Response assessment in paediatric low-grade glioma: recommendations from the Response Assessment in Pediatric Neuro-Oncology (RAPNO) working group

Jason Fangusaro; Olaf Witt; Pablo Hernáiz Driever; Asim K Bag; Peter de Blank; Nadja Kadom; Lindsay Kilburn; Robert M Lober; Nathan J Robison; Michael J Fisher; Roger J Packer; Tina Young Poussaint; Ludmila Papusha; Shivaram Avula; Alba A Brandes; Eric Bouffet; Daniel Bowers; Anton Artemov; Murali Chintagumpala; David Zurakowski; Martin van den Bent; Brigitte Bison; Kristen W Yeom; Walter Taal; Katherine E Warren

doi:10.1016/S1470-2045(20)30064-4

Response assessment in paediatric low-grade glioma: recommendations from the Response Assessment in Pediatric Neuro-Oncology (RAPNO) working group

Lancet Oncol. 2020 Jun;21(6):e305-e316. doi: 10.1016/S1470-2045(20)30064-4.

Authors

Jason Fangusaro¹, Olaf Witt², Pablo Hernáiz Driever³, Asim K Bag⁴, Peter de Blank⁵, Nadja Kadom⁶, Lindsay Kilburn⁷, Robert M Lober⁸, Nathan J Robison⁹, Michael J Fisher¹⁰, Roger J Packer¹¹, Tina Young Poussaint¹², Ludmila Papusha¹³, Shivaram Avula¹⁴, Alba A Brandes¹⁵, Eric Bouffet¹⁶, Daniel Bowers¹⁷, Anton Artemov¹³, Murali Chintagumpala¹⁸, David Zurakowski¹⁹, Martin van den Bent²⁰, Brigitte Bison²¹, Kristen W Yeom²², Walter Taal²³, Katherine E Warren²⁴

Affiliations

¹ Department of Pediatric Hematology, Oncology and Stem Cell Transplantation, Aflac Cancer Center, Emory University and the Children's Healthcare of Atlanta, Atlanta, GA, USA. Electronic address: jfangus@emory.edu.
² Department CCU Pediatric Oncology, Hopp Children's Cancer Center (KiTZ), University Hospital and German Cancer Research Center (DKFZ), Heidelberg, Germany.
³ Department of Pediatric Oncology and Hematology, Charité-Universitätsmedizin Berlin, Freie Universität Berlin, Humboldt-Universität zu Berlin, Berlin Institute of Health, Berlin, Germany.
⁴ Department of Diagnostic Imaging, St Jude Children's Research Hospital, Memphis, TN, USA.
⁵ Department of Pediatrics, University of Cincinnati and Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
⁶ Pediatric Neuroradiology, Emory University and the Children's Healthcare of Atlanta, Atlanta, GA, USA.
⁷ Department of Hematology and Oncology, National Medical Center, Washington, DC, USA.
⁸ Department of Neurosurgery, Dayton Children's Hospital and Wright State University Boonshoft School of Medicine, Dayton, OH, USA.
⁹ Division of Hematology and Oncology, Children's Hospital Los Angeles, Keck School of Medicine of the University of Southern California, Los Angeles, CA, USA.
¹⁰ Division of Oncology, Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.
¹¹ Center for Neuroscience and Behavioral Medicine, Children's National Hospital, Washington, DC, USA.
¹² Department of Radiology, Boston Children's Hospital and Harvard Medical School, Boston, MA, USA.
¹³ Department of Neuro-Oncology, Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology, Moscow, Russia.
¹⁴ Department of Radiology, Alder Hey Children's NHS Foundation Trust, Liverpool, UK.
¹⁵ Medical Oncology Department, AUSL-IRCCS Scienze Neurologiche, Bologna, Italy.
¹⁶ Division of Hematology/Oncology, The Hospital for Sick Children, Toronto, ON, Canada.
¹⁷ Division of Pediatric Hematology and Oncology, University of Texas Southwestern, Dallas, TX, USA.
¹⁸ Texas Children's Cancer Center, Baylor College of Medicine, Houston, TX, USA.
¹⁹ Department of Anesthesiology, Critical Care and Pain Medicine, Boston Children's Hospital and Harvard Medical School, Boston, MA, USA.
²⁰ Brain Tumor Center, Erasmus MC Cancer Institute, Rotterdam, Netherlands.
²¹ Diagnostic and Interventional Radiology, Universitatsklinikum Würzburg, Würzburg, Germany.
²² Department of Radiology, Lucile Packard Children's Hospital, Stanford University, Palo Alto, CA, USA.
²³ Department of Neurology/Neuro-Oncology, Erasmus MC Cancer Institute, Rotterdam, Netherlands.
²⁴ Department of Pediatric Oncology, Dana-Farber/Boston Children's Cancer and Blood Disorders Center, Dana-Farber Cancer Institute, Boston, MA, USA.

PMID: 32502457
DOI: 10.1016/S1470-2045(20)30064-4

Abstract

Paediatric low-grade gliomas (also known as pLGG) are the most common type of CNS tumours in children. In general, paediatric low-grade gliomas show clinical and biological features that are distinct from adult low-grade gliomas, and the developing paediatric brain is more susceptible to toxic late effects of the tumour and its treatment. Therefore, response assessment in children requires additional considerations compared with the adult Response Assessment in Neuro-Oncology criteria. There are no standardised response criteria in paediatric clinical trials, which makes it more difficult to compare responses across studies. The Response Assessment in Pediatric Neuro-Oncology working group, consisting of an international panel of paediatric and adult neuro-oncologists, clinicians, radiologists, radiation oncologists, and neurosurgeons, was established to address issues and unique challenges in assessing response in children with CNS tumours. We established a subcommittee to develop consensus recommendations for response assessment in paediatric low-grade gliomas. Final recommendations were based on literature review, current practice, and expert opinion of working group members. Consensus recommendations include imaging response assessments, with additional guidelines for visual functional outcomes in patients with optic pathway tumours. As with previous consensus recommendations, these recommendations will need to be validated in prospective clinical trials.

Publication types

Practice Guideline
Review

MeSH terms

Age of Onset
Central Nervous System Neoplasms / diagnostic imaging*
Central Nervous System Neoplasms / epidemiology
Central Nervous System Neoplasms / pathology
Central Nervous System Neoplasms / therapy*
Child
Consensus
Endpoint Determination / standards*
Female
Glioma / diagnostic imaging*
Glioma / epidemiology
Glioma / pathology
Glioma / therapy*
Humans
Magnetic Resonance Imaging / standards
Male
Neoplasm Grading
Neuroimaging / standards*
Perfusion Imaging / standards
Positron-Emission Tomography / standards
Predictive Value of Tests
Time Factors
Treatment Outcome
Tumor Burden