8%) experienced recurrences: one in the fixed spine and one in the mobile spine. Both recurrences occurred in latent Stage 1 tumors following en bloc resection. No osteochondroma-related deaths were observed. CONCLUSIONS In the present study, most patients underwent en bloc resection and were treated as EA cases. Both recurrences occurred in the Stage 1 tumor cohort. Therefore, although benign in character, osteochondromas still require careful management and thorough follow-up.

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Spinal stereotactic body radiotherapy following intralesional curettage with separation surgery for initial or salvage chordoma treatment

Neurosurgical FOCUS ◽

10.3171/2016.9.focus16373 ◽

2017 ◽

Vol 42 (1) ◽

pp. E4 ◽

Cited By ~ 15

Author(s):

Dennis T. Lockney ◽

Timothy Shub ◽

Benjamin Hopkins ◽

Natalie A. Lockney ◽

Nelson Moussazadeh ◽

...

Keyword(s):

Local Control ◽

Salvage Therapy ◽

En Bloc Resection ◽

Bloc Resection ◽

Local Control Rate ◽

High Dose ◽

En Bloc ◽

Intralesional Curettage ◽

Mobile Spine

OBJECTIVE Chordoma is a rare malignant tumor for which en bloc resection with wide margins is advocated as primary treatment. Unfortunately, due to anatomical constraints, en bloc resection to achieve wide or marginal margins is not feasible for many patients as the resulting morbidity would be prohibitive. The objective of this study was to evaluate the efficacy of intralesional curettage and separation surgery followed by spinal stereotactic body radiation therapy (SBRT) in patients with chordomas in the mobile spine. METHODS The authors performed a retrospective chart review of all patients with chordoma in the mobile spine treated from 2004 to 2016. Patients were identified from a prospectively collected database. Initially 22 patients were identified with mobile spine chordomas. With inclusion criteria of cytoreductive separation surgery followed closely by SBRT and a minimum of 6 months of follow-up imaging, 12 patients were included. Clinical and pathological characteristics of each patient were collected and data were analyzed. Patients were divided into two cohorts—those undergoing intralesional resection followed by SBRT as initial chordoma treatment at Memorial Sloan Kettering Cancer Center (MSKCC) (Cohort 1) and those undergoing salvage treatment following recurrence (Cohort 2). Treatment toxicities were classified according to the Common Terminology Criteria for Adverse Events version 4.03. Overall survival was analyzed using Kaplan-Meier analysis. RESULTS The 12 patients had a median post-SBRT follow-up time of 26 months. Cohort 1 had 5 patients with median post-SBRT follow-up time of 65.9 months and local control rate of 80% at last follow-up. Only one patient had disease progression, at 48.2 months following surgery and SBRT. Cohort 2 had 7 patients who had been treated at other institutions prior to undergoing both surgery and SBRT (salvage therapy) at MSKCC. The local control rate was 57.1% and the median follow-up duration was 10.7 months. One patient required repeat irradiation. Major surgery- and radiation-related complications occurred in 18% and 27% of patients, respectively. Epidural spinal cord compression scores were collected for each patient pre- and postoperatively. CONCLUSIONS The combination of surgery and SBRT provides excellent local control following intralesional curettage and separation surgery for chordomas in the mobile spine. Patients who underwent intralesional curettage and spinal SBRT as initial treatment had better disease control than those undergoing salvage therapy. High-dose radiotherapy may offer several biological benefits for tumor control.

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High-dose hypofractionated stereotactic body radiotherapy for spinal chordoma

Journal of Neurosurgery Spine ◽

10.3171/2021.2.spine202199 ◽

2021 ◽

pp. 1-10

Author(s):

Xuguang Chen ◽

Sheng-Fu L. Lo ◽

Chetan Bettegowda ◽

Daniel M. Ryan ◽

John M. Gross ◽

...

Keyword(s):

Stereotactic Body Radiotherapy ◽

En Bloc Resection ◽

Bloc Resection ◽

Surgical Margins ◽

High Dose ◽

Newly Diagnosed ◽

En Bloc ◽

Spinal Chordoma ◽

Mobile Spine

OBJECTIVE Spinal chordoma is locally aggressive and has a high rate of recurrence, even after en bloc resection. Conventionally fractionated adjuvant radiation leads to suboptimal tumor control, and data regarding hypofractionated regimens are limited. The authors hypothesized that neoadjuvant stereotactic body radiotherapy (SBRT) may overcome its intrinsic radioresistance, improve surgical margins, and allow preservation of critical structures during surgery. The purpose of this study is to review the feasibility and early outcomes of high-dose hypofractionated SBRT, with a focus on neoadjuvant SBRT. METHODS Electronic medical records of patients with spinal chordoma treated using image-guided SBRT between 2009 and 2019 at a single institution were retrospectively reviewed. RESULTS Twenty-eight patients with 30 discrete lesions (24 in the mobile spine) were included. The median follow-up duration was 20.8 months (range 2.3–126.3 months). The median SBRT dose was 40 Gy (range 15–50 Gy) in 5 fractions (range 1–5 fractions). Seventeen patients (74% of those with newly diagnosed lesions) received neoadjuvant SBRT, of whom 15 (88%) underwent planned en bloc resection, all with negative margins. Two patients (12%) developed surgical wound-related complications after neoadjuvant SBRT and surgery, and 4 (two grade 3 and two grade 2) experienced postoperative complications unrelated to the surgical site. Of the remaining patients with newly diagnosed lesions, 5 received adjuvant SBRT for positive or close surgical margins, and 1 received SBRT alone. Seven recurrent lesions were treated with SBRT alone, including 2 after failure of prior conventional radiation. The 2-year overall survival rate was 92% (95% confidence interval [CI] 71%–98%). Patients with newly diagnosed chordoma had longer median survival (not reached) than those with recurrent lesions (27.7 months, p = 0.006). The 2-year local control rate was 96% (95% CI 74%–99%). Among patients with radiotherapy-naïve lesions, no local recurrence was observed with a biologically effective dose ≥ 140 Gy, maximum dose of the planning target volume (PTV) ≥ 47 Gy, mean dose of the PTV ≥ 39 Gy, or minimum dose to 80% of the PTV ≥ 36 Gy (5-fraction equivalent doses). All acute toxicities from SBRT were grade 1–2, and no myelopathy was observed. CONCLUSIONS Neoadjuvant high-dose, hypofractionated SBRT for spinal chordoma is safe and does not increase surgical morbidities. Early outcomes at 2 years are promising, although long-term follow-up is pending.

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Long-term follow-up after colorectal endoscopic submucosal dissection in 182 cases

Endoscopy International Open ◽

10.1055/a-1321-1271 ◽

2021 ◽

Vol 09 (02) ◽

pp. E258-E262

Author(s):

Christian Suchy ◽

Moritz Berger ◽

Ingo Steinbrück ◽

Tsuneo Oyama ◽

Naohisa Yahagi ◽

...

Keyword(s):

Case Series ◽

En Bloc Resection ◽

Bloc Resection ◽

Published Data ◽

Low Grade ◽

Recurrence Rates ◽

En Bloc ◽

Long Term Follow Up

Abstract Background and study aims We previously reported a case series of our first 182 colorectal endoscopic submucosal dissections (ESDs). In the initial series, 155 ESDs had been technically feasible, with 137 en bloc resections and 97 en bloc resections with free margins (R0). Here, we present long-term follow-up data, with particular emphasis on cases where either en bloc resection was not achieved or en bloc resection resulted in positive margins (R1). Patients and methods Between September 2012 and October 2015, we performed 182 consecutive ESD procedures in 178 patients (median size 41.0 ± 17.4 mm; localization rectum vs. proximal rectum 63 vs. 119). Data on follow-up were obtained from our endoscopy database and from referring physicians. Results Of the initial cohort, 11 patients underwent surgery; follow-up data were available for 141 of the remaining 171 cases (82,5 %) with a median follow-up of 2.43 years (range 0.15–6.53). Recurrent adenoma was observed in 8 patients (n = 2 after margin positive en bloc ESD; n = 6 after fragmented resection). Recurrence rates were lower after en bloc resection, irrespective of involved margins (1.8 vs. 18,2 %; P < 0.01). All recurrences were low-grade adenomas and could be managed endoscopically. Conclusions The rate of recurrence is low after en bloc ESD, in particular if a one-piece resection can be achieved. Recurrence after fragmented resection is comparable to published data on piecemeal mucosal resection.

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