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. 2012 Jun;14(Suppl 1):i116–i120. doi: 10.1093/neuonc/nos104

NEUROSURGERY

PMCID: PMC3483350
Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-01. TOTAL SPONDILECTOMY IN A C2 SPINE TUMOR USING A PEDIATRIC CUSTOMIZED VERTEBRAL BODY PROSTHESIS: TECHNICAL CASE REPORT

Javier Ibáñez 1, Marta Brell 1, Manuel Tomás 2, Pedro Roldán 1, Mercedes Guibelalde 3, Angela Tavera 3, Jose Antonio Salinas 3

Abstract

Indications for complete C2 spondilectomy are uncommon and mainly limited to the treatment of those tumors in which radical resection offers a clear prognostic advantage. Surgeons are reluctant to perform this operation because it means dealing with critical neural and vascular structures and a complex reconstruction at the C1-C2 level. We report the case of a 6 year-old girl who presented with a posterior cervical painful mass and movement limitation. Radiological evaluation showed a huge C2 tumor affecting and destroying the whole vertebra. In a first-stage a posterior midline approach allowed subtotal tumor resection, releasing of neural structures and both vertebral arteries and an occipitocervical fusion. Pathology was diagnostic for aneurysmal bone cyst. As this pseudotumoral lesion shows high recurrence rates in cases of incomplete removal, a second surgical procedure was performed 5 weeks later. An anterior transoral approach was done with radical removal of the infiltrated C2 vertebral body. To obtain a stable anterior support, a customized titanium mesh-plate was used. It offers redistribution of the axial load of both C1 lateral masses to the C3 vertebral body and a secure screw fixation. The implant was specifically made for our pediatric patient, based on an original adult design. This constitutes the first case in which such prosthesis has been adapted and implanted in a child. Our patient did very well; she completely recovered without any neurological deficit. A total tumor resection could be demonstrated in the postoperative studies. There has been no recurrence after 6 months of follow-up. Although complete C2 spondilectomy is technically demanding, it offers a clear advantage in terms of tumor control and patient recovery in selected cases. A customized C2 vertebral body prosthesis can be adapted for pediatric use, making surgery easier and safer, enhancing the biomechanical strength and stability of the construction.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-02. PHOTODYNAMIC NEUROENDOSCOPIC DETECTION AND BIOPSY FOR INTRAVENTRICULAR LESIONS IN CENTRAL NERVOUS SYSTEM (CNS) GERMINOMA WITH A 5-AMINOLEVULNIC ACID (5-ALA)

Tomonari Suzuki 1, Kohei Fukuoka 1, Tomoyuki Kohga 2, Takaaki Yanagisawa 1, Junichi Adachi 2, Kazuhiko Mishima 2, Takamitsu Fujimaki 3, Masao Matsutani 2, Shoichiro Ishihara 2, Ryo Nishikawa 2

Abstract

BACKGROUND: Photodynamic detection with 5-amonolevulnic acid(5-ALA) during surgery has been employed recently for detection of malignant brain tumour lesions as in other malignancies. The usefulness of neuroendscopic fluorescence guided detection and sampling for the intraventricular CNS germinoma has not yet been studied and confirmed. OBJECTS: We describe the procedure and discuss the usefulness of this method for detecting and biopsy of intraventricular lesions in CNS germinoma. PATIENTS AND METHODS: Patients suspected with germinoma underwent photodynamic endoscopical detection of the intraventricular lesions. Three hours before the induction of general anesthesia, 20mg/kg of 5-ALA was administered orally. During endosopical surgery the wave peak of porphyrin fluorescence (635-nm) was observed with spectrometer by long-pass fiber through working channel after excitation with violet-blue (405-nm) light. Navigation system was employed for ventricular puncture in the absence of ventriculomegaly. For multiple lesions, sampling was tried for all lesions. RESULTS: 11 cases with neuroradiologically and clinically suspected intracranial germinoma ranging in age rom 14 years to 34 years underwent this procedure. The lesions were detected in pineal region, supracellar region, septum pellucidum and/or other regions on ventricular wall. All the tissues samples taken were confirmed as germinoma histopathologically. Fluorescence peak was observed in 10 of 11 patients(91%). The peak was not observed in a case whose lesions showed spontaneous regression after CT and cerebral angiography (CAG), before surgery. Procedure related morbidity was diplopia observed in two cases with a small lesion in pineal lesion, possibly caused by injury to tegmentum. CONCLUSIONS: Photodynamic neuroendoscopic detection with 5-ALA can be useful method to identify the possible intraventricular lesions of CNS germinoma for diagnosis.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-03. NEUROFIBROMATOSIS: LONG-TERM CONTROL OF SPINAL/PERIPHERAL MANIFESTATIONS

Robert Keating 1, Tiffani DeFreitas 1, Faisal Al Abbas 2, John Myseros 1, Amanda Yaun 1, Suresh Magge 1

Abstract

INTRODUCTION: Few clinical challenges are greater than the innumerable phenotypic manifestations seen with NF1. The role of surveillance radiographic studies/timing and therapeutic approach continue to be contested; best exemplified by patients with intraspinal/peripheral disease. Despite an inconsistent natural history, review of our institutional experience with NF1 patients and their peripheral disease was undertaken to assess long-term behavior of their intrinsic process and to offer guidance on treatment. METHODS: Over seventeen years, 17 patients(10M:7 F) underwent surgery for peripheral manifestations. Patients presented with progressive intraspinal disease with radiographic progression or clinical deterioration. Patients had concomitant intracranial surgical disease(4), hydrocephalus(2), and plexiform lesions requiring surgery(3). RESULTS: Spinal disease was observed in 17 pts(10 M:7 F) whereas plexiform lesions(requiring additional neurosurgery) were seen in 3 patients(2 patients had both manifestations). Surgical age was 12.63 yr(1.08-20.85 yr), time from radiographic diagnosis to surgery 1.61 yr(.02-4.7 yr). Follow-up was 3.44 yrs(2wk-16 yr) and average number of surgeries 1.95/pt. A total of 37 operations were done(nine à single spinal surgery,3 à two,1 à 3,1 à 4, and 3 à 5). Nine patients had surgery resulting from surveillance studies and all 9 had clinically improved outcomes, whereas 5/7 patients presenting acutely(neurological deterioration) had good outcomes(remaining two stable). Those followed by close radiographic surveillance had no appreciable morbidity although two patients had persistent deficits postop(despite overall improvement). In the non-surveillance group, one patient à CSF leak and another required additional surgery(in setting of bleeding). Persistent deficits however were seen in 4/7. No malignant transformations were noted in any group. CONCLUSION: Patients with NF1 are at significant risk of deterioration from spinal disease despite radiographic surveillance. Earlier intervention offers the best chance to avoid long-term deficits. Risk of morbidity and less successful surgery was higher in the de novo group of NF1 spinal patients than the surveillance cohort.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-04. SURGICAL MANAGEMENT OF PINEAL REGION TUMOURS IN CHILDREN

Benedetta Pettorini 1, Rafeed Al-Mahfoudh 2, Jawad Yousaf 1, Barry Pizer 1, Michael Jenkinson 2, Conor Mallucci 1

Abstract

INTRODUCTION: Pineal region tumours are rare in childhood. The management is still controversial due to a lack of consensus guidelines and the wide variability of pathologies in this area. The aim of this study is to review the management of a cohort of these tumours in a single centre and compare to available guidelines such as germ cell guidelines SIOP and CCLG. METHODS: Patients with pineal, tectal plate, midbrain and posterior third ventricular tumours were identified from the neuro-oncology database. Data collection included: presentation, germ cell markers, tumour size and location, imaging, histology, treatment, and control/relapse rates. Intra-operative MRI has been used since October 2009. RESULTS: Thirty patients were treated between 1998 and 2010. Median age at diagnosis was 12.7 years (range 3.6 to 17). Twenty-one patients (70%) presented with hydrocephalus. Eighteen (86%) were successfully treated with endoscopic third ventriculostomy alone. Seventeen patients (57%) had endoscopic biopsies, 14 of which were at the same time as third ventriculostomy (71% diagnostic). Eight (27%) had stereotactic biopsies (100% diagnostic). No morbidity or mortality was associated with endoscopic or stereotactic procedures. Thirteen tumours underwent resective surgery via an appropriate midline approach, taking into account the morphology and anatomy of each tumour. Significant tumour debulking was achieved in all patients, with gross total resection in 70% (100% for non astrocytic intrinsic tumours). There was one intraventricular haemorrage (full recovery) and one mild hemiparesis. There was no other surgical mortality. CONCLUSION: A multi-disciplinary approach is required for this heterogeneous group of paediatric tumours. The best option for hydrocephalus management is endoscopic third ventriculostomy. In our experience, tumour resection is effective and safe.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-05. VASOSPASM AFTER RESECTION OF BRAIN TUMOURS: CASE REPORTS AN REVIEW OF THE LITERATURE

Benedetta Pettorini 1, Stefano Parlato 1, Jawad Yousaf 1, Barry Pizer 1, Ram Kumar 1, Shivaram Avula 1, Conor Mallucci 1

Abstract

INTRODUCTION: Diffuse cerebral vasospasm after brain tumor resection is rarely reported. Diffuse cerebral vasospasm represents a complication following aneurysmal subarachnoid hemorrhage or after severe traumatic head injury. In the last several years, there have been an increasing number of cases of symptomatic cerebral vasospasm following intracranial tumor resection. Different causes of vasospasm after brain tumor resection have been described, such as significant intraoperative bleeding, tumour location, operative positioning, tumor pathology, vascular manipulation, release of vasoactive factors, and post- operative blood in the basal cisterns. In this study, we report three cases of delayed symptomatic vasospasm following resection of brain tumours. A review of the current literature and pathophysiology is discussed. METHOD: Three paediatric patients were identified who suffered from delayed symptomatic vasospasm following resection of brain tumours (2 girls, 1 boy; median age 7.1 years). Hospital records were reviewed regarding history, presenting features, physical examination, treatment and radiological findings. RESULTS: Three patients who underwent resection of brain tumour (1 vestibular schwannoma, 1 posterior fossa pylocytic astrocytoma, 1 supratentorial astrocytoma) developed delayed neurological symptoms (median 6 days after surgery). MRI and MRA showed cerebral vasospasm. Treatment involves elevation of the blood pressure (induced hypertension), hemodilution to improve cerebral blood flow, and maintenance of high normal circulating blood volume (hypervolemia). All patients recovered back to their post-operative baseline and MRA showed complete resolution of the vasospasm. CONCLUSIONS: Neurologic deterioration in the post-operative period following tumor resection, unexplained by other causes, could suggest possible vasospasm. Early recognition and prompt treatment using triple-H can significantly improve patient outcome.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-06. VERTEBROPLASTY OF ODONTOID TROUGH AN ANTERIOR CERVICAL APROACH IN A PEDIATRIC PATIENT WITH A C2 OSTEOBLASTOMA: A CASE REPORT

Mauricio Munoz 1,2,3

Abstract

OBJECTIVE AND IMPORTANCE: Benign osteoblastoma (OB) has a rather aggressive growth that potentially endangers adjacent structures as well as the spinal canal. There is little literature about OB of the second cervical vertebrae (C2) and its management. This is the first report of vertebroplasy of odontoid in a child with OB of the axis. CLINICAL PRESENTATION: An 11 y/o girl with painful torticolis, sudden onset of hemiparesis and a palpable posterior cervical mass showed at our E.R. Significant myelopathic findings and irregular breathing pattern were found on admission. A bone reconstructed CT scan showed a 360° C2 tumor with markedly hypertrophy of dens and posterior elements. Ventral and posterior compression of the upper medulla was confirmed on MRI. INTERVENTION: Posterior dural decompression was performed a day after admission. The biopsy reported benign OB. On a second procedure, through an open anterior cervical approach and using real-time biplanar fluoroscopy, a 14 G needle was introduced at C5 level and advanced to the tip of the odontoid. Then, a vertebroplasty with polymethyl methacrylate and barium sulfate was carried out. A third procedure delivered cement to the posterior aspect of C2 as to "seal" the open stumps of the tumor containing vertebrae. At present, she has total motor recovery; no head rotation impairment and no tumor growth on an 18 month follow up. Posterior fixation wasn't needed. CONCLUSION: Until now, complete resection or curettage of osteoblastic lessions has been the standard. A tendency to embolize awkward placed spine bone tumors has been developed recently. Although anterior open cervical dens vertebroplasty have been performed on adult patients very few times, it is also a feassible procedure on a pediatric patient by which to achieve stability and deprive an osteoblastic tumor from irrigation. This is the first pediatric case description.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-07. CLINICAL USEFULNESS OF INTRAOPERATIVE INTRAVENOUS ADMINISTRATION OF FLUORESCEIN SODIUM IN PEDIATRIC PATIENTS WITH BRAIN TUMORS

Hirohito Yano 1, Naoyuki Ohe 1, Noriyuki Nakayama 1, Jun Shinoda 2, Toru Iwama 1

Abstract

OBJECTIVE: We have reported the usefulness of high-dose fluorescence-guided surgery in adult patients with glioblastoma. However, there have been no reports about the surgeries of pediatric tumors using fluorescein sodium (Fl-Na). The goal of this study is to ascertain whether the intravenous injection of Fl-Na is useful and safety in pediatric patients with brain tumors. METHODS: We retrospectively analyzed 7 surgeries in 6 cases of pediatric brain tumors that were resected by Fl-Na guided surgery between January 2005 and December 2011. Four males and 2 females were included. Their mean age at the surgery was 8 years-old 9 months. Fl-Na (9.1-17.6 mg/kg, mean 15.6mg/kg) was intravenously injected after dural opening. There were 2 cases of anaplastic ependymoma, a case of germinoma, glioblastoma, gangliocytoma, mixed neuronal-glial tumor, respectively. All cases were intraaxial tumors including the germinoma existing in the right thalamus. Five cases located supratentorially, excluding the case of gangliocytoma, which located in the cerebellum. All cases were well enhanced with gadolinium on magnetic resonance images. The tumors were resected under the Fl-Na guidance of intraoperative visualization of their gadolinium enhancement. RESULTS: Three cases that were injected Fl-Na with dosage of more than 17 mg/kg, were stained deeply yellow. In contrast, other three cases (less than 17 mg/kg) were faintly stained. Three cases of superficial tumors and a case of germinoma were totally resected, however, one case of the thalamic glioblastoma, and a diffusely invaded gangliocytoma were only partially resected. No cases postoperatively showed serious side effects including renal dysfunctions. CONCLUSIONS: The Fl-Na guided surgery is convenient under the ordinary surgical microscope. This method is available for gross total resection of the superficial tumors, and serves as a real time navigator for the deep-seated tumors. Accordingly, this is also useful and safety in pediatric patients.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-08. ADJUVANT CHEMOTHERAPY FOR BRAIN TUMORS DELIVERED VIA A NOVEL INTRA-CAVITY MOLDABLE POLYMER MATRIX

Cheryl Rahman 1, Stuart Smith 1, Paul Morgan 2, Keith Langmack 2, Donald Macarthur 1, Felicity Rose 1, Kevin Shakesheff 1, Richard Grundy 1, Ruman Rahman 1

Abstract

INTRODUCTION: Administering chemotherapeutic agents locally into the surgical resection cavity circumvents the blood brain barrier, potentially targeting micro-deposits of cancer cells in the brain parenchyma whilst minimizing toxic doses systemically. Here we evaluate the clinical utility, toxicity and sustained drug release capability of a novel self-assembling formulation of poly(lactic-co-glycolic acid) (PLGA)/poly(ethylene glycol) (PEG) microparticles. METHODS: PLGA/PEG microparticle-based matrices were pasted around an ex vivo brain pseudo-resection cavity and analyzed using MRI and CT. Matrices were subjected to a standard isocentric high dose radiotherapy regime and microparticle structure assessed. In vitro toxicity of the polymer was assessed using tumor/endothelial cells and drug release of trichostatin A-, etoposide- and methotrexate-loaded matrices was determined. To verify drug activity, tumor cells were seeded onto drug-loaded matrices and viability assessed. RESULTS: PLGA/PEG matrices can be molded around a pseudo-resection cavity wall with no polymer-related artifact on MRI and CT scans. The polymer withstands fractionated radiotherapy, with no disruption of microparticle structure. No toxicity was evident when tumor or endothelial cells were grown on control matrices in vitro. Trichostatin A, etoposide and methotrexate were released from the matrices over a 1-3 week period in vitro and followed near zero-order kinetics after an initial burst, with released agents retaining cytotoxic capabilities. CONCLUSIONS: The PLGA/PEG delivery system offers an innovative intra-cavity approach to administer chemotherapeutics for improved local control of brain tumors. Matrices show good apposition around a resection cavity wall and are distinguishable in clinical scans. Matrices are non-toxic in vitro suggesting good biocompatibility in vivo. Active trichostatin A, etoposide and methotrexate can be released gradually from matrices, with radiotherapy unlikely to interfere with release. The PLGA/PEG mouldable drug-delivery system will be widely applicable for childhood brain tumours for which local control is an essential part of the treatment strategy.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-09. THE IMPACT OF SECOND-LOOK SURGERY UPON OUTCOME FOR CHILDREN WITH MEDULLOBLASTOMA AND OTHER CENTRAL NERVOUS SYSTEM (CNS) PRIMITIVE NEURO-ECTODERMAL TUMORS RESULTS FROM THE “HEAD START” TRIALS

Mark Krieger 1, S J Si 1, N Flores 1, K Haley 1, J Malvar 1, R Sposto 1, J Fangusaro 1, G Dhall 1, T B Davidson 1, J Finlay 1

Abstract

INTRODUCTION: Residual tumor after surgical resection carries a worse prognosis in childhood PNET. It is not known if post-chemotherapy resection improves this prognosis. We evaluated the impact of “second look” surgical resections in cases of medulloblastoma and other CNS PNETs. METHODS: 183 patients (MB= 122 and oPNET= 61) were enrolled in Head Start. 80 children initiated chemotherapy with residual tumor (MB= 54 and oPNET= 26). 29 of these patients had mestastatic disease (MB = 16, oPNET = 13.) 42 patients had persistent residual tumor at the end of induction chemotherapy (MB= 24 and oPNET= 18); 9 of these patients underwent “second look” surgical resections (MB= 7 and oPNET= 2). RESULTS: Overall, the 2-year EFS were 54.8 +/- 12.2 % and 28.6 +/- 12.8 %. For all 42 patients with persistent disease after induction chemotherapy, the 2-year EFS were 43 +/- 9.1%; for those undergoing second look surgery (n = 9) the 2 year EFS was 68.57 +/- 18.63 % and for those not undergoing second look surgery (n = 33) was 38.20 +/- 9.77 % (p = 0.19 NS). For MB with residual disease, the 2 year EFS was 54.8 +/- 12.2%; for those undergoing second look surgery (n = 7) the 2 year EFS was 80.0 +/- 17.89 % and for those not undergoing second look surgery (n = 17) was 49.02 +/- 13.38 % (p = 0.3521 NS). For patients with residual oPNET, the 2 year EFS was 28.6 +/- 12.8%; for those undergoing second look surgery (n = 2) the 2 year EFS was 50.0 +/- 35.36 % and for those not undergoing second look surgery (n = 16) was 25.39 +/- 13.53 % (p = 0.3875 NS). There were no differences in outcome between patients with localized versus metastatic disease. CONCLUSIONS: Second look surgical resection was used in only 20% of eligible patients. There was a trend towards improved outcomes of these patients. Further study using a multi-national meta-analysis is warranted.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-10. CONVECTION-ENHANCED DELIVERY OF CARMUSTINE IS EFFECTIVE AND NON-TOXIC IN PRECLINICAL MODELS OF DIFFUSE INTRINSIC PONTINE GLIOMA

Viola Caretti 1, Tonny Lagerweij 1, Pepijn Schellen 1, Marc Jansen 2, Dannis G van Vuurden 2, Esther Hulleman 1, Sander Idema 3, W Peter Vandertop 3, David P Noske 4, Gertjan Kaspers 5, Thomas Wurdinger 4

Abstract

BACKGROUND: Diffuse intrinsic pontine glioma (DIPG) is the pediatric brain tumor with the worst prognosis. A reason for inefficiency of chemotheraputics, such as carmustine, may be the lack of optimal drug delivery caused by an intact blood-brain barrier (BBB) and/or severe systemic toxicity reached at high drug doses. These roadblocks could be overcome by convection-enhanced delivery (CED), which may enable drugs to reach the infiltrating DIPG cells over relevant anatomical volumes. METHODS: CED was set up for local delivery in the murine brainstem. Carmustine (3.3 mg/mL; 15mM) diluted in water (5% glucose pH < 4) was delivered via CED for 30 minutes at a rate of 0.5 µL/minute in immunocompetent mice. CED of carmustine was then tested in our established E98-Fluc-mCherry (FM) DIPG and our newly developed VUMC-DIPG-3 DIPG mouse models. Blood withdrawal was performed twice, on the day of CED and seven days later. RESULTS: Increasing carmustine doses proportionally reduced primary DIPG cells viability in-vitro (IC50 ≈ 80 µM). After carmustine had shown no acute or chronic toxicities in immunocompetent mice, CED with carmustine was performed into the brainstem of mice with infiltrative E98-FM-DIPG tumours. E98-DIPG tumour mass shrunk, as measured by bioluminescence, and increased survival was reported in this group compared to the control group that received CED of vehicle solution. Next, survival in mice bearing VUMC-DIPG-3 tumours receiving carmustine-CED was also significantly longer than the control group. No differences in liver and kidney enzymes blood levels were found between the two groups, indicating no acute toxicity in these organs. CONCLUSIONS: Local delivery of carmustine in the brainstem of mice is non-toxic at the dose used and highly effective against orthotopic human E98-FM DIPG and murine VUMC-DIPG-3 tumors. Based on our preclinical analysis, we propose to further study the effect of carmustine delivered via CED into the brainstem of DIPG patients.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-11. THE POTENTIAL OF THERAGNOSTIC 124I-8H9 CONVECTION-ENHANCED DELIVERY IN DIFFUSE INTRINSIC PONTINE GLIOMA

Neal Luther 1, Zhiping Zhou 1, Pat Zanzonico 2, Nai-Kong Cheung 2, Mark Souweidane 1

Abstract

INTRODUCTION: No human study has directly evaluated a concentration-dependent volume of distribution of convection-enhanced delivery (CED)-infused agents in the brain. This remains a limitation of this therapeutic modality, as reasons for inefficacy in prior glioma CED clinical trials remain unclear. Because 124I activity in tissue can be directly measured in vivo with high resolution via positron-emission tomography (PET), and due to the potential therapeutic utility of radioimmunotherapy, we postulated 124I conjugated to the anti-glioma monoclonal antibody 8H9 produces a theoretically "theragnostic" agent against diffuse intrinsic pontine glioma (DIPG). METHODS: Safety and dosimetry of 131I-8H9 and 124I-8H9 delivered by CED in rodents and primate were evaluated. Fifteen rats underwent CED of 0.1-1.0 mCi of 131I-8H9 to the pons for toxicity evaluation. Six additional rats underwent CED of 10 µCi of 124I-8H9 to the pons for dosimetry, with PET performed immediately following CED and daily for one week. One primate underwent CED of 1.0 mCi of 124I-8H9 to the pons with gadolinium-albumin co-infusion for safety and dosimetry analysis. PET was performed 2 and 40 hours following surgery, and blood and CSF were collected for radioactivity 10 days following CED. Ratios of volume of infusion to volume of distribution (Vd/Vi) were calculated. RESULTS: One rat (1.0 mCi 131I-8H9 infusion) suffered toxicity necessitating early sacrifice. PET analysis in rats yielded a pontine absorbed dose of 37 Gy/mCi (>95% activity cleared within 72 hours). In the primate, no toxicity was observed, and absorbed pontine dose was 3.8 Gy/mCi. Activity decreased ten-fold between 2 and 40 hours following CED. Mean Vd was 0.14 cc3 (Vd/Vi = 14) in the rat and 6.2 cc3 (Vd/Vi = 9.5) in primate. CONCLUSION: These results demonstrate the safety and feasibility of 124I dosimetry via PET, and function to lay the pre-clinical framework of a trial evaluating 124I-8H9 for DIPG.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-12. GROSS TOTAL RESECTION IS THE THERAPEUTIC GOAL FOR CHILDHOOD AND ADOLESCENT MENINGIOMA: AN INDIVIDUAL PATIENT META-ANALYSIS

Rishi Kotecha 1, Elaine Pascoe 2, Elisabeth Rushing 3, Lucy Rorke-Adams 4, Ted Zwerdling 5, Xing Gao 6, Xin Li 7, Stephanie Greene 8, Abbas Amirjamshidi 9, Seung-Ki Kim 10, Marco Lima 11, Po-Cheng Hung 12, Fayçal Lakhdar 13, Nirav Mehta 14, Yuguang Liu 15, B Indira Devi 16, B Jayanand Sudhir 17, Morten Lund-Johansen 18, Flemming Gjerris 19, Catherine Cole 1, Nicholas Gottardo 1

Abstract

Meningiomas are the most common primary central nervous system tumor in adults, accounting for approximately 30% of all brain tumors. In stark contrast, childhood and adolescent meningioma is rare. Consequently, there is a paucity of data and management has largely been based on extrapolation from adult data. To better define prognostic factors for childhood and adolescent meningioma we undertook an individual patient meta-analysis, reviewing all reported case series in the literature over the past 21 years. A total of 35 case series of child and adolescent meningioma were identified and individual patient data were obtained from 30 studies via direct communication with investigators. A total of 677 children and adolescents with meningioma were identified. Of these 518 were eligible for relapse free survival (RFS) and 547 for overall survival (OS) analysis. For the whole cohort the 15 year RFS and OS were 64.6% (57.6-70.7%) and 73.3% (65.1-79.8%) respectively. Initial gross total resection was an independent prognostic factor for RFS (hazard ratio 0·16, 95% CI 0·10-0·25; p < 0·0001) and OS (0·21, 0·11-0·39; p < 0·0001) compared with subtotal resection. However, upfront radiotherapy was not associated with improved RFS (0·59, 0·30-1·16; p = 0·128) or OS (1·10, 0·53-2·28; p = 0·791). Patients with WHO grade III tumors had an almost 2.5 and 4 fold increased risk of recurrence compared with WHO grade II (2·49, 1·11-5·56; p = 0·027) and WHO grade I tumors (3·90, 2·10-7·26; p < 0·0001) respectively. We conclude that initial gross total resection is the most powerful independent prognostic factor for children and adolescents with meningioma, with no survival advantage for upfront radiotherapy. Therefore, aggressive surgical management, to achieve gross total resection, is the initial treatment of choice.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-13. THALAMIC TUMORS IN CHILDREN: THE ROLE OF SURGERY

Christian Dorfer 1, Irene Slavc 2, Karin Dieckmann 3, Kerstin Gruber 1, Maresa Schmook 4, Thomas Czech 1

Abstract

OBJECTIVE: To present our single center experience in the management of childhood thalamic tumors. METHODS: We retrospectively reviewed our patients operated on a thalamic tumor between 1980 and 2011. RESULTS: 43 children were operated (median age at diagnosis 9.1 years; (range from 1yrs to 19yrs; male:female ratio of 1:1). In15 patients with unilateral pilocytic astrocytomas 18 tumor resections and 3 stereotactic biopsies were performed. The extent of initial resection was categorized as > 90% in 9 and < 90% in 4. In 2 patients stereotactic biopsy alone was performed. 7/11 had an improvement of their preoperative hemiparesis after surgery. 5/15 children were treated with chemotherapy, 3/15 were treated with irradiation [1/15 focal (55Gy); 1/15 J125 seeds; 1/15 gamma knife (20 Gy)]. 15/15 are alive after a mean follow-up time of 7.3 years. In 21 children with a unilateral non-pilocytic tumor (LGG n = 7; HGG n = 13;PNET n = 1), 19 tumor resections and 6 stereotactic biopsies were performed. In 15 children with initial tumor resection the extent of resection was categorized as > 90% in 6 and < 90% in 9. 8/14 had no improvement of their preoperative hemiparesis after surgery. 12/21 children were treated with adjuvant chemotherapy and 14/21 with radiotherapy [11/14 focal (mean 58.1 Gy); 2/14 craniospinal (36Gy); 1/14 J125 seeds]. 13/21 are alive after a mean follow-up time of 5.0 years. In 7 bithalamic tumors 6/7 had chemotherapy and 5/7 radiotherapy after histologic diagnosis (3/7 stereotactic biopsy; 2/7 partial resection; 2/7 open biopsy). 4/7 rapidly progressed and died after a mean survival time of 14.1 months. 2/7 are long term survivors (8.3 years and 11.4 years). CONCLUSION: In pilocytic thalamic astrocytomas growth total resection is recommended. In non-pilocytic tumors there is no clear-cut benefit from attempting maximal resection.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-14. PROPHYLACTIC ENDOSCOPIC THIRD VENTRICULOSTOMY IN CHILDREN WITH POSTERIOR FOSSA TUMORS AND HYDROCEPHALUS: IS IT WARRANTED?

Andrew Griffin 1, Jeffrey Greenfield 1, Mark Souweidane 1

Abstract

OBJECTIVE: Recently it was proposed that endoscopic third ventriculostomy (ETV) be instituted prior to tumor removal in children presenting with hydrocephalus from a posterior fossa tumor. This recommendation comes from a recent clinical study indicating a reduced postoperative need for ventriculo-peritoneal (VP) shunting in patients undergoing ETV compared with those that did not. While the rationale may be valid, the true benefit of pre-resectional ETV would be based upon the actual requirement for postoperative hydrocephalus treatment. The purpose of this retrospective review was to examine the need of post-operative CSF diversion in a contemporary series of children with hydrocephalus due to posterior fossa tumors. METHODS: 89 children underwent 104 surgeries for posterior fossa tumors at New York Presbyterian Hospital and Memorial Sloan Kettering Cancer Center between 1995 and 2012. 48 of these patients had hydrocephalus on admission and were selected for analysis. Their ages ranged from 3 weeks to 16 years. 16 patients had medulloblastoma, 16 had pilocytic astrocytoma, 4 had rhabdomyosarcoma, 3 had ependymoma, 3 had teratoma, 3 had PNET, 2 had neuroblastoma, and 1 had hemangioblastoma. Patients were evaluated as to their need for any permanent CSF diversionary procedure (VPS or ETV) within 6 months following tumor removal. RESULTS: Prior to or concomitant with tumor resection, 16 patients were treated with a CSF diversionary procedure. 2 of these 16 patients (12%) required a subsequent CSF diversion procedure post-operatively. Of the 32 children without pre-resection CSF diversion, 3 patients (9%) required a VPS or ETV for symptomatic hydrocephalus. CONCLUSIONS: This contemporary series demonstrates a low need for (9%) post-operative CSF diversion in children with hydrocephalus due to posterior fossa tumors. Our findings suggest that the routine use of prophylactic ETV is not warranted as a justification for reducing post-resection CSF diversion due to the low incidence of symptomatic hydrocephalus.

Neuro Oncol. 2012 Jun;14(Suppl 1):i116–i120.

NS-15. REVERSIBLE CEREBRAL ARTERY VASOSPASM AFTER RESECTION OF CHILDHOOD MEDULLOBLASTOMA

Rishi R Lulla 1, Vamshi Rao 1, Abilash Haridas 1, Maura Ryan 1, Joshua L Goldstein 1, Mark Wainwright 1, Tadanori Tomita 1

Abstract

INTRODUCTION: Cerebral artery vasospasm after brain tumor resection is rare. We report a case of symptomatic vasospasm after resection of a posterior fossa tumor in a pediatric patient. CASE REPORT: A 10 year old girl underwent an uncomplicated posterior fossa craniotomy for resection of a right cerebellopontine classic medulloblastoma. Seven days post-operatively, she developed evidence of hydrocephalus and a ventriculoperitoneal shunt was placed. Two days later, she was noted to have transient episodes expressive aphasia and tingling in her hands and arms. Electroencephalogram showed no evidence of seizure activity. Magnetic resonance angiogram (MRA) and computed tomography angiography (CTA) of the brain revealed multiple short segments of smooth stenoses in the right and left posterior cerebral arteries (PCAs) and middle cerebral arteries (MCAs). A transcranial doppler (TCD) examination was performed and revealed a significant increase in time averaged velocity the right MCA, measuring 185 cm/sec. Elevated resistive indices of the other cerebral arteries were also noted together with an increased Lindegaard ratio, supporting a diagnosis of vasospasm in the right MCA. The patient was treated with hemodynamic augmentation, permissive hypertension and calcium channel blockade. Over the following 7 days, she improved clinically and the therapies were slowly weaned. Serial TCD examinations demonstrated reduction in elevated cerebral artery velocities. Repeat CTA 10 days after initiation of therapy revealed near complete resolution of the vasospasm. The patient went on to receive craniospinal radiation therapy (RT) with surveillance TCD examinations every 2 weeks. She completed RT as prescribed without clinical or radiographic evidence of recurrent vasospasm. CONCLUSION: Cerebral artery vasospasm is a rare complication of childhood brain tumor resection. Early recognition and prompt treatment can significantly improve patient outcomes. In addition to conventional neuroimaging, TCD may be useful for following response to therapy.

Articles from Neuro-Oncology are provided here courtesy of Society for Neuro-Oncology and Oxford University Press

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