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JOURNAL ARTICLE
Sujin Gang, Hyunhee Kwon, In Hye Song, Jung-Man Namgoong
BACKGROUND: Biliary intraepithelial neoplasia (BilIN), a noninvasive precursor of cholangiocarcinoma, can manifest malignant transformation. Since cholangiocarcinoma (CCA) may progress due to chronic inflammation in the bile ducts and gallbladder, choledochal cysts are considered a precursor to CCA. However, BilIN has rarely been reported in children, to date. METHODS: We reviewed medical records of patients (< 18 years of age, n = 329) who underwent choledochal cyst excision at Asan Medical Center from 2008 to 2022...
April 20, 2024: World Journal of Surgical Oncology
#22
JOURNAL ARTICLE
Francisco Maldonado, Ana Filipa Geraldo, Angela Guarnizo, Nicolás Fernández-Ponce, Lorena Baroni, Carlos Rugilo
The embryonal central nervous system (CNS) tumor with PLAGL1 (pleomorphic adenoma gene-like) amplification is a novel type of pediatric neoplasm with a distinct methylation profile, described for the first time in 2022. It may be located anywhere in the neuroaxis and, as its name implies, it is driven by the amplification and overexpression of one of the PLAG family genes. Although the associated clinical, immunohistopathological, and molecular characteristics are well characterized in the seminal report of this entity, data on the radiological features is still lacking...
April 20, 2024: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
#23
JOURNAL ARTICLE
Larisa Gorenstein, Shai Shrot, Michal Ben-Ami, Eve Stern, Michal Yalon, Chen Hoffmann, Shani Caspi, Michal Lurye, Amos Toren, Gadi Abebe-Campino, Dalit Modan-Moses
BACKGROUND AND PURPOSE: Multiple studies demonstrated hypothalamic-pituitary dysfunction in survivors of pediatric brain tumors. However, few studies investigated the trajectories of pituitary height in these patients and their associations with pituitary function. We aimed to evaluate longitudinal changes of pituitary height in children and adolescents with brain tumors, and their association with endocrine deficiencies. MATERIALS AND METHODS: We conducted a retrospective analysis of 193 pediatric patients (54...
April 17, 2024: Radiotherapy and Oncology
#24
JOURNAL ARTICLE
Kevin J Bielamowicz, Mary Beth Littrell, Gregory W Albert, Lora S Parker, Sateesh Jayappa, Kenneth Aldape, Murat Gokden
PURPOSE: Central nervous system (CNS) embryonal tumors are a diverse group of malignant tumors typically affecting pediatric patients that recently have been better defined, and this paper describes evolution of a unique type of embryonal tumor at relapse. METHODS: Two pediatric patients with CNS embryonal tumors with EWSR1-PLAGL1 rearrangements treated at Arkansas Children's Hospital with histopathologic and molecular data are described. RESULTS: These two patients at diagnosis were classified as CNS embryonal tumors with EWSR1-PLAGL1 rearrangements based on histologic appearance and molecular data...
April 19, 2024: Journal of Neuro-oncology
#25
JOURNAL ARTICLE
Priya H Marathe, Valeria Santibanez, Paul A Meyers, Maria L Padilla, Danielle N Friedman
Pleuroparenchymal fibroelastosis (PPFE) is a rare interstitial pneumonia with distinct clinicopathologic features. It has been associated with exposure to hematopoietic stem cell transplantation (HSCT) and classical alkylating agents. Here, we highlight PPFE as a late complication of childhood cancer therapy by describing the cases of four survivors of childhood cancer with a diagnosis of treatment-related PPFE. All patients received high-dose alkylating agents. PPFE should be considered in the differential diagnosis of restrictive lung disease in patients with history of exposure to alkylating agents or HSCT...
April 18, 2024: Pediatric Blood & Cancer
#26
JOURNAL ARTICLE
Rodrigo L P Romao, Jennifer H Aldrink, Lindsay A Renfro, Elizabeth A Mullen, Andrew J Murphy, Jack Brzezinski, Marcus M Malek, Daniel J Benedetti, Nicholas G Cost, Ethan Smith, Jeffrey S Dome, Andrew M Davidoff, Amy Treece, Lauren N Parsons, Conrad V Fernandez, Brett Tornwall, Robert C Shamberger, Arnold Paulino, John A Kalapurakal, James I Geller, Peter F Ehrlich
INTRODUCTION: The purpose of this study is to examine the outcomes in children with anaplastic bilateral Wilms tumor (BWT) from study AREN0534 in order to define potential prognostic factors and areas to target in future clinical trials. METHODS: Demographic and clinical data from AREN0534 study patients with anaplasia (focal anaplasia [FA], or diffuse anaplasia [DA]) were compared. Event-free survival (EFS) and overall survival (OS) were reported using Kaplan-Meier estimation with 95% confidence bands, and differences in outcomes between FA and DA compared using log-rank tests...
April 18, 2024: Pediatric Blood & Cancer
#27
JOURNAL ARTICLE
Alexandra E Kovach, Maximilian Wengyn, My H Vu, Andrew Doan, Gordana Raca, Deepa Bhojwani
BACKGROUND: Compared to other ethnicities, Hispanics/Latinos (H/L) have a high incidence of acute lymphoblastic leukemia (ALL), enrichment of unfavorable ALL genetic subtypes, and worse outcomes, even after correcting for socioeconomic factors. We previously demonstrated increased incidence of the high-risk genetic drivers IKZF1 deletion and IGH::CRLF2 rearrangement in H/L compared to non-H/L children with B-ALL. Here in an expanded pediatric cohort, we sought to identify novel genetic drivers and secondary genetic alterations in B-ALL associated with H/L ethnicity...
April 18, 2024: Pediatric Blood & Cancer
#28
JOURNAL ARTICLE
Lucia Pedace, Simone Pizzi, Luana Abballe, Maria Vinci, Celeste Antonacci, Sara Patrizi, Claudia Nardini, Francesca Del Bufalo, Sabrina Rossi, Giulia Pericoli, Francesca Gianno, Zein Mersini Besharat, Luca Tiberi, Angela Mastronuzzi, Elisabetta Ferretti, Marco Tartaglia, Franco Locatelli, Andrea Ciolfi, Evelina Miele
In vitro models of pediatric brain tumors (pBT) are instrumental for better understanding the mechanisms contributing to oncogenesis and testing new therapies; thus, ideally, they should recapitulate the original tumor. We applied DNA methylation (DNAm) and copy number variation (CNV) profiling to characterize 241 pBT samples, including 155 tumors and 86 pBT-derived cell cultures, considering serum vs serum-free conditions, late vs early passages, and dimensionality (2D vs 3D cultures). We performed a t-SNE classification and identified differentially methylated regions in tumors compared to cell models...
April 18, 2024: NPJ Precision Oncology
#29
JOURNAL ARTICLE
Rami W Eldaya, Richard J Fagan, Samir A Dagher, Angshumoy Roy, Sonika Dahyia, Gregory N Fuller, Max Wintermark, Matthew S Parsons, Thierry A G M Huisman
Primary intracranial sarcoma, DICER1 -mutant, is a rare, recently described entity in the fifth edition of the WHO Classification of CNS Tumors. Given the entity's rarity and recent description, imaging data on primary intracranial sarcoma, DICER1 -mutant, remains scarce. In this multicenter case series, we present detailed multimodality imaging features of primary intracranial sarcoma, DICER1 -mutant, with emphasis on the appearance of the entity on MR imaging. In total, 8 patients were included. In all 8 patients, the lesion demonstrated blood products on T1WI...
April 18, 2024: AJNR. American Journal of Neuroradiology
#30
JOURNAL ARTICLE
Joaquin Garcia-Solorio, Juan Carlos Núñez-Enriquez, Marco Jiménez-Olivares, Janet Flores-Lujano, Fernanda Flores-Espino, Carolina Molina-Garay, Alejandra Cervera, Diana Casique-Aguirre, José Gabriel Peñaloza-Gonzalez, Ma Del Rocío Baños-Lara, Ángel García-Soto, César Alejandro Galván-Díaz, Alberto Olaya-Vargas, Hilario Flores Aguilar, Minerva Mata-Rocha, Miguel Ángel Garrido-Hernández, Juan Carlos Solís-Poblano, Nuria Citlalli Luna-Silva, Lena Sarahi Cano-Cuapio, Pierre Mitchel Aristil-Chery, Fernando Herrera-Quezada, Karol Carrillo-Sanchez, Anallely Muñoz-Rivas, Luis Leonardo Flores-Lagunes, Elvia Cristina Mendoza-Caamal, Beatriz Eugenia Villegas-Torres, Vincent González-Osnaya, Elva Jiménez-Hernández, José Refugio Torres-Nava, Jorge Alfonso Martín-Trejo, María de Lourdes Gutiérrez-Rivera, Rosa Martha Espinosa-Elizondo, Laura Elizabeth Merino-Pasaye, María Luisa Pérez-Saldívar, Silvia Jiménez-Morales, Everardo Curiel-Quesada, Haydeé Rosas-Vargas, Juan Manuel Mejía-Arangure, Carmen Alaez-Verson
BACKGROUND: Recurrent genetic alterations contributing to leukemogenesis have been identified in pediatric B-cell Acute Lymphoblastic Leukemia (B-ALL), and some are useful for refining classification, prognosis, and treatment selection. IKZF1plus is a complex biomarker associated with a poor prognosis. It is characterized by IKZF1 deletion coexisting with PAX5 , CDKN2A/2B , or PAR1 region deletions. The mutational spectrum and clinical impact of these alterations have scarcely been explored in Mexican pediatric patients with B-ALL...
2024: Frontiers in Oncology
#31
JOURNAL ARTICLE
Juan Carlos Núñez-Enríquez, Rubí Romo-Rodríguez, Pedro Gaspar-Mendoza, Gabriela Zamora-Herrera, Lizeth Torres-Pineda, Jiovanni Amador-Cardoso, Jebea A López-Blanco, Laura Alfaro-Hernández, Lucero López-García, Arely Rosas-Cruz, Dulce Rosario Alberto-Aguilar, César Omar Trejo-Pichardo, Dalia Ramírez-Ramírez, Astin Cruz-Maza, Janet Flores-Lujano, Nuria Luna-Silva, Angélica Martínez-Martell, Karina Martínez-Jose, Anabel Ramírez-Ramírez, Juan Carlos Solis-Poblano, Patricia Zagoya-Martínez, Vanessa Terán-Cerqueda, Andrea Huerta-Moreno, Álvaro Montiel-Jarquín, Miguel Garrido-Hernández, Raquel Hernández-Ramos, Daniela Olvera-Caraza, Cynthia Shanat Cruz-Medina, Enoch Alvarez-Rodríguez, Lénica Anahí Chávez-Aguilar, Wilfrido Herrera-Olivares, Brianda García-Hidalgo, Lena Sarahí Cano-Cuapio, Claudia Guevara-Espejel, Gerardo Juárez-Avendaño, Juan Carlos Balandrán, Ma Del Rocío Baños-Lara, Mariana Cárdenas-González, Elena R Álvarez-Buylla, Sonia Mayra Pérez-Tapia, Diana Casique-Aguirre, Rosana Pelayo
The main objective of the National Project for Research and Incidence of Childhood Leukemias is to reduce early mortality rates for these neoplasms in the vulnerable regions of Mexico. This project was conducted in the states of Oaxaca, Puebla, and Tlaxcala. A key strategy of the project is the implementation of an effective roadmap to ensure that leukemia patients are the target of maximum benefit of interdisciplinary collaboration between researchers, clinicians, surveyors, and laboratories. This strategy guarantees the comprehensive management of diagnosis and follow-up samples of pediatric patients with leukemia, centralizing, managing, and analyzing the information collected...
2024: Frontiers in Oncology
#32
Azlann Arnett, David A Siegel, Shifan Dai, Trevor D Thompson, Jennifer Foster, Erika J di Pierro, Behnoosh Momin, Philip J Lupo, Andras Heczey
IMPORTANCE: Hepatocellular carcinoma accounts for approximately 80% of liver neoplasms. Globally, hepatocellular carcinoma ranks as the third most lethal cancer, with the number of deaths expected to further increase by 2040. In adults, disparities in incidence and survival are well described while pediatric epidemiology is not well characterized. OBJECTIVE: To describe incidence and survival for pediatric (ages 0-19 years) hepatocellular carcinoma cases and compare these measures to adults (ages ≥20 years) diagnosed with hepatocellular carcinoma...
April 5, 2024: medRxiv
#33
JOURNAL ARTICLE
Chen Wang, Runxi Fu, Yunkun Wang, Jia Wei, Ying Yu, Liuhua Hu, Chenran Zhang
Medulloblastoma (MB), a prevalent pediatric central nervous system tumor, is influenced by microRNAs (miRNAs) that impact tumor initiation and progression. However, the specific involvement of miRNAs in MB tumorigenesis remains unclear. Using single-cell RNA sequencing, we identified ROR2 expression in normal human fetal cerebellum. Subsequent analyses, including immunofluorescence, quantitative real-time PCR (qRT-PCR), and Western blot, assessed ROR2 expression in MB tissues and cell lines. We investigated miR-124-3p and miR-194-5p and their regulatory role in ROR2 expression through the dual-luciferase reporter, qRT-PCR, and western blot assays...
March 19, 2024: Cancer Gene Therapy
#34
JOURNAL ARTICLE
Daniela Kocher, Lei Cao, Romain Guiho, Melanie Langhammer, Yun-Lu Lai, Pauline Becker, Hiba Hamdi, Dennis Friedel, Florian Selt, David Vonhören, Julia Zaman, Gintvile Valinciute, Sonja Herter, Daniel Picard, Johanna Rettenmeier, Kendra K Maass, Kristian W Pajtler, Marc Remke, Andreas von Deimling, Stefan Pusch, Stefan M Pfister, Ina Oehme, David T W Jones, Sebastian Halbach, Tilman Brummer, Juan Pedro Martinez-Barbera, Olaf Witt, Till Milde, Romain Sigaud
INTRODUCTION: Patients with pediatric low-grade gliomas (pLGGs), the most common primary brain tumors in children, can often benefit from MAPK inhibitor (MAPKi) treatment. However, rapid tumor regrowth, also referred to as rebound growth, may occur once treatment is stopped, constituting a significant clinical challenge. METHODS: Four patient-derived pediatric glioma models were investigated to model rebound growth in vitro based on viable cell counts in response to MAPKi treatment and withdrawal...
April 17, 2024: Journal of Neuro-oncology
#35
JOURNAL ARTICLE
Ajami Gikandi, Susan N Chi, Kee Kiat Yeo, Allison F O'Neill, David S Shulman, Steven G DuBois, Natalie B Collins
BACKGROUND: Immune checkpoint inhibitors (ICI) have improved outcomes in a variety of adult cancers and are prescribed with increasing frequency across oncology. However, patterns of off-label use of ICI in pediatrics remain unclear. METHODS: This is a single-institution, retrospective cohort study evaluating off-label ICI use in pediatric and young adult patients with cancer treated at our institution from 2014 to 2022. Response was based on clinician assessment derived from clinical records...
April 2024: Cancer Medicine
#36
JOURNAL ARTICLE
Julie Savagner, Anne Ducassou, Bastien Cabarrou, Gregory Hangard, Marion Gambart, Anne-Isabelle Bertozzi, Eloise Baudou, Sergio Boetto, Delphine Larrieu, Anne Laprie
OBJECTIVE: As craniospinal irradiation (CSI) is delivered more frequently by helical tomotherapy (HT) with few reports about late effects, we analysed all patients treated in our centre over an 11-year period. METHODS AND MATERIALS: Our study included all patients that underwent CSI by HT, between September 2009 and January 2020, in the Department of Radiation Oncology of the Toulouse Cancer Institute. Acute radiotherapy toxicities were reported and medium- to long-term outcomes analysed...
May 2024: Clinical and Translational Radiation Oncology
#37
EDITORIAL
Roger J Packer
No abstract text is available yet for this article.
April 17, 2024: Neuro-oncology
#38
JOURNAL ARTICLE
Betty Y Zhang, Ashley R Wilson-Smith, Elizabeth A Connolly, Madeleine C Strach, Nathan Ussher, Tristan Yan, Vivek A Bhadri
BACKGROUND: Extrapleural pneumonectomy (EPP) is a complex surgical procedure involving en-bloc resection of the parietal and visceral pleura, lung, pericardium, and ipsilateral diaphragm. Small case series of pleural-based sarcoma of predominantly pediatric patients suggest EPP may be a life-prolonging surgical option. We aimed to describe the characteristics and outcomes of adults who underwent EPP at a specialized sarcoma center. METHODS: Clinicopathologic variables, surgical details, and follow-up information were extracted for patients undergoing EPP for pleural-based sarcoma between August 2017 and December 2020...
April 2024: Cancer reports
#39
JOURNAL ARTICLE
Alexandra Widita Swipratami Pangarso, Sri Mulatsih, Mei Neni Sitaresmi, Susanne Verhulst, Gertjan Kaspers, Saskia Mostert
BACKGROUND: Although most children with cancer die in low- and middle-income countries, palliative care receives limited attention in these settings. This study explores parents' perspectives on experiences and needs of children dying from cancer. METHODS: Home visits were conducted to interview parents of children, who were treated for cancer at an Indonesian academic hospital and died between 2019 and 2020, using semi-structured questionnaires. RESULTS: Parents of 49 children (response rate 74%) were interviewed...
April 16, 2024: Pediatric Blood & Cancer
#40
JOURNAL ARTICLE
Jonathan Metts, Wei Xue, Zhengya Gao, Sapna Oberoi, Aaron R Weiss, Rajkumar Venkatramani, Douglas J Harrison
BACKGROUND: Novel therapies are needed for relapsed and refractory rhabdomyosarcoma (RRMS). Phase II clinical trials in RRMS have typically utilized radiologic response as the primary activity endpoint, an approach that poses several limitations in RRMS. In this analysis, we aimed to estimate an event-free survival (EFS) endpoint for RRMS that could be used as a benchmark for future studies. PROCEDURE: We performed a retrospective study of patients with RRMS enrolling on 13 single-agent phase II Children's Oncology Group and legacy group trials from 1997 to 2016...
April 16, 2024: Pediatric Blood & Cancer
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