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      Spinal meningiomas in pediatric patients – A case series and literature review

      case-report

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          Abstract

          Background:

          Meningiomas are the most frequent intracranial tumors in the adult population; however, they are rare in pediatric patients. In children, meningiomas often require further diagnosis of genetic comorbidities. As many as, 50% of young patients with meningiomas suffer from neurofibromatosis type 2 (NF2). Spinal meningiomas include only 10% of pediatric meningiomas.

          Case Description:

          Between 2000 and 2017, three children were hospitalized in the Neurosurgery Department. The patients reported prolonged periods of increasing neurological symptoms. In each case, a total gross tumor resection was performed. Histopathology result in each patient was meningioma psammomatosum. Only one girl required adjuvant radiotherapy (RTH) due to recurrent tumors. Magnetic resonance imaging (MRI) showed spinal nerves schwannomas and bilateral vestibular schwannomas in two patients with NF2.

          Conclusion:

          A slow tumor growth is characteristic of spinal meningiomas. Back pain is a frequent initial symptom of a slowly growing tumor mass. Subsequently, neurological deficits gradually increase. Patients require a long follow-up period and control MRI-scan. Children with diagnosed spinal meningioma should be strictly controlled because of the high risk of their developing other tumors associated with NF2. Surgical resection is the primary treatment modality of meningiomas. Adjuvant RTH should be recommended only for selected patients.

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          Most cited references22

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          Medical therapies for meningiomas.

          Meningiomas are the most common primary brain tumor in adults. Although the majority of these tumors can be effectively treated with surgery and radiation therapy, an important subset of patients have inoperable tumors, or develop recurrent disease after surgery and radiotherapy, and require some form of medical therapy. There are increasing numbers of studies evaluating various medical therapies but the results remain disappointing. Chemotherapies and hormonal therapies have been generally ineffective, although somatostatin analogues may have therapeutic potential. There is also increasing interest in targeted molecular therapies. Agents inhibiting platelet derived growth factor receptors and epidermal growth factor receptors have shown little efficacy, but molecular agents inhibiting vascular endothelial growth factor receptors appear to have some promise. As with other tumors, advances in the medical therapies for meningiomas will require improved understanding of the molecular pathogenesis of these tumors, more predictive preclinical models, and efficient mechanisms for conducting clinical trials, given the small population of eligible patients.
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            Spinal meningiomas: critical review of 131 surgically treated patients.

            This study was undertaken to analyze the functional outcome of surgically treated spinal meningiomas and to determine factors for surgical morbidity. Between January 1990 and December 2006 a total of 131 patients underwent surgical resection of a spinal menigioma. There were 114 (87%) female and 17 (13%) male patients. Age ranged from 17 to 88 years (mean 69 years). The mean follow-up period was 61 months (range 1-116 months) including a complete neurological examination and postoperative MRI studies. The pre- and postoperative neurological state was graded according to the Frankel Scale. Surgery was performed under standard microsurgical conditions with neurophysiological monitoring. In 73% the lesion was located in the thoracic region, in 16% in the cervical region, in 5% at the cervico-thoracic junction, in 4.5% at the thoraco-lumbar junction and in 1.5% in the lumbar region. Surgical resection was complete in 127 patients (97%) and incomplete in 4 patients (3%). At the last follow-up the neurological state was improved or unchanged in 126 patients (96.2%) and worse in 4 patients (3%). Permanent operative morbidity and mortality rates were 3 and 0.8%, respectively. Extensive tumour calcification proved to be a significant factor for surgical morbidity (P < 0.0001). Radical resection of spinal meningiomas can be performed with good functional results. Extensive tumor calcification, especially in elderly patients proved to harbor an increased risk for surgical morbidity.
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              Meningiomas in children and adolescents: a meta-analysis of individual patient data.

              The epidemiological, prognostic, and therapeutic features of child and adolescent meningioma are poorly defined. Clinical knowledge has been drawn from small case series and extrapolation from adult studies. This study was done to pool and analyse the clinical evidence on child and adolescent meningioma. Searches of PubMed, Medline, and Embase identified 35 case series of child and adolescent meningioma completed over the past 21 years. Individual patient data were obtained from 30 studies via direct communication with investigators. Primary outcomes were relapse-free survival (RFS) and overall survival. Prognostic variables were extent of initial surgery, use of upfront radiotherapy, age, sex, presence of neurofibromatosis, tumour location, and tumour grade. RFS and overall survival were analysed using Kaplan-Meier survival curves and multivariable Cox regression models. From a total of 677 children and adolescents with meningioma, 518 were eligible for RFS analysis and 547 for overall survival analysis. Multivariable analysis showed that patients who underwent initial gross-total resection had better RFS (hazard ratio 0·16, 95% CI 0·10-0·25; p<0·0001) and overall survival (0·21, 0·11-0·39; p<0·0001) than those who had subtotal resection. No significant benefit was seen for upfront radiotherapy in terms of RFS (0·59, 0·30-1·16; p=0·128) or overall survival (1·10, 0·53-2·28; p=0·791). Patients with neurofibromatosis type 2 (NF2) had worse RFS than those without neurofibromatosis (2·36, 1·23-4·51; p=0·010). There was a significant change in overall survival with time between patients with NF2 compared with those without neurofibromatosis (1·45, 1·09-1·92; p=0·011); although overall survival was initially better for patients with NF2 than for those without neurofibromatosis, overall survival at 10 years was worse for patients with NF2. Patients with WHO grade III tumours had worse RFS than those with WHO grade I (3·90, 2·10-7·26; p<0·0001) and grade II tumours (2·49, 1·11-5·56; p=0·027). Extent of initial surgical resection is the strongest independent prognostic factor for child and adolescent meningioma. No benefit for upfront radiotherapy was noted. Hence, aggressive surgical management, to achieve gross-total resection, is the initial treatment of choice. In the event of a subtotal resection, repeat resection is recommended to achieve maximum extirpation. Close observation is warranted for patients who have a subtotal resection or who have WHO grade III tumours. Patients without neurofibromatosis should have a minimum 10-year follow-up, whereas patients with NF2 should be considered a special risk category, necessitating life-long follow-up. None. Copyright © 2011 Elsevier Ltd. All rights reserved.
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                Author and article information

                Contributors
                Journal
                Surg Neurol Int
                Surg Neurol Int
                Surgical Neurology International
                Scientific Scholar (USA )
                2229-5097
                2152-7806
                2022
                30 September 2022
                : 13
                : 445
                Affiliations
                [1 ]Department of Neurosurgery, St Lukas Hospital in Tarnów, Poland,
                [2 ]Department of Childrens’ Neurosurgery, Faculty of Medicine, Jagiellonian University Medical College, Kraków, Poland.
                Author notes
                [* ] Corresponding author: Paula Piątek, Department of Neurosurgery, St Lukas Hospital in Tarnów, Poland. paulapiatek89@ 123456gmail.com
                Article
                10.25259/SNI_365_2022
                10.25259/SNI_365_2022
                9610126
                36324961
                3346f034-522c-4902-a46f-289bbc744a96
                Copyright: © 2022 Surgical Neurology International

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

                History
                : 20 April 2022
                : 09 September 2022
                Categories
                Case Report

                Surgery
                neurofibromatosis type 2,pediatric meningiomas,spinal meningiomas
                Surgery
                neurofibromatosis type 2, pediatric meningiomas, spinal meningiomas

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