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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 journalArticleAcademicpeer-review

Harvard

Chavaz, L, Janssens, GO, Bolle, S, Mandeville, H, Ramos-Albiac, M, van Beek, K, Benghiat, H, Hoeben, B, Morales la Madrid, A, Seidel, C, Kortmann, R-D, Hargrave, D, Gandola, L, Pecori, E, van Vuurden, DG, Biassoni, V, Massimino, M, Kramm, CM & von Bueren, AO 2022, 'Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma: A Retrospective Analysis of the SIOP-E-HGG/DIPG Project', Frontiers in oncology, vol. 12, 926196. https://doi.org/10.3389/fonc.2022.926196

APA

Chavaz, L., Janssens, G. O., Bolle, S., Mandeville, H., Ramos-Albiac, M., van Beek, K., Benghiat, H., Hoeben, B., Morales la Madrid, A., Seidel, C., Kortmann, R-D., Hargrave, D., Gandola, L., Pecori, E., van Vuurden, D. G., Biassoni, V., Massimino, M., Kramm, C. M., & von Bueren, A. O. (2022). Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma: A Retrospective Analysis of the SIOP-E-HGG/DIPG Project. Frontiers in oncology, 12, [926196]. https://doi.org/10.3389/fonc.2022.926196

Vancouver

Chavaz L, Janssens GO, Bolle S, Mandeville H, Ramos-Albiac M, van Beek K et al. Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma: A Retrospective Analysis of the SIOP-E-HGG/DIPG Project. Frontiers in oncology. 2022 Jun 22;12:926196. doi: 10.3389/fonc.2022.926196

Author

BibTeX

@article{b740e5da01804aacb542343e3084915d,
title = "Neurological Symptom Improvement After Re-Irradiation in Patients With Diffuse Intrinsic Pontine Glioma: A Retrospective Analysis of the SIOP-E-HGG/DIPG Project",
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 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{\textquoteright} characteristics and clinical benefits, we used a chi-square or Fisher{\textquoteright}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.",
keywords = "adolescent, child, diffuse intrinsic pontine glioma (DIPG), radiotherapy, re-irradiation (re-RT)",
author = "Lara Chavaz and Janssens, {Geert O.} and Stephanie Bolle and Henry Mandeville and Monica Ramos-Albiac and {van Beek}, Karen and Helen Benghiat and Bianca Hoeben and {Morales la Madrid}, Andres and Clemens Seidel and Rolf-Dieter Kortmann and Darren Hargrave and Lorenza Gandola and Emilia Pecori and {van Vuurden}, {Dannis G.} and Veronica Biassoni and Maura Massimino and Kramm, {Christof M.} and {von Bueren}, {Andre O.}",
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. Andr{\'a}s 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 {\textcopyright} 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.",
year = "2022",
month = jun,
day = "22",
doi = "10.3389/fonc.2022.926196",
language = "English",
volume = "12",
journal = "Frontiers in oncology",
issn = "2234-943X",
publisher = "Frontiers Media S.A.",

}

RIS

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