Surgical outcomes in spinal cord ependymomas and the importance of extent of resection in children and young adults

Safaee, Michael; Oh, Michael C.; Mummaneni, Praveen V.; Weinstein, Philip; Ames, Christopher P.; Chou, Dean; Berger, Mitchel S.; Parsa, Andrew T.; Gupta, Nalin

doi:10.3171/2013.12.peds13383

Cited by 42 publications

(33 citation statements)

References 55 publications

(54 reference statements)

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“…However, the role of RT after STR is controversial. We reviewed the literature on pediatric patients with grade II/III spinal ependymoma and found 13 cases that were subtotally resected (Table 1) [4,[6][7][8][9][10][11][12]. The median age was 9 (range 1 to 20).…”

Section: Dear Editormentioning

confidence: 99%

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Radiation therapy after subtotal resection of pediatric grade II/III spinal ependymomas: what is the evidence?

et al. 2015

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Dear Editor:We read with interest a recent paper published by Yang et al. The authors reported their institutional experience of 15 grade II spinal cord ependymomas in children [1]. Gross total resection (GTR) was achieved in 12 cases. The remaining three underwent subtotal resection (STR) followed by radiotherapy (RT). While no tumor recurrence occurred in the GTR group, residual tumor regrowth was observed in two STR cases. Consequently, the author concluded that postoperative radiotherapy should be considered carefully due to uncertain therapeutic efficacy.We agree with the authors that there is a paucity of published data on the use of adjuvant radiotherapy in children with grade II/III spinal ependymomas. Pediatric intraspinal ependymomas are considered more aggressive than their adult counterparts [2]. All efforts must be made to achieve GRT [3]. It is generally agreed that RT is not necessary if the tumor has been removed completed [4]. In adults, progression-free survival (PFS) has been shown to be significantly prolonged among those who received RT after STR in a meta-analysis [5]. However, the role of RT after STR is controversial. We reviewed the literature on pediatric patients with grade II/III spinal ependymoma and found 13 cases that were subtotally resected (Table 1) [4, 6-12]. The median age was 9 (range 1 to 20). There were 5 males and 8 females. All except three were located in the lower spinal cord (thoracic, lumbar, and conus). Ten received adjuvant RT with an average dose of 40 Gy. The rate of local recurrence in those who received RT was 40 % (4/10), while two of the three who did not receive RT had local recurrence. The median PFS was 68 months. For the three patients who had information on further treatment after local recurrence, two received STR followed by RT and postsalvage recurrence occurred in both cases. The other patient received RT only and experienced no recurrence. The median follow-up was 92 months. All patients were alive at the last followup except two. One received RT and the other one did not.Contrary to the opinion of Yang et al., our review of the literature suggests that there may be some evidence for beneficial effect of RT after STR in pediatric grade II/III intraspinal ependymomas. Because of the rarity of this tumor in the pediatric population, potentially different tumor biology between children and adults, and the risks of RT on the developing nervous and osseous tissues, we recommend a multi-institutional trial to investigate the role of RT after STR in the local control of these tumors and its effect on patient survival.

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Section: Dear Editormentioning

confidence: 99%

“…All efforts must be made to achieve GRT [3]. It is generally agreed that RT is not necessary if the tumor has been removed completed [4]. In adults, progression-free survival (PFS) has been shown to be significantly prolonged among those who received RT after STR in a meta-analysis [5].…”

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confidence: 99%

Radiation therapy after subtotal resection of pediatric grade II/III spinal ependymomas: what is the evidence?

et al. 2015

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“…Subtotal resection, lack of initial radiation treatment and a young age at diagnosis are factors that have been associated with worse prognosis . In some studies, pediatric patients have a less predictable outcome than their adult counterparts, even after gross total resection . Extraneural metastases from primary intradural MPEs are rare , as are advanced cases in which the recurrence involves the adjacent soft tissues , and cases that have both intradural and soft tissue involvement at initial presentation .…”

Section: Introductionmentioning

confidence: 99%

Clinicopathologic features of anaplastic myxopapillary ependymomas

et al. 2019

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Myxopapillary ependymomas (MPE) are considered benign (World Health Organization (WHO) grade I) neoplasms with favorable prognosis. However, malignant behavior occurs in a small subset. To our knowledge, only five anaplastic MPEs have been reported without consensus on diagnostic criteria. We retrieved 14 anaplastic MPEs from the pathology archives of six institutions. Each tumor included at least two of the following features: ≥5 mitoses per 10 high power fields, Ki-67 labeling index (LI) ≥10%, microvascular proliferation (MVP) and spontaneous necrosis. These features were typically encountered in the foci of hypercellularity and reduced mucin. There were eight male and six female patients (age range 6-57 years, median = 16.5). Ten tumors displayed anaplasia at initial resection, and 4 were anaplastic at a second surgery for recurrence (ranging from 9 months to 14 years following initial resection). The Ki-67 LI ranged between 8% and 40% in the anaplastic foci and <3% in the foci of classic MPE. There was documented cerebrospinal fluid (CSF) dissemination in seven cases, recurrence following an anaplastic diagnosis in three cases and bone or soft tissue invasion in two cases. One patient suffered lung metastases. Two cases evaluated by targeted next-generation sequencing and one evaluated by fluorescence in situ hybridization (FISH) showed nonspecific chromosomal gains. We conclude that although rare, anaplastic MPE occurs in both pediatric and adult patients, similar to other ependymomas. At a minimum, closer follow-up is recommended, given the concern for aggressive biologic potential. Further study is needed to determine WHO grading criteria and genetic indicators of tumor progression.

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“…The majority of primary intracranial ependymomas are from the IV ventricle and their disseminations were located in the cervical region. Knowledge about the origin of the tumor and the proper evolution before and after surgery could prevent the appearance of disseminations, which do not depend on the degree or histological type [23,24].…”

Section: Discussionmentioning

confidence: 99%

Intraspinalependymomas and Their Association with Metastasis/ Disseminations in Patients Over a Period of 22 Years

2018

IJP

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Introduction: Ependymomas are neuroepithelial tumors of variable morphological appearance whose treatment of choice is surgical. They represent 13% of intraspinal tumors and constitute 40% of spinal tumors in adults. In Cuba there are few studies about this pathology, hence the main objective is to evaluate the behavior of intraspinalependymomas in the Neurology and Neurosurgery Institute. Patients and Methods: A retrospective descriptive study of 47 patients operated on with a histological diagnosis of intraspinalependymomas in a period of 22 years was carried out. Results: In the patients studied, 51% started with a radicular syndrome. In ependymomas, the most frequent histology was myxopapillary (34%) and the terminal filum was the location that prevailed (48.9%). A total resection was achieved in 51.1%, postoperative complications were not frequent, being the cerebrospinal fluid fistula (19.1%) the most frequent. Half of the subjects evolved favorably. 40.4% of the intraspinalependymomas studied were from intracranial tumors, and of these 73.7% recurred. Primary spinal tumors did not disseminate in 92.9% of cases. Conclusions: Intraspinalependymomas may be primary of the spine or disseminations / metastases of other locations of the neuraxis. The disseminations do not depend on the degree or histological type. Spinal ependymomas have a high rate of tumor recurrence. The adequate postoperative functional recovery depends on an early diagnosis of intratrachealependymomas and the degree of surgical resection.

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Surgical outcomes in spinal cord ependymomas and the importance of extent of resection in children and young adults

Cited by 42 publications

References 55 publications

Radiation therapy after subtotal resection of pediatric grade II/III spinal ependymomas: what is the evidence?

Radiation therapy after subtotal resection of pediatric grade II/III spinal ependymomas: what is the evidence?

Clinicopathologic features of anaplastic myxopapillary ependymomas

Intraspinalependymomas and Their Association with Metastasis/ Disseminations in Patients Over a Period of 22 Years

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