Management of Pediatric Myxopapillary Ependymoma: The Role of Adjuvant Radiation

Agbahiwe, Harold; Wharam, Moody D.; Batra, Sachin; Cohen, Kenneth J.; Terezakis, Stephanie A.

doi:10.1016/j.ijrobp.2012.05.001

Cited by 56 publications

(47 citation statements)

References 19 publications

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“…Similarly, our findings are consistent with studies in which the authors have found a distinct role for adjuvant radiotherapy in pediatric patients. Agbahiwe and colleagues 1 showed in a study of 16 pediatric patients with MPE that adjuvant radiotherapy improves local tumor control in those treated with GTR and STR. Their suggestion of treating pediatric MPE patients with adjuvant radiotherapy is one that the authors of several smaller studies have also proposed based on more anecdotal evidence.…”

Section: Discussionmentioning

confidence: 99%

“…Pica and colleagues 27 and Schild and colleagues 33 found that adjuvant radiotherapy was associated with improved outcomes when given in high doses (> 50.4 Gy) but not when given in low doses. Agbahiwe and colleagues 1 showed that radiotherapy was associated with local tumor control in at least one subpopulation: pediatric patients. Myxopapillary ependymomas appear to be particularly aggressive in these younger individuals.…”

Section: ©Aans 2013mentioning

confidence: 99%

“…However, to account for patients included in multiple studies, 8 manuscripts were excluded. 1,6,16,23,[27][28][29]32 In total, 28 articles with 475 patients were included in this integrative analysis. [2][3][4][5][7][8][9][10][11][12][13][14][15][17][18][19][20][21][23][24][25][26]30,31,[34][35][36][37] …”

Section: Article Selectionmentioning

confidence: 99%

“…7 Moreover, adjuvant radiotherapy in pediatric MPE patients appears to be associated with improved local tumor control. 1 To test whether MPEs are more aggressive in children than in adults and whether adjuvant radiotherapy might prove efficacious in children, we compared 2 groups of patients: one with individuals from those studies in which no patient was older than 19 years of age (pediatric patients), and another with patients from all other studies. There were 8 exclusively pediatric studies including 43 patients.…”

Section: Pediatric Patients With Mpementioning

confidence: 99%

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Tumor control after surgery for spinal myxopapillary ependymomas: distinct outcomes in adults versus children

Feldman

Clark²,

Safaee³

et al. 2013

SPI

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Object Myxopapillary ependymomas (MPEs) are rare WHO Grade I tumors found in the conus medullaris, cauda equina, and filum terminale. Treatment generally consists of resection with or without adjuvant radiotherapy. Evidence-based guidelines for surgical management are lacking due to the rarity of this tumor. Methods An English-language PubMed search was performed using the key words “myxopapillary” and “ependymoma.” Reports describing fewer than 3 patients or those lacking data on the extent of resection or radiotherapy were excluded. A total of 28 articles describing 475 patients met the authors' inclusion criteria. Patients were grouped by extent of resection and whether or not they underwent adjuvant radiotherapy. Differences in recurrence rates were assessed by chi-square test. Results The overall recurrence rate was 15.5% in patients treated by gross-total resection (GTR) and 32.6% in patients treated by subtotal resection (STR), irrespective of whether they underwent adjuvant therapy (p < 0.001). Regardless of the extent of resection, adjuvant radiotherapy was not associated with a decrease in recurrence rates. The overall recurrence rate was 15.6% in patients who underwent GTR and radiotherapy compared with 15.9% in patients who underwent GTR alone (p = 0.58), and it was 29.3% in patients who underwent STR and radiotherapy compared with 35.1% in those who underwent STR alone (p = 0.53). The difference between recurrence rates for patients who underwent GTR alone versus STR and radiotherapy was statistically significant (p = 0.02). Subgroup analysis demonstrated significantly higher recurrence rates in pediatric patients compared with adults (40.5% vs 23.4%, respectively; p = 0.02). Even in the setting of GTR alone, recurrence rates were higher in pediatric patients (65% vs 7.6%; p < 0.001). Conclusions Gross-total resection alone is associated with decreased recurrence rates compared with STR with or without radiotherapy. The authors' results suggest that treatment goals should include attempted GTR whenever possible. The observation that children benefitted from radiation therapy to a greater extent than did adults suggests that biological differences between tumors in these patient populations warrants more rigorous scientific studies.

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Section: Discussionmentioning

confidence: 99%

Section: ©Aans 2013mentioning

confidence: 99%

Section: Article Selectionmentioning

confidence: 99%

Section: Pediatric Patients With Mpementioning

confidence: 99%

See 2 more Smart Citations

Tumor control after surgery for spinal myxopapillary ependymomas: distinct outcomes in adults versus children

Feldman

Clark²,

Safaee³

et al. 2013

SPI

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“…1,3,4,7,10,12 In a pediatric population of 15 children with MPE from the Johns Hopkins Hospital, the local control rate at 5 and 10 3 Resection and adjuvant radiotherapy were performed in 4 patients; 3 of these patients remained disease-free compared with 2 of 3 patients with GTR alone who experienced disease recurrence. Only 6 pediatric cases were available for analysis in the present series.…”

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confidence: 99%

Outcomes following myxopapillary ependymoma resection: the importance of capsule integrity

Abdulaziz¹,

Mallory²,

Bydon

et al. 2015

FOC

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OBJECT While extent of resection has been shown to correlate with outcomes after myxopapillary ependymoma (MPE) resection, the effect of capsular violation has not been well studied. The role of adjuvant radiation also remains controversial. In this paper the authors' goals were to evaluate outcomes following resection of MPE based on intraoperative capsular violation and to explore the role of adjuvant radiotherapy in cases of capsular violation. METHODS A retrospective review of patients undergoing resection of MPE at 2 academic institutions between 1990 and 2013 was performed. Cases with dissemination at presentation, less than 12 months of follow-up, or incomplete records were excluded. Extent of resection was defined as en bloc if all visible tumor was removed without capsular violation, gross-total resection (GTR) if all visible tumor was removed, but with capsular violation, and subtotal resection (STR) if a known residual was left at the time of surgery. Postoperative MR images were reviewed to confirm the extent of resection. Primary outcomes were progression-free survival (PFS) and overall recurrence rates. The effects of extent of resection, capsular violation, and adjuvant radiotherapy on recurrence rates and PFS were analyzed using Kaplan-Meier statistics. Associations between recurrence and preoperative variables were evaluated using Fisher exact methods and t-tests where appropriate. RESULTS Of the 107 patients reviewed, 58 patients (53% were male) met inclusion criteria. The mean age at surgery was 40.8 years (range 7–68 years). The median follow-up was 51.5 months (range 12–243 months). Extent of resection was defined as en bloc in 46.5% (n = 27), GTR in 34.5% (n = 20), and STR in 18.9% (n = 11). No recurrences were noted in the en bloc group, compared with 15% (n = 3) and 45% (n = 5) in the GTR and STR groups. En bloc resection was achieved most frequently in tumors involving the conus. Twelve patients (20%) underwent adjuvant radiotherapy following either STR or GTR. The overall recurrence rate was 13.8% (n = 8), and the 5-year PFS was 81%. Capsular violation was associated with a higher recurrence rate (p = 0.005). Adjuvant radiotherapy showed a nonsignificant trend of lower recurrence rates (16.7% vs 31.6%, p = 0.43) and longer PFS at 5 years (83.3% vs 49.9%, p = 0.16) in cases of capsular violation. CONCLUSIONS A strong correlation between capsular violation and recurrence was found following removal of MPE and should be assessed when defining extent of resection in future studies. Although the use of adjuvant radiotherapy in cases of capsular violation showed a trend toward improved PFS, further investigation is needed to establish its role as salvage therapy also appears to be effective at halting disease progression.

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Outcomes following limited‐volume proton therapy for multifocal spinal myxopapillary ependymoma

Looi

Indelicato

Vega

et al. 2020

Pediatric Blood & Cancer

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Purpose Spinal myxopapillary ependymoma (MPE) often presents with a multifocal distribution, complicating attempts at resection. There remains no standard approach to irradiating these patients. We report disease control and toxicity in pediatric patients with multifocal spinal MPE treated with limited‐volume proton therapy. Materials/methods Twelve patients (≤21 years old) with multifocal spinal MPE were treated between 2009 and 2018 with limited‐volume brain‐sparing proton therapy. Median age was 13.5 years (range, 7‐21). Radiotherapy was given as adjuvant therapy after primary surgery in five patients (42%) and for recurrence in seven (58%). No patient received prior radiation. Eleven patients (92%) had evidence of gross disease at radiotherapy. Eleven patients received 54 GyRBE; one received 50.4 GyRBE. Treatment toxicity was graded per the CTCAEv4.0. We estimated disease control and survival using the Kaplan‐Meier product‐limit method. Results The median follow‐up was 3.6 years (range, 1.8‐10.6). The five‐year actuarial rates of local control, progression‐free survival, and overall survival were 100%, 92%, and 100%, respectively. One patient experienced an out‐of‐field recurrence in the spine superior to the irradiated region. No patients developed in‐field recurrences. Following surgery and irradiation, one patient developed grade three spinal kyphosis and one patient developed grade 2 unilateral L5 neuropathy. Conclusion 54 GyRBE to a limited volume appears effective for disseminated spinal MPE in both the primary and salvage settings, sparing children the toxicity of full craniospinal irradiation. Compared with historical reports, this approach using proton therapy improves the therapeutic ratio, resulting in minimal side effects and high rates of disease control.

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Management of Pediatric Myxopapillary Ependymoma: The Role of Adjuvant Radiation

Cited by 56 publications

References 19 publications

Tumor control after surgery for spinal myxopapillary ependymomas: distinct outcomes in adults versus children

Tumor control after surgery for spinal myxopapillary ependymomas: distinct outcomes in adults versus children

Outcomes following myxopapillary ependymoma resection: the importance of capsule integrity

Outcomes following limited‐volume proton therapy for multifocal spinal myxopapillary ependymoma

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