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JOURNAL ARTICLE
Bryan K Li, Alexandre Vasiljevic, Christelle Dufour, Fupan Yao, Ben L B Ho, Mei Lu, Eugene I Hwang, Sridharan Gururangan, Jordan R Hansford, Maryam Fouladi, Sumihito Nobusawa, Annie Laquerriere, Marie-Bernadette Delisle, Jason Fangusaro, Fabien Forest, Helen Toledano, Palma Solano-Paez, Sarah Leary, Diane Birks, Lindsey M Hoffman, Alexandru Szathmari, Cécile Faure-Conter, Xing Fan, Daniel Catchpoole, Li Zhou, Kris Ann P Schultz, Koichi Ichimura, Guillaume Gauchotte, Nada Jabado, Chris Jones, Delphine Loussouarn, Karima Mokhtari, Audrey Rousseau, David S Ziegler, Shinya Tanaka, Scott L Pomeroy, Amar Gajjar, Vijay Ramaswamy, Cynthia Hawkins, Richard G Grundy, D Ashley Hill, Eric Bouffet, Annie Huang, Anne Jouvet
Pineoblastomas (PBs) are rare, aggressive pediatric brain tumors of the pineal gland with modest overall survival despite intensive therapy. We sought to define the clinical and molecular spectra of PB to inform new treatment approaches for this orphan cancer. Tumor, blood, and clinical data from 91 patients with PB or supratentorial primitive neuroectodermal tumor (sPNETs/CNS-PNETs), and 2 pineal parenchymal tumors of intermediate differentiation (PPTIDs) were collected from 29 centres in the Rare Brain Tumor Consortium...
February 2020: Acta Neuropathologica
#2
JOURNAL ARTICLE
Eugene I Hwang, Marcel Kool, Peter C Burger, David Capper, Lukas Chavez, Sebastian Brabetz, Chris Williams-Hughes, Catherine Billups, Linda Heier, Alok Jaju, Jeff Michalski, Yimei Li, Sarah Leary, Tianni Zhou, Andreas von Deimling, David T W Jones, Maryam Fouladi, Ian F Pollack, Amar Gajjar, Roger J Packer, Stefan M Pfister, James M Olson
PURPOSE: Children with histologically diagnosed high-risk medulloblastoma, supratentorial primitive neuroectodermal tumor of the CNS (CNS-PNET), and pineoblastoma (PBL) have had poor survival despite intensive treatment. We included these patients in this Children's Oncology Group trial. Molecular profiling later revealed tumor heterogeneity that was not detectable at protocol inception. Enrollment of patients with CNS-PNET/PBL was subsequently discontinued, and outcomes for this part of the study are reported here...
October 17, 2018: Journal of Clinical Oncology
#3
JOURNAL ARTICLE
Alya Elshahoubi, Eman Khattab, Hadeel Halalsheh, Kawther Khaleifeh, Eric Bouffet, Nisreen Amayiri
BACKGROUND: Results of high-dose chemotherapy (HDCT) protocols for the management of malignant central nervous system (CNS) tumors in infants are mostly reported in high-income countries. We evaluated the feasibility and results of such protocols in a middle-income country (Jordan). METHODS: A retrospective study of infants' charts with CNS tumors between 2006 and 2015 who were treated according to HeadStart (HS) protocols. Data included patients' demographics, chemotherapy complications, and cost...
January 2019: Pediatric Blood & Cancer
#4
REVIEW
Patrick Sin-Chan, Bryan K Li, Ben Ho, Adriana Fonseca, Annie Huang
PURPOSE OF REVIEW: Malignant embryonal brain tumors (EBTs) of childhood span a wide clinical spectrum but can share remarkably similar morphologic features. This overlap presents significant diagnostic challenges, particularly for tumor entities that are rarely encountered in clinical practice and for which diagnostic criteria were poorly defined. This review will provide an update on the evolving characterization and treatment of rare EBTs. RECENT FINDINGS: Rapid advances in genomic tools have led to the discovery of robust molecular markers, and identification of novel tumor types and subtypes for almost all major categories of pediatric brain tumors...
July 11, 2018: Current Oncology Reports
#5
JOURNAL ARTICLE
Teresa de Rojas, Francisco Bautista, Miguel Flores, Lucía Igual, Raquel Rubio, Eduardo Bardón, Lucía Navarro, Laura Murillo, Raquel Hladun, Adela Cañete, Miguel Garcia-Ariza, Carmen Garrido, Ana Fernández-Teijeiro, Eduardo Quiroga, Carlota Calvo, Anna Llort, Inmaculada de Prada, Luis Madero, Ofelia Cruz, Lucas Moreno
Non-medulloblastoma CNS embryonal tumors (former PNET/Pineoblastomas) are aggressive malignancies with poor outcome that have been historically treated with medulloblastoma protocols. The purpose of this study is to present a tumor-specific, real-world data cohort of patients with CNS-PNET/PB to analyze quality indicators that can be implemented to improve the outcome of these patients. Patients 0-21 years with CNS-PNET treated in eight large institutions were included. Baseline characteristics, treatment and outcome [progression-free and overall survival (PFS and OS respectively)] were analyzed...
March 2018: Journal of Neuro-oncology
#6
JOURNAL ARTICLE
Fumiyuki Yamasaki, Takeshi Takayasu, Ryo Nosaka, Ikuno Nishibuchi, Hiroshi Kawaguchi, Manish Kolakshyapati, Shumpei Onishi, Taiichi Saito, Kazuhiko Sugiyama, Masao Kobayashi, Kaoru Kurisu
PURPOSE: The purpose of this study is to investigate the incidence of cystic malacia in long-term survivors of pediatric brain tumors treated with high-dose cranial irradiation. MATERIALS AND METHODS: Between 1997 and 2015, we treated 41 pediatric patients (26 males, 15 females; age ranging from 3.3 to 15.7 years, median 9-year-old) of pediatric brain tumors [17 medulloblastomas, 7 primitive neuroectodermal tumors (PNET), 3 pineoblastomas, 6 non-germinomatous germ cell tumors (NGGCT), 8 gliomas (including 4 ependymomas, 1 anaplastic astrocytoma, 1 oligodendroglioma, 1 pilocytic astrocytoma, 1 astroblastoma)] with high-dose craniospinal irradiation...
June 2017: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
#7
JOURNAL ARTICLE
Regina I Jakacki, Peter C Burger, Mehmet Kocak, James M Boyett, Joel Goldwein, Minesh Mehta, Roger J Packer, Nancy J Tarbell, Ian F Pollack
BACKGROUND: Supratentorial PNETs (sPNET) are uncommon embryonal malignancies of the central nervous system whose prognosis has historically been poor. We evaluated the outcome and prognostic factors of children with sPNET treated prospectively on a Children's Oncology Group trial. PROCEDURE: Following surgery, patients received craniospinal radiotherapy with concurrent carboplatin followed by six months of maintenance chemotherapy with cyclophosphamide and vincristine...
May 2015: Pediatric Blood & Cancer
#8
JOURNAL ARTICLE
W Reith, R Mühl-Benninghaus, A Simgen, U Yilmaz
Germ cell tumors, which constitute approximately 3-5% of tumors of the central nervous system (CNS), can be subdivided into germinomas, embryonal carcinomas, yolk sac tumors, choriocarcinomas, teratomas and mixed germ cell tumors. The diagnosis of intracranial germ cell tumor is based on the clinical symptoms, detection of tumor markers, such as alpha fetoprotein (AFP) and the beta subunit of human chorionic gonadotropin (beta-hCG) in blood and cerebrospinal fluid (CSF), magnetic resonance imaging (MRI) of the brain and spinal cord, CSF cytology and histology...
August 2014: Der Radiologe
#9
JOURNAL ARTICLE
Carsten Friedrich, Klaus Müller, Katja von Hoff, Robert Kwiecien, Torsten Pietsch, Monika Warmuth-Metz, Nicolas U Gerber, Peter Hau, Joachim Kuehl, Rolf D Kortmann, André O von Bueren, Stefan Rutkowski
Central nervous system primitive neuroectodermal tumors (CNS-PNET) and pineoblastomas (PBL) are rare in adulthood. Knowledge on clinical outcome and the efficacy and toxicities of chemotherapy in addition to radiotherapy is limited. Patients older than 21 years at diagnosis were followed in the observational arm of the prospective pediatric multicenter trial HIT 2000. After surgery, craniospinal irradiation and maintenance or sandwich chemotherapy were recommended. Radiotherapy was normo- (35.2 Gy; tumor region, 55...
February 2014: Journal of Neuro-oncology
#10
MULTICENTER STUDY
Sébastien Perreault, Robert M Lober, Anne-Sophie Carret, Guohua Zhang, Linda Hershon, Jean-Claude Décarie, Kristen Yeom, Hannes Vogel, Paul G Fisher, Sonia Partap
Embryonal tumors of the central nervous system (CNS) share histological features and were therefore initially grouped as primitive neuroectodermal tumors (PNET) and treated similarly. We sought to determine the relapse patterns of specific embryonal CNS tumors. We conducted a historical cohort study of children diagnosed with CNS embryonal tumors from January 2000 to December 2011 in two pediatric neuro-oncology centers. Patients of 21 years of age or younger at time of presentation with a diagnosis of medulloblastoma, supratentorial PNET, pineoblastoma or atypical teratoid/rhabdoid tumor (ATRT) and at least one surveillance MRI were included...
November 2013: Journal of Neuro-oncology
#11
MULTICENTER STUDY
Carsten Friedrich, André O von Bueren, Katja von Hoff, Nicolas U Gerber, Holger Ottensmeier, Frank Deinlein, Martin Benesch, Robert Kwiecien, Torsten Pietsch, Monika Warmuth-Metz, Andreas Faldum, Joachim Kuehl, Rolf D Kortmann, Stefan Rutkowski
BACKGROUND: Especially in young children, primitive neuroectodermal tumors of the central nervous system (CNS-PNET) and pineoblastomas are associated with an unfavorable outcome, and only a few prospective trials have been conducted thus far. METHODS: From January 2001 through January 2005, 17 eligible children aged <4 years with CNS-PNET not otherwise specified (n = 8), ependymoblastoma (n = 1), or pineoblastoma (n = 8) confirmed by central review were prospectively treated in the trial HIT 2000...
February 2013: Neuro-oncology
#12
JOURNAL ARTICLE
André O von Bueren, Joachim Gerss, Christian Hagel, Haoyang Cai, Marc Remke, Martin Hasselblatt, Burt G Feuerstein, Sarah Pernet, Olivier Delattre, Andrey Korshunov, Stefan Rutkowski, Stefan M Pfister, Michael Baudis
Little is known about frequency, association with clinical characteristics, and prognostic impact of DNA copy number alterations (CNA) on survival in central primitive neuroectodermal tumors (CNS-PNET) and tumors of the pineal region. Searches of MEDLINE, Pubmed, and EMBASE--after the original description of comparative genomic hybridization in 1992 and July 2010--identified 15 case series of patients with CNS-PNET and tumors of the pineal region whose tumors were investigated for genome-wide CNA. One additional case study was identified from contact with experts...
September 2012: Journal of Neuro-oncology
#13
JOURNAL ARTICLE
Suzanne Miller, Jennifer H Ward, Hazel A Rogers, James Lowe, Richard G Grundy
Pediatric embryonal brain tumors can be difficult to classify. Atypical teratoid rhabdoid tumors (ATRT) contain rhabdoid cells, while primitive neuroectodermal tumors (PNETs) are composed of "small round blue cells." Loss of INI1 is a common event in ATRT; therefore, we investigated if the loss of INI1 protein expression was also observed in central nervous system (CNS) PNET and pineoblastoma. A histological review of 42 CNS PNETs and six pineoblastomas was performed. INI1 expression was assessed by immunohistochemistry...
January 2013: Brain Pathology
#14
JOURNAL ARTICLE
Suzanne Miller, Hazel A Rogers, Paul Lyon, Vikki Rand, Martyna Adamowicz-Brice, Steven C Clifford, James T Hayden, Sara Dyer, Stefan Pfister, Andrey Korshunov, Marie-Anne Brundler, James Lowe, Beth Coyle, Richard G Grundy
Central nervous system primitive neuroectodermal tumor (CNS PNET) and pineoblastoma are highly malignant embryonal brain tumors with poor prognoses. Current therapies are based on the treatment of pediatric medulloblastoma, even though these tumors are distinct at both the anatomical and molecular level. CNS PNET and pineoblastoma have a worse clinical outcome than medulloblastoma; thus, improved therapies based on an understanding of the underlying biology of CNS PNET and pineoblastoma are needed. To this end, we characterized the genomic alterations of 36 pediatric CNS PNETs and 8 pineoblastomas using Affymetrix single nucleotide polymorphism arrays...
August 2011: Neuro-oncology
#15
JOURNAL ARTICLE
Po Zhao, Alexis Strohl, Corina Gonzalez, Thomas Fishbein, Sandra Rosen-Bronson, Bhaskar Kallakury, Metin Ozdemirli
Transplant-transmitted malignances are rare but devastating events. Primary brain tumors are the least common among reported donor-derived malignancies. We report a case of donor-transmitted pineoblastoma, a PNET, in a two-yr-old male recipient, who presented with a rapidly growing mass in the right mandible, four months after multiple visceral organ transplantation. The recipient had liver, pancreas, and small bowel transplants because of end-stage liver failure and short gut syndrome, which was secondary to large bowel resection for management of gastroschisis complicated by intestinal volvulus...
June 2012: Pediatric Transplantation
#16
JOURNAL ARTICLE
Amy Rosenfeld, Morris Kletzel, Reggie Duerst, David Jacobsohn, Paul Haut, Joanna Weinstein, Alfred Rademaker, Colleen Schaefer, Lauren Evans, Molly Fouts, Stewart Goldman
High risk/recurrent CNS tumors have a poor prognosis. We studied tandem high dose chemotherapy (HDC) with hematopoietic progenitor stem cell rescues (HPCR) as potentially curative therapy. Twenty-four patients (mean age 6.8 years) were enrolled, 19 underwent HDC/HPCR. Diagnoses were medulloblastoma (n = 9), germ cell tumor (n = 4), high grade astrocytoma (n = 2), supratentorial PNET (n = 1), pineoblastoma (n = 2), or papillary meningioma (n = 1). Cytoreduction regimen #1 consisted of carboplatin (500 mg/m(2)) x 3 days, etoposide (250 mg/m(2)) x 3 days, and thiotepa (300 mg/m(2)) x 3 days...
April 2010: Journal of Neuro-oncology
#17
JOURNAL ARTICLE
Stewart J Kellie, Christopher K F Wong, Luciano Dalla Pozza, Keith D Waters, Liane Lockwood, David C Mauger, Les White
BACKGROUND: Chemotherapy is used as an alternative to irradiation or to minimize the irradiation exposure among infants with medulloblastoma or other CNS embryonal tumors. Adjuvant chemotherapy is commonly used in older children with high-risk medulloblastoma to improve survival or to allow a reduction in the craniospinal irradiation dose in standard-risk patients. However, optimal multimodality therapy, including the precise role of chemotherapy, has not been defined for these groups of patients...
September 2002: Medical and Pediatric Oncology
#18
JOURNAL ARTICLE
N Ishii, H Hiraga, Y Sawamura, Y Shinohe, K Nagashima
Primitive neuroectodermal tumors (PNET) occur either in the central nervous system (CNS; central PNET, cPNET) or in the peripheral sites (peripheral PNET, pPNET). Recent molecular approaches have been defining a new concept of PNET, that is, the pPNET including Ewing's sarcoma (ES) which expresses MIC2 glycoprotein and shows the specific chimeric gene of EWS-FLI1. The expression of MIC2 and the genetic rearrangement of EWS-FLI1 are considered to be highly specific to the pPNET/ES. This study examined the expression of MIC2 and EWS-FLI1 gene by means of immunohistochemistry and reverse transcriptase-polymerase chain reaction (RT-PCR) on various small round cell tumors originating in the CNS or non-CNS organs...
March 2001: Neuropathology: Official Journal of the Japanese Society of Neuropathology
#19
JOURNAL ARTICLE
U R Kees, D Spagnolo, L A Hallam, J Ford, P R Ranford, D L Baker, D F Callen, J A Biegel
Pineoblastoma is a rare, but highly malignant tumor of the central nervous system (CNS) in children and is classified as a central primitive neuroectodermal tumor (PNET). Despite notable recent advances in understanding the molecular genetic basis of malignancies, the pathogenesis of PNETs remains enigmatic. There is scant information on the cytogenetics of PNETs arising in the pineal gland and the only three reported cases did not show any common aberrations. Here we report the establishment and characterization of a new pineoblastoma cell line, PER-480...
January 15, 1998: Cancer Genetics and Cytogenetics
#20
JOURNAL ARTICLE
E Galanis, J C Buckner, P J Schomberg, J E Hammack, C Raffel, B W Scheithauer
PURPOSE: Embryonal tumors of the CNS include, among others, medulloblastoma, cerebral neuroblastoma, pineoblastoma, and primitive neuroectodermal tumors (PNETs). Almost all data on the treatment of embryonal CNS tumors are derived from the pediatric population, since these tumors are uncommon in adulthood. The purpose of this study was to examine the rate and duration of response to chemotherapy of advanced embryonal CNS tumors in adults. PATIENTS AND METHODS: We retrospectively studied all adult (> 18 years of age) patients with advanced embryonal tumors of the CNS who received chemotherapy at our institution between 1976 and 1994...
August 1997: Journal of Clinical Oncology
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