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Balogun JA, Udayakumaran S, Collange NZ. Surgical treatment of pediatric low-grade glioma in developing countries. Childs Nerv Syst 2024; 40:3129-3134. [PMID: 38709257 DOI: 10.1007/s00381-024-06448-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/16/2024] [Accepted: 05/01/2024] [Indexed: 05/07/2024]
Abstract
Pediatric low-grade gliomas constitute the most common brain tumors worldwide, though with some peculiarities in the presentation and surgical care in different parts of the world. The symptomatology in developing countries is likely to be over longer periods with a tendency to delayed diagnosis due to cultural, religious beliefs, manpower, and infrastructural deficits. Thus, the children present with large tumors with attendant morbidities and an increased risk of mortalities from surgery. Surgery is mainly by "general" neurosurgeons due to the paucity of trained pediatric neurosurgeons. The pre-operative imaging may be limited to anatomic MR imaging, and in some cases, CT scans, without expansive neuropsychological evaluation. The armamentarium available to the neurosurgeon may warrant large openings to access the tumor, and there may be limited possibility for intra-operative mapping of "eloquent" brain functions when this is deemed necessary. Complicating pre-operative acute hydrocephalus can result in two operations that further worsen the catastrophic spending associated with brain tumor surgeries in these climes. While these challenges appear daunting but certainly have not been enough to deter the "can do" spirit of neurosurgeons in developing countries, it is essential to strengthen the training of pediatric neurosurgeons in LMICs and provide a platform for the advocacy of better infrastructure for the surgical management of these tumors.
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Affiliation(s)
- James A Balogun
- Department of Neurological Surgery, University College Hospital, Ibadan, Nigeria.
- Division of Neurosurgery, Department of Surgery, College of Medicine, University of Ibadan, No 1 Queen Elizabeth Road, Mokola, Ibadan, Nigeria.
| | - Suhas Udayakumaran
- Division of Paediatric Neurosurgery, Department of Neurosurgery, Amrita Institute of Medical Sciences and Research Centre, Kochi, India
| | - Nelci Z Collange
- Centro de Neurocirurgia Pediátrica (CENEPE), Pediatric Neurosurgical Center, São Paulo, Brazil
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Gorodezki D, Zipfel J, Bevot A, Nägele T, Ebinger M, Schuhmann MU, Schittenhelm J. Prognostic utility and characteristics of MIB-1 labeling index as a proliferative activity marker in childhood low-grade glioma: a retrospective observational study. J Cancer Res Clin Oncol 2024; 150:178. [PMID: 38580878 PMCID: PMC10997709 DOI: 10.1007/s00432-024-05701-w] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/26/2023] [Accepted: 03/13/2024] [Indexed: 04/07/2024]
Abstract
PURPOSE The prognostic utility of MIB-1 labeling index (LI) in pediatric low-grade glioma (PLGG) has not yet conclusively been described. We assess the correlation of MIB-1 LI and tumor growth velocity (TGV), aiming to contribute to the understanding of clinical implications and the predictive value of MIB-1 LI as an indicator of proliferative activity and progression-free survival (PFS) in PLGG. METHODS MIB-1 LI of a cohort of 172 nonependymal PLGGs were comprehensively characterized. Correlation to TGV, assessed by sequential MRI-based three-dimensional volumetry, and PFS was analyzed. RESULTS Mean MIB-1 LI accounted for 2.7% (range: < 1-10) and showed a significant decrease to 1.5% at secondary surgery (p = .0013). A significant difference of MIB-1 LI in different histopathological types and a correlation to tumor volume at diagnosis could be shown. Linear regression analysis showed a correlation between MIB-1 LI and preoperative TGV (R2 = .55, p < .0001), while correlation to TGV remarkably decreased after incomplete resection (R2 = .08, p = .013). Log-rank test showed no association of MIB-1 LI and 5-year PFS after incomplete (MIB-1 LI > 1 vs ≤ 1%: 48 vs 46%, p = .73) and gross-total resection (MIB-1 LI > 1 vs ≤ 1%: 89 vs 95%, p = .75). CONCLUSION These data confirm a correlation of MIB-1 LI and radiologically detectable TGV in PLGG for the first time. Compared with preoperative TGV, a crucially decreasing correlation of MIB-1 LI and TGV after surgery may result in limited prognostic capability of MIB-1 LI in PLGG.
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Affiliation(s)
- David Gorodezki
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany.
| | - Julian Zipfel
- Department of Neurosurgery, Section of Pediatric Neurosurgery, University Hospital Tübingen, Tübingen, Germany
| | - Andrea Bevot
- Department of Neuropediatrics and Developmental Neurology, University Hospital Tübingen, Tübingen, Germany
| | - Thomas Nägele
- Department of Neuroradiology, University Hospital Tübingen, Tübingen, Germany
| | - Martin Ebinger
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany
| | - Martin U Schuhmann
- Department of Neurosurgery, Section of Pediatric Neurosurgery, University Hospital Tübingen, Tübingen, Germany
| | - Jens Schittenhelm
- Institute of Pathology, Department of Neuropathology, University Hospital Tübingen, Tübingen, Germany
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Antkowiak L, Zimny M, Starszak K, Sordyl R, Mandera M. Surgical Treatment of Pediatric Incidentally Found Brain Tumors: A Single-Center Experience. Brain Sci 2023; 13:brainsci13050746. [PMID: 37239218 DOI: 10.3390/brainsci13050746] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/27/2023] [Revised: 04/28/2023] [Accepted: 04/29/2023] [Indexed: 05/28/2023] Open
Abstract
There remains much debate about the correct management of incidentally found brain tumors in the pediatric population. This study aimed to evaluate the efficacy and safety of surgical treatment of incidentally found pediatric brain tumors. A retrospective analysis of pediatric patients who underwent surgical resection of incidentally found brain tumors between January 2010 and April 2016 was performed. A total of seven patients were included. The median age at the time of diagnosis was 9.7 years. The reasons for performing neuroimaging were as follows: impeded speech development (n = 2), shunt control (n = 1), paranasal sinuses control (n = 1), behavior changes (n = 1), head trauma (n = 1), and preterm birth (n = 1). Five patients underwent gross total tumor resection (71.4%), while subtotal resection was performed in two patients (28.6%). There was no surgery-related morbidity. Patients were followed up for a mean of 79 months. One patient with atypical neurocytoma experienced tumor recurrence 45 months following primary resection. All patients remained neurologically intact. The majority of pediatric incidentally found brain tumors were histologically benign. Surgery remains a safe therapeutic approach associated with favorable long-term outcomes. Considering the expected long lifetime of pediatric patients, as well as the psychological burden associated with having a brain tumor as a child, surgical resection can be considered an initial approach.
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Affiliation(s)
- Lukasz Antkowiak
- Department of Pediatric Neurosurgery, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
| | - Mikolaj Zimny
- Department of Neurosurgery, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
| | - Krzysztof Starszak
- Department of Pediatric Neurosurgery, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
- Department of Human Anatomy, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
| | - Ryszard Sordyl
- Department of Pediatric Neurosurgery, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
| | - Marek Mandera
- Department of Pediatric Neurosurgery, Medical University of Silesia in Katowice, 40-752 Katowice, Poland
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Gorodezki D, Zipfel J, Queudeville M, Sosa J, Holzer U, Kern J, Bevot A, Schittenhelm J, Nägele T, Ebinger M, Schuhmann MU. Resection extent and BRAF V600E mutation status determine postoperative tumor growth velocity in pediatric low-grade glioma: results from a single-center cohort analysis. J Neurooncol 2022; 160:567-576. [PMID: 36319795 PMCID: PMC9758245 DOI: 10.1007/s11060-022-04176-4] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/18/2022] [Accepted: 10/18/2022] [Indexed: 12/23/2022]
Abstract
PURPOSE Despite excellent long-term overall survival rates, pediatric low-grade gliomas (pLGG) show high variety of clinical behavior regarding progress or senescence post incomplete resection (IR). This study retrospectively analyzes tumor growth velocity (TGV) of pLGG before surgery and after IR to investigate the impact of surgical extent, tumor location and molecular BRAF status on postoperative residual tumor growth behavior. METHODS Of a total of 172 patients with pLGG receiving surgical treatment, 107 underwent IR (66%). Fifty-three vs 94 patients could be included in the pre- and post-operative cohort, respectively, and were observed over a mean follow-up time of 40.2 vs 60.1 months. Sequential three-dimensional MRI-based tumor volumetry of a total of 407 MRI scans was performed to calculate pre- and postoperative TGV. RESULTS Mean preoperative TGV of 0.264 cm3/month showed significant deceleration of tumor growth to 0.085 cm3/month, 0.024 cm3/month and -0.016 cm3/month after 1st, 2nd, and 3rd IR, respectively (p < 0.001). Results remained significant after excluding patients undergoing (neo)adjuvant treatment. Resection extent showed correlation with postoperative reduction of TGV (R = 0.97, p < 0.001). ROC analysis identified a residual cut-off tumor volume > 2.03 cm3 associated with a higher risk of progress post IR (sensitivity 78,6%, specificity 76.3%, AUC 0.88). Postoperative TGV of BRAF V600E-mutant LGG was significantly higher than of BRAF wild-type LGG (0.123 cm3/month vs. 0.016 cm3/month, p = 0.047). CONCLUSION This data suggests that extensive surgical resection may impact pediatric LGG growth kinetics post incomplete resection by inducing a significant deceleration of tumor growth. BRAF-V600E mutation may be a risk factor for higher postoperative TGV.
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Affiliation(s)
- David Gorodezki
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany.
| | - Julian Zipfel
- Section of Pediatric Neurosurgery, Department of Neurosurgery, University Hospital Tübingen, Tübingen, Germany
| | - Manon Queudeville
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany
- Clinic for Pediatric Hematology and Oncology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany
| | - Jordana Sosa
- Section of Pediatric Neurosurgery, Department of Neurosurgery, University Hospital Tübingen, Tübingen, Germany
| | - Ursula Holzer
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany
| | - Jan Kern
- Department of Neuropediatrics and Developmental Neurology, University Hospital Tübingen, Tübingen, Germany
| | - Andrea Bevot
- Department of Neuropediatrics and Developmental Neurology, University Hospital Tübingen, Tübingen, Germany
| | - Jens Schittenhelm
- Department of Neuropathology, Institute of Pathology, University Hospital Tübingen, Tübingen, Germany
| | - Thomas Nägele
- Department of Neuroradiology, University Hospital Tübingen, Tübingen, Germany
| | - Martin Ebinger
- Department of Hematology and Oncology, University Children's Hospital Tübingen, Tübingen, Germany
| | - Martin U Schuhmann
- Section of Pediatric Neurosurgery, Department of Neurosurgery, University Hospital Tübingen, Tübingen, Germany
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Kanno H, Kanetsuna Y, Shinonaga M. Anaplastic myxopapillary ependymoma: A case report and review of literature. World J Clin Oncol 2021; 12:1072-1082. [PMID: 34909401 PMCID: PMC8641005 DOI: 10.5306/wjco.v12.i11.1072] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/23/2021] [Revised: 05/17/2021] [Accepted: 10/11/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Myxopapillary ependymoma (MPE) is a pathological grade I tumor that arises in the filum terminale. MPE with anaplastic features is extremely rare, and only 5 cases have shown malignancy at the time of recurrence.
CASE SUMMARY The patient (a 46-year-old woman) had undergone a MPE operation 30 years ago. After subtotal resection of the tumor located in L4-S1, it had a solid component that extended to the adjacent subcutaneous region. Histologically, the tumor consisted of a typical MPE with anaplastic features. The anaplastic areas of the tumor showed hypercellularity, a rapid mitotic rate, vascular proliferation, and connective tissue proliferation. Pleomorphic cells and atypical mitotic figures were occasionally observed. The MIB-1 index in this area was 12.3%. The immunohistochemical study showed immunoreactivity for vimentin, glial fibrillary acidic protein and S100. The morphological pattern and immunohistochemical profile were consistent with anaplastic MPE. The patient tolerated surgery well without new neurological deficits. She underwent local irradiation for the residual tumor and rehabilitation.
CONCLUSION Although extremely rare, anaplastic MPE occurs in both pediatric and adult patients, similar to other ependymomas. At a minimum, close monitoring is recommended, given concerns about aggressive biological potential. In the future, further study is needed to determine the WHO classification criteria and genetic indicators of tumor progression. The possibility of malignant transformation of MPE should be taken into account, and patients with MPE should be treated with care and follow-up.
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Affiliation(s)
- Hiroshi Kanno
- Department of Neurosurgery, International University of Health and Welfare Atami Hospital, Atami 413-0012, Shizuoka, Japan
| | - Yukiko Kanetsuna
- Department of Pathology, International University of Health and Welfare Atami Hospital, Atami 413-0012, Shizuoka, Japan
| | - Masamichi Shinonaga
- Department of Neurosurgery, International University of Health and Welfare Atami Hospital, Atami 413-0012, Shizuoka, Japan
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