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Diffuse intrinsic pontine

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https://www.readbyqxmd.com/read/28178670/new-in-vivo-avatars-of-diffuse-intrinsic-pontine-gliomas-dipg-from-stereotactic-biopsies-performed-at-diagnosis
#1
Alexandre Plessier, Ludivine Le Dret, Pascale Varlet, Kévin Beccaria, Joëlle Lacombe, Sébastien Mériaux, Françoise Geffroy, Laurence Fiette, Patricia Flamant, Fabrice Chrétien, Thomas Blauwblomme, Stéphanie Puget, Jacques Grill, Marie-Anne Debily, David Castel
Diffuse Instrinsic Pontine Glioma is the most aggressive form of High Grade Gliomas in children. The lack of biological material and the absence of relevant models have hampered the development of new therapeutics. Their extensive infiltration of the brainstem renders any surgical resection impossible and until recently biopsies were considered not informative enough and therefore not recommended. Thus, most models were derived from autopsy material. We aimed to develop relevant in vivo DIPG models that mimic this specific disease and its molecular diversity from tumor material obtained at diagnosis...
February 2, 2017: Oncotarget
https://www.readbyqxmd.com/read/28161497/survival-benefit-for-patients-with-diffuse-intrinsic-pontine-glioma-dipg-undergoing-re-irradiation-at-first-progression-a-matched-cohort-analysis-on-behalf-of-the-siop-e-hgg-dipg-working-group
#2
Geert O Janssens, Lorenza Gandola, Stephanie Bolle, Henry Mandeville, Monica Ramos-Albiac, Karen van Beek, Helen Benghiat, Bianca Hoeben, Andres Morales La Madrid, Rolf-Dieter Kortmann, Darren Hargrave, Johan Menten, Emilia Pecori, Veronica Biassoni, Andre O von Bueren, Dannis G van Vuurden, Maura Massimino, Dominik Sturm, Max Peters, Christof M Kramm
BACKGROUND: Overall survival (OS) of patients with diffuse intrinsic pontine glioma (DIPG) is poor. The purpose of this study is to analyse benefit and toxicity of re-irradiation at first progression. METHODS: At first progression, 31 children with DIPG, aged 2-16 years, underwent re-irradiation (dose 19.8-30.0 Gy) alone (n = 16) or combined with systemic therapy (n = 15). At initial presentation, all patients had typical symptoms and characteristic MRI features of DIPG, or biopsy-proven high-grade glioma...
February 2, 2017: European Journal of Cancer
https://www.readbyqxmd.com/read/28120771/diffuse-intrinsic-pontine-gliomas-in-children-interest-of-robotic-frameless-assisted-biopsy-a-technical-note
#3
H A Coca, H Cebula, M Benmekhbi, M P Chenard, N Entz-Werle, F Proust
INTRODUCTION: Diffuse intrinsic pontine gliomas (DIPG) constitute 10-15% of all brain tumors in the pediatric population; currently prognosis remains poor, with an overall survival of 7-14 months. Recently the indication of DIPG biopsy has been enlarged due to the development of molecular biology and various ongoing clinical and therapeutic trials. Classically a biopsy is performed using a stereotactic frame assisted procedure but the workflow may sometimes be heavy and more complex especially in children...
December 2016: Neuro-Chirurgie
https://www.readbyqxmd.com/read/28116951/neonatal-respiratory-failure-caused-by-congenital-diffuse-intrinsic-pontine-glioma
#4
Katie M Satrom, Rachel A Phelan, Christopher L Moertel, H Brent Clark, Dana E Johnson, Thomas N George
The authors present a case of diffuse intrinsic pontine glioma presenting in a newborn with stridor and respiratory distress that progressed to respiratory failure. Magnetic resonance imaging (MRI) of the brain revealed findings compatible with the diagnosis of diffuse intrinsic pontine glioma. The family pursued palliative care and postmortem examination confirmed WHO grade III astrocytoma.
January 1, 2017: Journal of Child Neurology
https://www.readbyqxmd.com/read/28110411/development-of-the-siope-dipg-network-registry-and-imaging-repository-a-collaborative-effort-to-optimize-research-into-a-rare-and-lethal-disease
#5
Sophie E M Veldhuijzen van Zanten, Joshua Baugh, Brooklyn Chaney, Dennis De Jongh, Esther Sanchez Aliaga, Frederik Barkhof, Johan Noltes, Ruben De Wolf, Jet Van Dijk, Antonio Cannarozzo, Carin M Damen-Korbijn, Jan A Lieverst, Niclas Colditz, Marion Hoffmann, Monika Warmuth-Metz, Brigitte Bison, David T W Jones, Dominik Sturm, Gerrit H Gielen, Chris Jones, Esther Hulleman, Raphael Calmon, David Castel, Pascale Varlet, Géraldine Giraud, Irene Slavc, Stefaan Van Gool, Sandra Jacobs, Filip Jadrijevic-Cvrlje, David Sumerauer, Karsten Nysom, Virve Pentikainen, Sanna-Maria Kivivuori, Pierre Leblond, Natasha Entz-Werle, Andre O von Bueren, Antonis Kattamis, Darren R Hargrave, Péter Hauser, Miklos Garami, Halldora K Thorarinsdottir, Jane Pears, Lorenza Gandola, Giedre Rutkauskiene, Geert O Janssens, Ingrid K Torsvik, Marta Perek-Polnik, Maria J Gil-da-Costa, Olga Zheludkova, Liudmila Shats, Ladislav Deak, Lidija Kitanovski, Ofelia Cruz, Andres Morales La Madrid, Stefan Holm, Nicolas Gerber, Rejin Kebudi, Richard Grundy, Enrique Lopez-Aguilar, Marta Zapata-Tarres, John Emmerik, Tim Hayden, Simon Bailey, Veronica Biassoni, Maura Massimino, Jacques Grill, William P Vandertop, Gertjan J L Kaspers, Maryam Fouladi, Christof M Kramm, Dannis G van Vuurden
Diffuse intrinsic pontine glioma (DIPG) is a rare and deadly childhood malignancy. After 40 years of mostly single-center, often non-randomized trials with variable patient inclusions, there has been no improvement in survival. It is therefore time for international collaboration in DIPG research, to provide new hope for children, parents and medical professionals fighting DIPG. In a first step towards collaboration, in 2011, a network of biologists and clinicians working in the field of DIPG was established within the European Society for Paediatric Oncology (SIOPE) Brain Tumour Group: the SIOPE DIPG Network...
January 21, 2017: Journal of Neuro-oncology
https://www.readbyqxmd.com/read/28108511/biomarker-based-pet-imaging-of-diffuse-intrinsic-pontine-glioma-in-mouse-models
#6
Susanne Kossatz, Brandon Carney, Melanie E Schweitzer, Giuseppe Carlucci, Vesselin Miloushev, Uday B Maachani, Prajwal Rajappa, Kayvan Keshari, David J Pisapia, Wolfgang A Weber, Mark M Souweidane, Thomas Reiner
Diffuse intrinsic pontine glioma (DIPG) is a childhood brainstem tumor with a universally poor prognosis. Here, we characterize on a positron emission tomography (PET) probe for imaging DIPG in vivo. In human histological tissues, the probes target, poly(ADP)ribose polymerase 1 (PARP1), was highly expressed in DIPG compared to normal brain. PET imaging allowed for the sensitive detection of DIPG in a genetically engineered mouse model (GEMM), and probe uptake correlated to histologically determined tumor infiltration...
January 20, 2017: Cancer Research
https://www.readbyqxmd.com/read/28107439/development-of-a-dipg-orthotopic-model-in-mice-using-an-implantable-guide-screw-system
#7
Miguel Marigil, Naiara Martinez-Velez, Pablo D Domínguez, Miguel Angel Idoate, Enric Xipell, Ana Patiño-García, Marisol Gonzalez-Huarriz, Marc García-Moure, Marie-Pierre Junier, Hervé Chneiweiss, Elías El-Habr, Ricardo Diez-Valle, Sonia Tejada-Solís, Marta M Alonso
OBJECTIVE: In this work we set to develop and to validate a new in vivo frameless orthotopic Diffuse Intrinsic Pontine Glioma (DIPG) model based in the implantation of a guide-screw system. METHODS: It consisted of a guide-screw also called bolt, a Hamilton syringe with a 26-gauge needle and an insulin-like 15-gauge needle. The guide screw is 2.6 mm in length and harbors a 0.5 mm central hole which accepts the needle of the Hamilton syringe avoiding a theoretical displacement during insertion...
2017: PloS One
https://www.readbyqxmd.com/read/28093680/the-international-diffuse-intrinsic-pontine-glioma-registry-an-infrastructure-to-accelerate-collaborative-research-for-an-orphan-disease
#8
Joshua Baugh, Ute Bartels, James Leach, Blaise Jones, Brooklyn Chaney, Katherine E Warren, Jenavieve Kirkendall, Renee Doughman, Cynthia Hawkins, Lili Miles, Christine Fuller, Tim Hassall, Eric Bouffet, Adam Lane, Darren Hargrave, Jacques Grill, Lindsey M Hoffman, Chris Jones, Alex Towbin, Sharon A Savage, Michelle Monje, Xiao-Nan Li, David S Ziegler, Sophie Veldhuijzen van Zanten, Christof M Kramm, Dannis G van Vuurden, Maryam Fouladi
Diffuse intrinsic pontine glioma (DIPG), a rare, often fatal childhood brain tumor, remains a major therapeutic challenge. In 2012, investigators, funded by the DIPG Collaborative (a philanthropic partnership among 29 private foundations), launched the International DIPG Registry (IDIPGR) to advance understanding of DIPG. Comprised of comprehensive deidentified but linked clinical, imaging, histopathological, and genomic repositories, the IDIPGR uses standardized case report forms for uniform data collection; serial imaging and histopathology are centrally reviewed by IDIPGR neuro-radiologists and neuro-pathologists, respectively...
January 16, 2017: Journal of Neuro-oncology
https://www.readbyqxmd.com/read/28052119/pre-clinical-study-of-panobinostat-in-xenograft-and-genetically-engineered-murine-diffuse-intrinsic-pontine-glioma-models
#9
Tammy Hennika, Guo Hu, Nagore G Olaciregui, Kelly L Barton, Anahid Ehteda, Arjanna Chitranjan, Cecilia Chang, Andrew J Gifford, Maria Tsoli, David S Ziegler, Angel M Carcaboso, Oren J Becher
BACKGROUND: Diffuse intrinsic pontine glioma (DIPG), or high-grade brainstem glioma (BSG), is one of the major causes of brain tumor-related deaths in children. Its prognosis has remained poor despite numerous efforts to improve survival. Panobinostat, a histone deacetylase inhibitor, is a targeted agent that has recently shown pre-clinical efficacy and entered a phase I clinical trial for the treatment of children with recurrent or progressive DIPG. METHODS: A collaborative pre-clinical study was conducted using both a genetic BSG mouse model driven by PDGF-B signaling, p53 loss, and ectopic H3...
2017: PloS One
https://www.readbyqxmd.com/read/28035748/a-phase-i-study-of-perifosine-with-temsirolimus-for-recurrent-pediatric-solid-tumors
#10
Oren J Becher, Stephen W Gilheeney, Yasmin Khakoo, David C Lyden, Sofia Haque, Kevin C De Braganca, Jill M Kolesar, Jason T Huse, Shakeel Modak, Leonard H Wexler, Kim Kramer, Ivan Spasojevic, Ira J Dunkel
BACKGROUND: The PI3K/AKT/mTOR pathway is aberrantly activated in many pediatric solid tumors including gliomas and medulloblastomas. Preclinical data in a pediatric glioma model demonstrated that the combination of perifosine (AKT inhibitor) and temsirolimus (mTOR inhibitor) is more potent at inhibiting the axis than either agent alone. We conducted this study to assess pharmacokinetics and identify the maximum tolerated dose for the combination. PROCEDURE: We performed a standard 3+3 phase I, open-label, dose-escalation study in patients with recurrent/refractory pediatric solid tumors...
December 30, 2016: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/27765855/molecular-drug-imaging-89zr-bevacizumab-pet-in-children-with-diffuse-intrinsic-pontine-glioma
#11
Marc Jansen, Sophie Em Veldhuijzen van Zanten, Dannis G Van Vuurden, Marc Huisman, Danielle J Vugts, Otto S Hoekstra, Guus A M S van Dongen, Gert-Jan Jl Kaspers
: Predictive tools to guide therapy in children with brain tumors are urgently needed. We introduced molecular imaging to facilitate this. We investigated whether bevacizumab can reach the tumor in children with diffuse intrinsic pontine glioma (DIPG) by measuring the tumor uptake of zirconium-89((89)Zr)-labeled bevacizumab by PET. In addition we evaluated the safety of the procedure in children and determined the optimal timing of imaging. METHODS: Patients received 0...
October 20, 2016: Journal of Nuclear Medicine: Official Publication, Society of Nuclear Medicine
https://www.readbyqxmd.com/read/27713169/a-phase-1-study-of-oral-ridaforolimus-in-pediatric-patients-with-advanced-solid-tumors
#12
Andrew D J Pearson, Sara M Federico, Isabelle Aerts, Darren R Hargrave, Steven G DuBois, Robert Iannone, Ryan D Geschwindt, Ruixue Wang, Frank G Haluska, Tanya M Trippett, Birgit Geoerger
PURPOSE: Ridaforolimus is an investigational, potent, selective mTOR inhibitor. This study was conducted to determine the recommended phase 2 dose (RP2D), maximum tolerated dose, safety, pharmacokinetics, and antitumor activity of oral ridaforolimus in children with advanced solid tumors. EXPERIMENTAL DESIGN: In this phase 1, multicenter, open-label study in children aged 6 to <18 years with advanced solid tumors, ridaforolimus was administered orally for 5 consecutive days/week in 28-day cycles until progression, unacceptable toxicity, or consent withdrawal...
October 4, 2016: Oncotarget
https://www.readbyqxmd.com/read/27698822/bioinformatics-analysis-of-the-molecular-mechanism-of-diffuse-intrinsic-pontine-glioma
#13
Lei Deng, Pengju Xiong, Yunhui Luo, Xiao Bu, Suokai Qian, Wuzhao Zhong
The present study aimed to elucidate key molecular mechanisms in the progression of diffuse intrinsic pontine glioma (DIPG). The gene expression profile GSE50021, which consisted of 35 pediatric DIPG samples and 10 normal brain samples, was downloaded from the Gene Expression Omnibus database. The differentially-expressed genes (DEGs) in the pediatric DIPG samples were identified. Gene Ontology (GO), Kyoto Encyclopedia of Genes and Genomes (KEGG) and Reactome pathways of DEGs were enriched and analyzed. The protein-protein interaction (PPI) network of the DEGs was constructed and functional modules of the PPI network were disclosed using ClusterONE...
October 2016: Oncology Letters
https://www.readbyqxmd.com/read/27538997/polo-like-kinase%C3%A2-1-as-a-potential-therapeutic-target-in-diffuse-intrinsic-pontine-glioma
#14
Vladimir Amani, Eric W Prince, Irina Alimova, Ilango Balakrishnan, Diane Birks, Andrew M Donson, Peter Harris, Jean M Mulcahy Levy, Michael Handler, Nicholas K Foreman, Sujatha Venkataraman, Rajeev Vibhakar
BACKGROUND: Diffuse intrinsic pontine gliomas (DIPGs) are highly aggressive, fatal, childhood tumors that arise in the brainstem. DIPGs have no effective treatment, and their location and diffuse nature render them inoperable. Radiation therapy remains the only standard of care for this devastating disease. New therapeutic targets are needed to develop novel therapy for DIPG. METHODS: We examined the expression of PLK1 mRNA in DIPG tumor samples through microarray analysis and found it to be up regulated versus normal pons...
2016: BMC Cancer
https://www.readbyqxmd.com/read/27438806/discrepant-longitudinal-volumetric-and-metabolic-evolution-of-diffuse-intrinsic-pontine-gliomas-during-treatment-implications-for-current-response-assessment-strategies
#15
U Löbel, S Hwang, A Edwards, Y Li, X Li, A Broniscer, Z Patay
INTRODUCTION: Based on clinical observations, we hypothesized that in infiltrative high-grade brainstem neoplasms, such as diffuse intrinsic pontine glioma (DIPG), longitudinal metabolic evaluation of the tumor by magnetic resonance spectroscopy (MRS) may be more accurate than volumetric data for monitoring the tumor's biological evolution during standard treatment. METHODS: We evaluated longitudinal MRS data and corresponding tumor volumes of 31 children with DIPG...
October 2016: Neuroradiology
https://www.readbyqxmd.com/read/27422681/diffuse-intrinsic-pontine-glioma-in-children-document-or-treat
#16
Beuriat Pierre-Aurélien, Szathmari Alexandru, Di Rocco Federico, Kanold Justyna, Mottolese Carmine, Frappaz Didier
BACKGROUND: The place of stereotactic biopsies in the management of diffuse intrinsic pontine gliomas (DIPGs) in children has changed over the years. Nonetheless, stereotactic biopsy remains a surgical procedure with its risks. One complication that has not been reported previously in case of a biopsy of a DIPG is metastatic seeding along the tract of the biopsy. We report the first 2 cases in the literature. CASE DESCRIPTION: One 8-year-old and one 9-year-old boy were admitted for a typical DIPG...
September 2016: World Neurosurgery
https://www.readbyqxmd.com/read/27392443/pediatric-thalamic-glioma-with-h3f3a-k27m-mutation-which-was-detected-before-and-after-malignant-transformation-a-case-report
#17
Kenichi Ishibashi, Takeshi Inoue, Hiroko Fukushima, Yusuke Watanabe, Yoshiyasu Iwai, Hiroaki Sakamoto, Kai Yamasaki, Jyunichi Hara, Tomoko Shofuda, Daiksuke Kanematsu, Ema Yoshioka, Yonehiro Kanemura
PURPOSE: Histone H3.3 (H3F3A) mutation in the codon for lysine 27 (K27M) has been found as driver mutations in pediatric glioblastoma and has been suggested to play critical roles in the pathogenesis of thalamic gliomas and diffuse intrinsic pontine gliomas. We report a case of thalamic glioma with H3F3A K27M mutation, which was detected in both the primary tumor diagnosed as diffuse astrocytoma obtained during the first surgery and also in the tumor diagnosed as anaplastic astrocytoma obtained at the second surgery...
December 2016: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
https://www.readbyqxmd.com/read/27391980/a-novel-magnetic-resonance-imaging-segmentation-technique-for-determining-diffuse-intrinsic-pontine-glioma-tumor-volume
#18
Ranjodh Singh, Zhiping Zhou, Jamie Tisnado, Sofia Haque, Kyung K Peck, Robert J Young, Apostolos John Tsiouris, Sunitha B Thakur, Mark M Souweidane
OBJECTIVE Accurately determining diffuse intrinsic pontine glioma (DIPG) tumor volume is clinically important. The aims of the current study were to 1) measure DIPG volumes using methods that require different degrees of subjective judgment; and 2) evaluate interobserver agreement of measurements made using these methods. METHODS Eight patients from a Phase I clinical trial testing convection-enhanced delivery (CED) of a therapeutic antibody were included in the study. Pre-CED, post-radiation therapy axial T2-weighted images were analyzed using 2 methods requiring high degrees of subjective judgment (picture archiving and communication system [PACS] polygon and Volume Viewer auto-contour methods) and 1 method requiring a low degree of subjective judgment (k-means clustering segmentation) to determine tumor volumes...
November 2016: Journal of Neurosurgery. Pediatrics
https://www.readbyqxmd.com/read/27382528/bromodomain-inhibitor-review-bromodomain-and-extra-terminal-family-protein-inhibitors-as-a-potential-new-therapy-in-central-nervous-system-tumors
#19
REVIEW
Elizabeth Wadhwa, Theodore Nicolaides
The bromodomain and extraterminal (BET) family proteins associate with transcriptional activation through interaction with acetylated chromatin, therefore playing a key role as epigenetic regulators. BET proteins serve to regulate the expression of importance oncogenes, including those involved in apoptosis as well as cell cycle progression. Due to this potential as an epigenetic target, small molecule inhibition of BET proteins have been investigated and demonstrate promising activity in both solid and hematologic malignancies, including brain tumors...
May 21, 2016: Curēus
https://www.readbyqxmd.com/read/27379495/pre-radiation-chemotherapy-improves-survival-in-pediatric-diffuse-intrinsic-pontine-gliomas
#20
Z Gokce-Samar, P A Beuriat, C Faure-Conter, C Carrie, S Chabaud, L Claude, F Di Rocco, C Mottolese, A Szathmari, C Chabert, D Frappaz
BACKGROUND: The median survival of patients with diffuse intrinsic pontine glioma (DIPG) remains less than 1 year. The BSG 98 pre-irradiation chemotherapy protocol showed a significant increase in overall survival. In contrast to current treatment strategies, patients did not have to undergo surgical stereotactic biopsy, which can sometimes lead to complications, to be included in this protocol. MATERIALS AND METHODS: We retrospectively reviewed all the cases of DIPG that were treated in our department from September 15, 2004 to September 15, 2014...
August 2016: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
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