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MULTICENTER STUDY
Jihane Adelon, Christelle Dufour, Stéphanie Foulon, Julien Masliah Planchon, David Meyronnet, Franck Bourdeaut, Gilles Palenzuela, Fanny Fouyssac, Sandra Raimbault, Emilie De Carli, Sébastien Klein, Anne Pagnier, Anne-Isabelle Bertozzi, Angélique Rome, Audrey David, Sylvie Chabaud, Cécile Faure-Conter
PURPOSE: High-risk medulloblastomas (HR-MB) may not respond to induction chemotherapy, with either post-induction stable (SD) or progressive disease (PD). There is no consensus regarding their optimal management. METHODS: A retrospective, multicentre study investigated patients with non-responder HR-MB treated according to the PNET HR + 5 protocol (NCT00936156) between 01/01/2009 and 31/12/2018. After two courses of etoposide and carboplatin (induction), patients with SD or PD were analyzed...
July 2021: Journal of Neuro-oncology
#22
RANDOMIZED CONTROLLED TRIAL
Adam S Levy, Mark Krailo, Susan Chi, Doojduen Villaluna, Linda Springer, Chris Williams-Hughes, Maryam Fouladi, Amar Gajjar
BACKGROUND: Approximately 30% of children with medulloblastoma (MB) experience recurrence, which is usually incurable. This study compared the overall survival (OS) of patients receiving temozolomide (TMZ) and irinotecan with that of patients receiving TMZ, irinotecan, and bevacizumab for recurrent MB/central nervous system (CNS) primitive neuroectodermal tumor (PNET). METHODS: Patients with relapsed/refractory MB or CNS PNET were randomly assigned to receive TMZ (150 mg/m2 /day PO on days 1-5) and irinotecan (50 mg/m2 /day IV on days 1-5) with or without bevacizumab (10 mg/kg IV on days 1 and 15)...
August 2021: Pediatric Blood & Cancer
#23
JOURNAL ARTICLE
M R Muehler, V R Rendell, L L Bergmann, E R Winslow, S B Reeder
OBJECTIVES: Ferumoxytol is an ultra-small superparamagnetic iron oxide (USPIO) agent that is taken up by splenic tissue. This study describes our initial institutional experience of ferumoxytol-enhanced MRI (feMRI) for differentiating intrapancreatic splenules (IPS) from other pancreatic lesions. METHODS: In this retrospective study, patients with computed tomographic imaging that identified small enhancing lesions in the tail of the pancreas subsequently underwent feMRI for further characterization...
December 30, 2020: Abdominal Radiology
#24
JOURNAL ARTICLE
Christelle Dufour, Stephanie Foulon, Anne Geoffray, Julien Masliah-Planchon, Dominique Figarella-Branger, Léa Guerrini-Rousseau, Cecile Faure-Conter, Celine Icher, Anne-Isabelle Bertozzi, Pierre Leblond, Tasnime Akbaraly, Franck Bourdeaut, Nicolas André, Celine Chappé, Pascale Schneider, Emilie De Carli, Pascal Chastagner, Claire Berger, Julien Lejeune, Christine Soler, Natacha Entz-Werlé, Marie-Bernadette Delisle
BACKGROUND: High-risk medulloblastoma are defined by the presence of metastatic disease and/or incomplete resection and/or unfavorable histopathology and/or tumors with MYC amplification. We aimed to assess the 3-year progression-free survival (PFS) and define the molecular characteristics associated with PFS in patients aged 5 to 19 years with newly diagnosed high-risk medulloblastoma treated according to the phase 2 trial PNET HR+5. METHODS: All children received postoperative induction chemotherapy (etoposide and carboplatin), followed by 2 high-dose thiotepa courses (600 mg/m 2) with hematological stem cell support...
December 30, 2020: Neuro-oncology
#25
JOURNAL ARTICLE
Stefan Dietzsch, Felix Placzek, Klaus Pietschmann, André O von Bueren, Christiane Matuschek, Albrecht Glück, Matthias Guckenberger, Volker Budach, Jutta Welzel, Christoph Pöttgen, Heinz Schmidberger, Frank Heinzelmann, Frank Paulsen, Montserrat Pazos Escudero, Rudolf Schwarz, Dagmar Hornung, Carmen Martini, Anca Ligia Grosu, Georg Stueben, Karolina Jablonska, Juergen Dunst, Heidi Stranzl-Lawatsch, Karin Dieckmann, Beate Timmermann, Torsten Pietsch, Monika Warmuth-Metz, Brigitte Bison, Robert Kwiecien, Martin Benesch, Nicolas U Gerber, Michael A Grotzer, Stefan M Pfister, Steven C Clifford, Katja von Hoff, Sabine Klagges, Stefan Rutkowski, Rolf-Dieter Kortmann, Martin Mynarek
Purpose: We aimed to compare treatment results in and outside of a randomized trial and to confirm factors influencing outcome in a large retrospective cohort of nonmetastatic medulloblastoma treated in Austria, Switzerland and Germany. Methods and Materials: Patients with nonmetastatic medulloblastoma (n = 382) aged 4 to 21 years and primary neurosurgical resection between 2001 and 2011 were assessed. Between 2001 and 2006, 176 of these patients (46.1%) were included in the randomized HIT SIOP PNET 4 trial...
November 2020: Advances in Radiation Oncology
#26
JOURNAL ARTICLE
Gauri Deshpande, Sridhar Epari, Chhavi Gupta, Omshree Shetty, Mamta Gurav, Girish Chinnaswamy, Tejpal Gupta
PURPOSE: The purpose is to highlight the primary intracranial (meningeal-based) occurrence of Ewing sarcoma/primitive neuroectodermal tumor (ES/PNET). METHODS: This report is a collation of clinicopathological features of eight cases of molecularly and clinicoradiologically confirmed primary (non-metastatic) intracranial (non-osseous) meningeal ES/PNET. RESULTS: The age range was 1 to 33 years with a median age of 9 years. Male to female ratio was 0...
August 6, 2020: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
#27
JOURNAL ARTICLE
Bunchoo Pongtanakul, Nongnuch Sirachainan, Pacharapan Surapolchai, Pimlak Charoenkwan, Worawut Choeyprasert, Patcharee Komwilaisak, Su-On Chainansamit, Nittaya Witsanuyothin, Yujinda Lertrakul, Piya Rujkijyanont, Somjai Kanjanapongkul, Darintr Sosothikul, Kleebsabai Sanpakit, Thirachit Chotsampancharoen, Somporn Wangruangsathit, Nintita Sripaiboonkij Thokanit, Sommaphun Tabjareon, Suradej Hongeng
BACKGROUND: Few epidemiological studies of pediatric central nervous system (CNS) tumors have been performed using data from Southeast Asian national registries. Therefore, we aimed to examine data on CNS tumors from the first national childhood CNS tumor registry in Thailand. METHODS: Newly diagnosed children with benign and malignant primary CNS tumors from 20 nationwide hospitals were included. Two eras in the Thai registry were studied to compare national protocol effectiveness, including 2003-2005 (before establishment of a pediatric CNS tumor protocol) and 2011-2012 (post-establishment)...
July 24, 2020: Journal of Neuro-oncology
#28
JOURNAL ARTICLE
Francesco Verde, Roberta Galatola, Valeria Romeo, Teresa Perillo, Raffaele Liuzzi, Luigi Camera, Michele Klain, Roberta Modica, Antongiulio Faggiano, Vincenzo Napolitano, Annamaria Colao, Arturo Brunetti, Simone Maurea
BACKGROUND: Magnetic Resonance Imaging (MRI) is a useful imaging modality to assess the presence of Pancreatic Neuroendocrine Tumors (PNETs), allowing repeat monitoring examinations in MEN-1 patients. OBJECTIVES: We aimed to compare the diagnostic accuracy of conventional MRI sequences identifying which better depict the presence of PNETs in MEN-1 patients. METHOD: We performed a retrospective analysis of consecutive MEN-1 patients who underwent a conventional MRI protocol to monitor previously proven PNETs...
June 24, 2020: Neuroendocrinology
#29
REVIEW
Lazaro De Lima, Mehmet Beşir Sürme, Marco Gessi, Angela Mastronuzzi, Evelina Miele, Gianpiero Tamburrini, Luca Massimi
INTRODUCTION: High-grade neuroepithelial tumor with BCOR alteration (HGNET BCOR) has been recently classified as a new category of tumors among those previously known as PNET. They are molecularly characterized by the mutation of the BCOR gene, a corepressor of BCL6 a gene (which has an important role in immune responses). Only case reports and very small series have been published so far; therefore, their behavior and management are still under investigation. The goal of the present case-based review is to provide a summary about the state of the art on these tumors...
August 2020: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
#30
JOURNAL ARTICLE
Ashwin Prajapati, Ashish Gulia, Prateek Hegde, Ajay Puri
Background: Reconstruction modalities described after distal fibula resection varies from no reconstruction to size matched allograft or reconstruction with ipsilateral proximal fibula. Every procedure has their own limitation. We used Prolene mesh to reconstruct lateral restraint (Meshplasty) after distal fibulectomy. Question: Is a minimal reconstruction using prolene mesh (meshplasty) adequate to restore ankle stability and function post distal fibula resection? Method: This retrospective analysis was performed in patients who underwent distal fibulectomy at the tertiary cancer hospital in India, between 01/01/2006 and 31/12/2016...
May 2020: Journal of Clinical Orthopaedics and Trauma
#31
JOURNAL ARTICLE
John G Aversa, Francine Blumental De Abreu, Sho Yano, Liqiang Xi, Donald W Hadley, Irini Manoli, Mark Raffeld, Samira M Sadowski, Naris Nilubol
BACKGROUND: Li-Fraumeni syndrome is a cancer predisposition syndrome caused by germline TP53 tumor suppressor gene mutations, with no previous association with pancreatic neuroendocrine tumors (PNETs). Here we present the first case of PNET associated with Li-Fraumeni syndrome. CASE PRESENTATION: This is a 43-year-old female who underwent laparoscopic distal pancreatectomy at age 39 for a well-differentiated grade 2 cystic PNET. When the patient was 41 years old, her seven-year-old daughter was found to have an astrocytoma and a germline TP53 mutation...
March 30, 2020: BMC Cancer
#32
JOURNAL ARTICLE
M Feretis, T Wang, E Ghorani, A Balakrishnan, S J Harper, A Jah, E L Huguet, R K Praseedom, S-S Liau
BACKGROUND: Non-functional pancreatic neuroendocrine tumours (NF-PNETs) are rare and have highly variable outcomes. Current guidelines recommend surveillance for NF-PNETs <2 cm. Patients who ultimately have surgical resection are at risk of disease recurrence, and data to support postoperative surveillance protocols are lacking. The aims of this study were to i) identify post-operative predictors of recurrence and ii) risk stratify patients at risk of recurrence. METHODS: Consecutive patients who underwent surgery for NF-PNETs between 2002 and 2015 were identified retrospectively...
October 2019: Pancreatology: Official Journal of the International Association of Pancreatology (IAP) ... [et Al.]
#33
JOURNAL ARTICLE
Raamis Khwaja, Emmanuel Mantilla, Karen Fink, Edward Pan
This case report describes a patient with a rare occurrence of primary spinal intramedullary Ewing's sarcoma (ES) in the cervical and thoracic spine. The older age of disease occurrence, uncommon location in the cervical and thoracic spine, and EWSR1 gene fusion as the basis of diagnosis are unique features of this case. There is no clear protocol for treatment of primary extraskeletal ES of the spine, with controversy between evidence for pursuing surgery versus a combination of radiation and chemotherapy...
August 2019: Anticancer Research
#34
JOURNAL ARTICLE
Mehdi Kardoust Parizi, Arman Mousavi, Niloofar Sadeghyar
A primary primitive neuroectodermal tumor (PNET) of the urinary bladder is a very rare bladder tumor. There are few described cases in the literature to date. We presented a 70-year-old man with primary nonmetastatic bladder PNET. The diagnosis was confirmed according to immunohistochemistry evaluation. The patient underwent bladder-sparing protocol using radiotherapy and standard systemic multidrug chemotherapy (vincristine + doxorubicin + cyclophosphamide). At the end of three years of follow-up, we performed radical cystoprostatectomy due to newly diagnosed high-grade transitional cell carcinoma of the bladder at the site far from the primary bladder PNET...
September 12, 2018: Turkish Journal of Urology
#35
Shilpa Mehta, Larisa Rusyn, Howard Ginsburg, Cristina Hajdu, Brenda Kohn
Pancreatic neuroendocrine tumors (PNETs) occur in the context of tuberous sclerosis complex (TSC). To date, PNETs in association with TSC have been described almost exclusively in adults and in the context of TSC2. We present the evaluation of a PNET in a young child with TSC1. A 3-year, 6-month-old boy with TSC1 was found on surveillance to have a small pancreatic lesion measuring 0.4 cm on magnetic resonance imaging (MRI). The lesion showed interval enlargement to 1 cm on serial MRI studies during the ensuing 16 weeks...
June 1, 2019: Journal of the Endocrine Society
#36
JOURNAL ARTICLE
Abtin Doroudinia, Sara Ahmadi, Payam Mehrian, Mihan Pourabdollah
Primary Ewing sarcoma (ES) or primitive neuroectodermal tumour (PNET) is a rare tumour in adults and primary renal involvement is extremely rare. Patients with renal ES or PNET respond to and would benefit from conventional ES treatment according to ES study protocols. Here, we report a case of a young woman, presenting with right flank pain and haematuria. After ultrasound and CT evaluation, a right middle pole renal mass was detected. The patient underwent radical right nephrectomy, and a grade 4 ES with peritoneal involvement was documented...
January 28, 2019: BMJ Case Reports
#37
JOURNAL ARTICLE
Mohamed Taha, Nur M Mohammed, Stephen Crowther, Rustom P Manecksha, Arun Z Thomas
We report the first case of a synchronous ipsilateral primitive neuroectodermal tumour (PNET) and clear cell renal cell carcinoma of the kidney. A 37-year-old man presented to the emergency department with a 24-hour history of colicky abdominal pain and visible haematuria. He had no relevant surgical or medical history. Physical examination was unremarkable apart from mild left flank tenderness. Triphasic CT of the abdomen and pelvis showed two solid lesions in the left kidney. Further staging CT of the chest showed no evidence of local or distal metastasis...
December 9, 2018: BMJ Case Reports
#38
JOURNAL ARTICLE
Shonan Sho, Colin M Court, Paul Winograd, Paul A Toste, Joseph R Pisegna, Michael Lewis, Timothy R Donahue, Oscar J Hines, Howard A Reber, David W Dawson, James S Tomlinson
BACKGROUND: Patients with early-stage pancreatic neuroendocrine tumors (PNETs) may develop metastatic recurrences despite undergoing potentially curative pancreas resections. We sought to identify factors predictive of metastatic recurrences and develop a prognostication strategy to predict recurrence-free survival (RFS) in resected PNETs. METHODS: Patients with localized PNETs undergoing surgical resection between 1989 and 2015 were identified. Univariate and multivariate analysis were used to identify potential predictors of post-resection metastasis...
October 23, 2018: Journal of Gastrointestinal Surgery
#39
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
#40
JOURNAL ARTICLE
Amit Tirosh, Neige Journy, Les R Folio, Choonsik Lee, Christiane Leite, Jianhua Yao, William Kovacs, W Marston Linehan, Ashkan Malayeri, Electron Kebebew, Amy Berrington de González
Purpose To assess the potential ionizing radiation exposure from CT scans for both screening and surveillance of patients with von Hippel-Lindau (VHL) syndrome. Materials and Methods For this retrospective study, abdomen-pelvic (AP) and chest-abdomen-pelvic (CAP) CT scans were performed with either a three-phase (n = 1242) or a dual-energy virtual noncontrast protocol (VNC; n = 149) in 747 patients with VHL syndrome in the National Institutes of Health Clinical Center between 2009 and 2015 (mean age, 47.6 years ± 14...
January 2019: Radiology
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