Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma: A Retrospective Analysis of the SIOP-E-HGG/DIPG Project
Chavaz, Lara; Janssens, Geert O; Bolle, Stephanie; Mandeville, Henry; Ramos-Albiac, Monica; Van Beek, Karen; Benghiat, Helen; Hoeben, Bianca; Morales La Madrid, Andres; Seidel, Clemens; Kortmann, Rolf-Dieter; Hargrave, Darren; Gandola, Lorenza; Pecori, Emilia; van Vuurden, Dannis G; Biassoni, Veronica; Massimino, Maura; Kramm, Christof M; von Bueren, Andre O
(2022) Frontiers in oncology, volume 12, pp. 1 - 8
(Article)
Abstract
Purpose: The aim of this study is to investigate the spectrum of neurological triad improvement in patients with diffuse intrinsic pontine glioma (DIPG) treated by re-irradiation (re-RT) at first progression. Methods: We carried out a re-analysis of the SIOP-E retrospective DIPG cohort by investigating the clinical benefits after re-RT with
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a focus on the neurological triad (cranial nerve deficits, ataxia, and long tract signs). Patients were categorized as “responding” or “non-responding” to re-RT. To assess the interdependence between patients’ characteristics and clinical benefits, we used a chi-square or Fisher’s exact test. Survival according to clinical response to re-RT was calculated by the Kaplan–Meier method. Results: As earlier reported, 77% (n = 24/31) of patients had any clinical benefit after re-RT. Among 25/31 well-documented patients, 44% (n = 11/25) had improvement in cranial nerve palsies, 40% (n = 10/25) had improvement in long-tract signs, and 44% (11/25) had improvement in cerebellar signs. Clinical benefits were observed in at least 1, 2, or 3 out of 3 symptoms of the DIPG triad, in 64%, 40%, and 24%, respectively. Patients irradiated with a dose ≥20 Gy versus <20 Gy may improve slightly better with regard to ataxia (67% versus 23%; p-value = 0.028). The survival from the start of re-RT to death was not different between responding and non-responding DIPG patients (p-value = 0.871). Conclusion: A median re-irradiation dose of 20 Gy provides a neurological benefit in two-thirds of patients with an improvement of at least one symptom of the triad. DIPG patients receiving ≥20 Gy appear to improve slightly better with regard to ataxia; however, we need more data to determine whether dose escalation up to 30 Gy provides additional benefits.
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Keywords: adolescent, child, diffuse intrinsic pontine glioma (DIPG), radiotherapy, re-irradiation (re-RT), Oncology, Cancer Research, Journal Article
ISSN: 2234-943X
Publisher: Frontiers Media S. A.
Note: Funding Information: DH was supported by the National Institute for Health Research/Biomedical Research Centre at Great Ormond Street Hospital for Children, NHS Foundation Trust, and University College London. Funding Information: HM was supported by the National Institute of Health Research/Biomedical Research Centre at The Royal Marsden NHS Foundation Trust, Sutton. Funding Information: We thank Dr. András Treszl for statistical advice. We would like to thank the CANSEARCH Foundation for continuous support. Funding Information: This work was supported in part by Deutsche Kinderkrebsstiftung. Publisher Copyright: Copyright © 2022 Chavaz, Janssens, Bolle, Mandeville, Ramos-Albiac, Van Beek, Benghiat, Hoeben, Morales La Madrid, Seidel, Kortmann, Hargrave, Gandola, Pecori, van Vuurden, Biassoni, Massimino, Kramm and von Bueren.
(Peer reviewed)