Hugo Câmara-Costa1, Anika Resch2, Virginie Kieffer3, Clémence Lalande4, Geraldina Poggi5, Colin Kennedy6, Kim Bull6, Gabriele Calaminus7, Jacques Grill4, François Doz8, Stefan Rutkowski2, Maura Massimino9, Rolf-Dieter Kortmann10, Birgitta Lannering11, Georges Dellatolas12, Mathilde Chevignard13. 1. National Institute of Health and Medical Research, INSERM U1178, Paris, France. Electronic address: hugocamaracosta@gmail.com. 2. University Medical Center Hamburg-Eppendorf, Hamburg, Germany. 3. Saint Maurice Hospitals, Saint Maurice, France. 4. Institut Gustave Roussy, Villejuif, France. 5. Scientific Institute, IRCCS Eugenio Medea, Bosisio Parini, Lecco, Italy. 6. University of Southampton, Faculty of Medicine, Southampton, United Kingdom. 7. Paediatric Oncology, University of Muenster, Muenster, Germany. 8. Institut Curie and University Paris Descartes, Sorbonne Paris Cité, Paris, France. 9. Fondazione IRCCS, Istituto Nazionale Tumori, Milan, Italy. 10. Department of Radiation Therapy, University of Leipzig, Leipzig, Germany. 11. Paediatric Oncology, University of Gothenburg, Gothenburg, Sweden. 12. National Institute of Health and Medical Research, INSERM U1178, Paris, France. 13. Rehabilitation Department for Children With Acquired Neurological Injury, Saint Maurice Hospitals, Saint Maurice, and Sorbonne Universités, UPMC Universités Paris, INSERM CNRS, Paris, France.
Abstract
PURPOSE: In the European HIT-SIOP PNET4 randomized controlled trial, children with standard risk medulloblastoma were allocated to hyperfractionated radiation therapy (HFRT arm, including a partially focused boost) or standard radiation therapy (STRT arm), followed, in both arms, by maintenance chemotherapy. Event-free survival was similar in both arms. Previous work showed that the HFRT arm was associated with worse growth and better questionnaire-based executive function, especially in children <8 years of age at diagnosis. Therefore, the aim of this study was to compare performance-based cognitive outcomes between treatment arms. METHODS AND MATERIALS: Neuropsychological data were collected prospectively in 137 patients. Using the Wechsler Intelligence Scales, Kaufman Assessment Battery for Children, and Raven's Progressive Matrices, we estimated full-scale intelligence quotient (FSIQ) and, when available, verbal IQ (VIQ), performance IQ (PIQ), working memory index (WMI), and processing speed index (PSI). RESULTS: Among the 137 participants (HFRT arm n=71, STRT arm n=66, 63.5% males), mean (±SD) ages at diagnosis and assessment respectively were 9.3 (±3.2) years of age (40.8% < 8 years of age at diagnosis) and 14.6 (±4.3) years of age. Mean (±SD) FSIQ was 88 (±19), and mean intergroup difference was 3.88 (95% confidence interval: -2.66 to 10.42, P=.24). No significant differences were found in children >8 years of age at diagnosis. In children <8 years of age at diagnosis, a marginally significant trend toward higher VIQ was found in those treated in the HFRT arm; a similar trend was found for PSI but not for PIQ, WMI, or FSIQ (mean intergroup differences were: 12.02 for VIQ [95% CI: 2.37-21.67; P=.02]; 3.77 for PIQ [95% CI: -5.19 to 12.74; P>.10]; 5.20 for WMI [95% CI: -2.07 to 12.47; P>.10]; 10.90 for PSI [95% CI: -1.54 to 23.36; P=.08]; and 5.28 for FSIQ [95% CI: -4.23 to 14.79; P>.10]). CONCLUSIONS:HFRT was associated with marginally higher VIQ in children <8 years of age at diagnosis, consistent with a previous report using questionnaire-based data. However, overall cognitive ability was not significantly different.
RCT Entities:
PURPOSE: In the European HIT-SIOP PNET4 randomized controlled trial, children with standard risk medulloblastoma were allocated to hyperfractionated radiation therapy (HFRT arm, including a partially focused boost) or standard radiation therapy (STRT arm), followed, in both arms, by maintenance chemotherapy. Event-free survival was similar in both arms. Previous work showed that the HFRT arm was associated with worse growth and better questionnaire-based executive function, especially in children <8 years of age at diagnosis. Therefore, the aim of this study was to compare performance-based cognitive outcomes between treatment arms. METHODS AND MATERIALS: Neuropsychological data were collected prospectively in 137 patients. Using the Wechsler Intelligence Scales, Kaufman Assessment Battery for Children, and Raven's Progressive Matrices, we estimated full-scale intelligence quotient (FSIQ) and, when available, verbal IQ (VIQ), performance IQ (PIQ), working memory index (WMI), and processing speed index (PSI). RESULTS: Among the 137 participants (HFRT arm n=71, STRT arm n=66, 63.5% males), mean (±SD) ages at diagnosis and assessment respectively were 9.3 (±3.2) years of age (40.8% < 8 years of age at diagnosis) and 14.6 (±4.3) years of age. Mean (±SD) FSIQ was 88 (±19), and mean intergroup difference was 3.88 (95% confidence interval: -2.66 to 10.42, P=.24). No significant differences were found in children >8 years of age at diagnosis. In children <8 years of age at diagnosis, a marginally significant trend toward higher VIQ was found in those treated in the HFRT arm; a similar trend was found for PSI but not for PIQ, WMI, or FSIQ (mean intergroup differences were: 12.02 for VIQ [95% CI: 2.37-21.67; P=.02]; 3.77 for PIQ [95% CI: -5.19 to 12.74; P>.10]; 5.20 for WMI [95% CI: -2.07 to 12.47; P>.10]; 10.90 for PSI [95% CI: -1.54 to 23.36; P=.08]; and 5.28 for FSIQ [95% CI: -4.23 to 14.79; P>.10]). CONCLUSIONS: HFRT was associated with marginally higher VIQ in children <8 years of age at diagnosis, consistent with a previous report using questionnaire-based data. However, overall cognitive ability was not significantly different.
Authors: Cara F Levitch; Benjamin Malkin; Lauren Latella; Whitney Guerry; Sharon L Gardner; Jonathan L Finlay; Stephen A Sands Journal: Neurooncol Pract Date: 2021-05-18
Authors: Vijay Ramaswamy; Marc Remke; Eric Bouffet; Simon Bailey; Steven C Clifford; Francois Doz; Marcel Kool; Christelle Dufour; Gilles Vassal; Till Milde; Olaf Witt; Katja von Hoff; Torsten Pietsch; Paul A Northcott; Amar Gajjar; Giles W Robinson; Laetitia Padovani; Nicolas André; Maura Massimino; Barry Pizer; Roger Packer; Stefan Rutkowski; Stefan M Pfister; Michael D Taylor; Scott L Pomeroy Journal: Acta Neuropathol Date: 2016-04-04 Impact factor: 17.088
Authors: Hugo Câmara-Costa; Kim S Bull; Colin Kennedy; Andreas Wiener; Gabriele Calaminus; Anika Resch; Virginie Kieffer; Clémence Lalande; Geraldina Poggi; Katja von Hoff; Jacques Grill; François Doz; Stefan Rutkowski; Maura Massimino; Rolf-Dieter Kortmann; Birgitta Lannering; Georges Dellatolas; Mathilde Chevignard Journal: Neurooncol Pract Date: 2017-02-10