Harmonization of postmortem donations for pediatric brain tumors and molecular characterization of diffuse midline gliomas.

Adolescent Adult Animals Autopsy Biomarkers, Tumor / genetics Brain Neoplasms / genetics Child Child, Preschool Female Gene Expression Profiling Gene Expression Regulation, Neoplastic Glioma / genetics Histones / genetics Humans Infant Male Mice, Inbred NOD Mice, SCID Mutation Tumor Cells, Cultured Xenograft Model Antitumor Assays Young Adult

Journal

Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288

Informations de publication

Date de publication:
02 07 2020
Historique:
received: 07 04 2020
accepted: 11 06 2020
entrez: 4 7 2020
pubmed: 4 7 2020
medline: 2 12 2020
Statut: epublish

Résumé

Children diagnosed with brain tumors have the lowest overall survival of all pediatric cancers. Recent molecular studies have resulted in the discovery of recurrent driver mutations in many pediatric brain tumors. However, despite these molecular advances, the clinical outcomes of high grade tumors, including H3K27M diffuse midline glioma (H3K27M DMG), remain poor. To address the paucity of tissue for biological studies, we have established a comprehensive protocol for the coordination and processing of donated specimens at postmortem. Since 2010, 60 postmortem pediatric brain tumor donations from 26 institutions were coordinated and collected. Patient derived xenograft models and cell cultures were successfully created (76% and 44% of attempts respectively), irrespective of postmortem processing time. Histological analysis of mid-sagittal whole brain sections revealed evidence of treatment response, immune cell infiltration and the migratory path of infiltrating H3K27M DMG cells into other midline structures and cerebral lobes. Sequencing of primary and disseminated tumors confirmed the presence of oncogenic driver mutations and their obligate partners. Our findings highlight the importance of postmortem tissue donations as an invaluable resource to accelerate research, potentially leading to improved outcomes for children with aggressive brain tumors.

Identifiants

DOI: 10.1038/s41598-020-67764-2 PMID: 32616776 PMC: PMC7331588
pubmed: 32616776
doi: 10.1038/s41598-020-67764-2
pii: 10.1038/s41598-020-67764-2
pmc: PMC7331588
doi:

Substances chimiques

Biomarkers, Tumor 0
Histones 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

10954

Références

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Auteurs

Madhuri Kambhampati (M)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

Eshini Panditharatna (E)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Department of Pediatric Oncology, Dana-Farber Cancer Institute, Boston, MA, USA.

Sridevi Yadavilli (S)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

Karim Saoud (K)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

Sulgi Lee (S)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA.

Augustine Eze (A)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

M I Almira-Suarez (MI)

Department of Pathology, Children's National Hospital, Washington, DC, USA.
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA.

Lauren Hancock (L)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Center for Cancer and Immunology Research, Children's National Hospital, Washington, DC, USA.

Erin R Bonner (ER)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA.

Jamila Gittens (J)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.
PTC Therapeutics, South Plainfield, NJ, USA.

Mojca Stampar (M)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA.

Krutika Gaonkar (K)

Center for Data-Driven Discovery in Biomedicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Adam C Resnick (AC)

Center for Data-Driven Discovery in Biomedicine, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Cassie Kline (C)

Pediatric Hematology-Oncology and Neurology, UCSF Benioff Children's Hospital, San Francisco, CA, USA.
Division of Oncology, Children's Hospital of Philadelphia, Philadelphia, PA, 19104, USA.

Cheng-Ying Ho (CY)

Department of Pathology and Neurology, University of Maryland School of Medicine, Baltimore, MD, USA.

Angela J Waanders (AJ)

Feinberg School of Medicine, Northwestern University, Chicago, IL, USA.

Maria-Magdalena Georgescu (MM)

NeuroMarkers PLLC, Houston, TX, USA.

Naomi E Rance (NE)

Department of Pathology, University of Arizona College of Medicine, Tucson, AZ, USA.

Yong Kim (Y)

Pediatric Oncology Branch, National Cancer Institute, Bethesda, MD, USA.

Courtney Johnson (C)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

Brian R Rood (BR)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Center for Cancer and Immunology Research, Children's National Hospital, Washington, DC, USA.

Lindsay B Kilburn (LB)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Center for Cancer and Immunology Research, Children's National Hospital, Washington, DC, USA.

Eugene I Hwang (EI)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Center for Cancer and Immunology Research, Children's National Hospital, Washington, DC, USA.

Sabine Mueller (S)

Pediatric Hematology-Oncology and Neurology, UCSF Benioff Children's Hospital, San Francisco, CA, USA.
Department of Oncology, Children's Research Center, University Children's Hospital Zürich, Zurich, Switzerland.

Roger J Packer (RJ)

Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.

Miriam Bornhorst (M)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA. mbornhorst@childrensnational.org.
Brain Tumor Institute, Children's National Hospital, Washington, DC, USA. mbornhorst@childrensnational.org.
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA. mbornhorst@childrensnational.org.

Javad Nazarian (J)

Center for Genetic Medicine Research, Children's National Hospital, Washington, DC, USA. Javad.Nazarian@kispi.uzh.ch.
Department of Oncology, Children's Research Center, University Children's Hospital Zürich, Zurich, Switzerland. Javad.Nazarian@kispi.uzh.ch.
The George Washington University School of Medicine and Health Sciences, Washington, DC, USA. Javad.Nazarian@kispi.uzh.ch.

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Classifications MeSH

questionsmedicales.fr › Personnes › Tranches d'âge › Adolescent › Harmonization of postmortem donations for...
questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Harmonization of postmortem donations for...
questionsmedicales.fr › Eucaryotes › Animaux › Harmonization of postmortem donations for...
questionsmedicales.fr › Sciences sociales › Criminologie › Sciences légales › Médecine légale › Autopsie › Harmonization of postmortem donations for...
questionsmedicales.fr › Facteurs biologiques › Marqueurs biologiques › Marqueurs biologiques tumoraux › Harmonization of postmortem donations for...
questionsmedicales.fr › Maladies du système nerveux › Tumeurs du système nerveux › Tumeurs du système nerveux central › Tumeurs du cerveau › Harmonization of postmortem donations for...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Harmonization of postmortem donations for...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Enfant d'âge préscolaire › Harmonization of postmortem donations for...
questionsmedicales.fr › Techniques d'investigation › Techniques génétiques › Analyse de profil d'expression de gènes › Harmonization of postmortem donations for...
questionsmedicales.fr › Phénomènes génétiques › Régulation de l'expression des gènes › Régulation de l'expression des gènes tumoraux › Harmonization of postmortem donations for...
questionsmedicales.fr › Tumeurs › Tumeurs par type histologique › Tumeurs du tissu nerveux › Tumeurs neuroectodermiques › Tumeurs neuroépitheliales › Gliome › Harmonization of postmortem donations for...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Nucléoprotéines › Histone › Harmonization of postmortem donations for...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Harmonization of postmortem donations for...
questionsmedicales.fr › Personnes › Tranches d'âge › Nourrisson › Harmonization of postmortem donations for...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Rodentia › Muridae › Murinae › Souris › Lignées consanguines de souris › Souris de lignée NOD › Harmonization of postmortem donations for...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Rodentia › Muridae › Murinae › Souris › Souches mutantes de souris › Souris SCID › Harmonization of postmortem donations for...
questionsmedicales.fr › Phénomènes génétiques › Variation génétique › Mutation › Harmonization of postmortem donations for...
questionsmedicales.fr › Cellules › Cellules cultivées › Cellules cancéreuses en culture › Harmonization of postmortem donations for...
questionsmedicales.fr › Techniques d'investigation › Transplantation tumorale › Tests d'activité antitumorale sur modèle de xénogreffe › Harmonization of postmortem donations for...
questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Jeune adulte › Harmonization of postmortem donations for...