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 et al.
In: Frontiers in oncology, Vol. 12, 926196, 22.06.2022.Research output: Contribution to journal › Article › Academic › peer-review
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TY - JOUR
T1 - Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma
T2 - A Retrospective Analysis of the SIOP-E-HGG/DIPG Project
AU - Chavaz, Lara
AU - Janssens, Geert O.
AU - Bolle, Stephanie
AU - Mandeville, Henry
AU - Ramos-Albiac, Monica
AU - van Beek, Karen
AU - Benghiat, Helen
AU - Hoeben, Bianca
AU - Morales la Madrid, Andres
AU - Seidel, Clemens
AU - Kortmann, Rolf-Dieter
AU - Hargrave, Darren
AU - Gandola, Lorenza
AU - Pecori, Emilia
AU - van Vuurden, Dannis G.
AU - Biassoni, Veronica
AU - Massimino, Maura
AU - Kramm, Christof M.
AU - von Bueren, Andre O.
N1 - 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.
PY - 2022/6/22
Y1 - 2022/6/22
N2 - 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 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.
AB - 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 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.
KW - adolescent
KW - child
KW - diffuse intrinsic pontine glioma (DIPG)
KW - radiotherapy
KW - re-irradiation (re-RT)
UR - http://www.scopus.com/inward/record.url?scp=85133775633&partnerID=8YFLogxK
U2 - 10.3389/fonc.2022.926196
DO - 10.3389/fonc.2022.926196
M3 - Article
C2 - 35814457
VL - 12
JO - Frontiers in oncology
JF - Frontiers in oncology
SN - 2234-943X
M1 - 926196
ER -
ID: 25080561