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      Peripheral primitive neuroectodermal tumors of the spine: a case report and review of the literature

      case-report

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          Abstract

          Background

          Peripheral primitive neuroectodermal tumors are extremely rare tumors in the spine; only 18 cases of extra-dural peripheral primitive neuroectodermal tumor cervical region have been reported. The aim of this report is to highlight the challenges in diagnosis and management of this condition.

          Case presentation

          We present a case of 5-year-old Moroccan boy, who presented with torticollis for 1 month. Computed tomography scan and Magnetic resonance imaging of the cervical spine revealed an extradural, dumbbell-shaped mass with extra-spinal extension at the left C1–C6 level. Multiple biopsy specimens were obtained. Histological examination revealed a highly cellular neoplasm composed of diffuse sheets of tumor cells having monomorphic, round to oval, finely vesicular nuclei. Immunohistochemical findings confirmed the diagnosis of intraspinal peripheral primitive neuroectodermal tumor.

          Conclusion

          After this illustrative case, we review the literature on clinicopathological and therapeutic aspects. In practice, it is important to consider the diagnosis of peripheral primitive neuroectodermal tumor in children and adolescents with an apparent soft-tissue mass located in the spine.

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          Primary spinal extradural Ewing's sarcoma (primitive neuroectodermal tumor): Report of a case and meta-analysis of the reported cases in the literature

          Saeed Saeedinia, Mohsen Nouri, Meysam Alimohammadi … (2012)
          Background: Primary spinal primitive neuroectodermal tumors (PNET) and/or spinal extraskeletal Ewing's sarcoma family tumors (ESET) are rare lesions appearing in the spinal extradural space. One hundred forty-one primary spinal PNETs, including 29 intramedullary lesions, have been reported in the literature. Encountering a case of primary epidural EES/peripheral PNET (pPNET) in sacral level, which is the fifth one occurring at this level in the literature, we have tried to conduct a meta-analysis of the reported cases. Case Description: A 44-year-old lady with epidural EES/pPNET is reported here. She was once operated for L5/S1 herniated disc, which did not ameliorate her symptoms. The clinical, imaging, surgical, and histopathologic characteristics of our case are presented and wide search of the literature is also done. All the reports were level 3 or less evidences and most of the series had missing parts. 106 cases of primary intraspinal (extradural/extramedullary-intradural) EES/pPNET and 29 cases of primary intramedullary PNET (CNS-PNET) have been reported in the literature. The most common clinical presentation in both entities was muscle weakness proportionate to the tumor location. Distant metastasis occurred in 38 of 99 (38%) cases of primary intraspinal EES/pPNET, while the rate of metastasis was 48% in patients with PNETs occurring in the intramedullary region (P > 0.05). One-year survival rate of the patients who underwent chemo-radiation after total or subtotal resection was better than those who did not receive chemotherapy or radiotherapy, or did not have total or subtotal resection. However, this difference was not repeated in 2-year survival rate in any of the tumor groups. Conclusion: It seems that total or subtotal removal of the tumor and adjuvant chemo- and radiation therapy can improve the outcome in these patients.
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            Extraskeletal Ewing's sarcoma: a case report and review of the literature.

            K O'Rourke, Brett Kennedy, B Hurson … (2000)
            Case report. To report on the diagnosis and current treatment of a rare tumor about the cervical spine. Extraskeletal Ewing's sarcoma (EES) is rare and has not been previously described about the cervical spine. We present a case of a 24-year-old man with a large mass in the posterior triangle of the neck extending through the vertebral foramens of the cervical vertebrae. This was identified as an extraskeletal Ewing's sarcoma. Traditional treatment paradigms have been associated with a poor prognosis. Since the recommendations of the Intergroup Rhabdomyosarcoma Study II study of multimodal chemotherapy and radiotherapy, this tumor has a significantly better prognosis. Surgical debulking of the tumor was necessary to relieve cord compression. Histologic analysis was used to confirm both magnetic resonance imaging and computed tomography diagnosis. A chemoradiation therapy program was commenced in accordance with Intergroup Rhabdomyosarcoma Study II recommendations. Computed tomography and magnetic resonance imaging demonstrated a large lobulated mass extending through the exit foramens of C2/C3 and C3/C4. The mass was entirely extraskeletal and extradural. Histologic examination of the excised mass showed microscopy consistent with extraskeletal Ewing's sarcoma. After surgical debulking and chemoradiation, the patient made a complete recovery. A review of the literature confirms that extraskeletal Ewing's sarcoma is a rare tumor and particularly so in the region of the cervical spine. Early diagnosis and surgical debulking combined with current multimodality chemoradiation programs can produce a favorable outcome.
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              Spinal epidural extraskeletal Ewing sarcoma: MR findings in two cases.

              C Choi, Mildred K. Cho, Eugene Rhim … (2001)
              We report the CT myelography and MR findings of two cases of extraskeletal Ewing sarcoma involving the spinal epidural and paravertebral spaces in a middle-aged man (case 1) and a young woman (case 2). In both cases CT myelography showed epidural and paravertebral masses on one side, with widening of the ipsilateral neural foramina at the C5-C6 level in case 1 and at the C7-T1 level in case 2. On MR images, the masses were isointense to muscle on T1-weighted images, hyperintense on T2-weighted images, and showed moderate enhancement on contrast-enhanced T1-weighted images. In one case, all pulse sequences showed linear signal voids, representing the vertebral artery encasement within the mass. The intradural component connected with the main mass was detected in the other case.
              Article 0 comments Cited 15 times – based on 0 reviews     Review now
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                Author and article information

                Contributors
                Mouna Khmou: +212 671 15 45 71 , mouna.khmou@yahoo.fr
                Abderrahmane Malihy: amalihy@gmail.com
                Najat Lamalmi: abiradil@hotmail.com
                Lamia Rouas: rouaslamiab@yahoo.fr
                Zaitouna Alhamany: alhamany.z@gmail.com
                Journal
                Journal ID (nlm-ta): BMC Res Notes
                Journal ID (iso-abbrev): BMC Res Notes
                Title: BMC Research Notes
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1756-0500
                Publication date (Electronic): 9 September 2016
                Publication date PMC-release: 9 September 2016
                Publication date Collection: 2016
                Volume: 9
                Issue: 1
                Electronic Location Identifier: 438
                Affiliations
                [1 ]Department of Pathology, Children’s Hospital, Ibn Sina University Hospital, Ibn Sina bd Ibn Rochd, Souissi, 10100 Rabat, Morocco
                [2 ]Faculty of Medicine and Pharmacy Rabat, University Mohammed V, Rabat, Morocco
                Article
                Publisher ID: 2246
                DOI: 10.1186/s13104-016-2246-5
                PMC ID: 5016941
                PubMed ID: 27613377
                SO-VID: 03713fdc-d479-465b-aa20-338ea58d46b8
                Copyright © © The Author(s) 2016
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 29 January 2016
                Date accepted : 6 September 2016
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2016

                ScienceOpen disciplines: Medicine
                Keywords: pnet,spine,extra-dural,tumors,childhood
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: pnet, spine, extra-dural, tumors, childhood

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