Alison L Cameron1, Markus C Elze2, Michela Casanova3, Birgit Geoerger4, Mark N Gaze5, Veronique Minard-Colin4, Kieran McHugh6, Rick R van Rijn7, Anna Kelsey8, Hélène Martelli9, Henry Mandeville10, Gianni Bisogno11, Stephen Lowis12, Milind Ronghe13, Daniel Orbach14, Cecile Guizani2, Sabine Fürst-Recktenwald2, Julia C Chisholm15, Johannes H M Merks16. 1. Bristol Haematology and Oncology Centre, University Hospitals Bristol and Weston NHS Foundation Trust, Bristol, United Kingdom. Electronic address: Alison.Cameron@UHBW.nhs.uk. 2. F. Hoffmann-La Roche Ltd, Basel, Switzerland. 3. Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori, Milano, Italy. 4. Department of Pediatric and Adolescent Oncology, Gustave Roussy Cancer Center, University Paris-Saclay, INSERM U1015, Villejuif, France. 5. Department of Oncology, University College London Hospitals NHS Foundation Trust, London, United Kingdom. 6. Department of Radiology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom. 7. Department of Radiology and Nuclear Medicine, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, the Netherlands. 8. Department of Paediatric Histopathology, Manchester University NHS Foundation Trust, Royal Manchester Children's Hospital, Manchester, United Kingdom. 9. Department of Paediatric Surgery, Hôpital Bicêtre-Hôpitaux Universitaires Paris-Saclay, Assistance Publique-Hôpitaux de Paris, Paris, France. 10. Department of Radiotherapy, the Royal Marsden NHS Foundation Trust, and the Institute of Cancer Research, Sutton, United Kingdom. 11. Department of Women's and Children's Health, University of Padova, Padova, Italy. 12. Department of Paediatric and Adolescent Oncology, Bristol Royal Hospital for Children, Bristol, United Kingdom. 13. Department of Paediatric Oncology, Royal Hospital for Children, Glasgow, United Kingdom. 14. SIREDO Oncology Center, Institut Curie, PSL University, Paris, France. 15. Children and Young People's Unit, the Royal Marsden NHS Foundation Trust, and the Institute of Cancer Research, Sutton, United Kingdom. 16. Princess Máxima Center for Pediatric Oncology, Utrecht, the Netherlands; Department of Pediatric Oncology, Emma Children's Hospital-Academic Medical Center (EKZ-AMC), Amsterdam, the Netherlands.
Abstract
PURPOSE: There is limited evidence to define the role of radiation therapy in children with metastatic rhabdomyosarcoma (mRMS). In the international BERNIE study, children with mRMS or non-RMS soft tissue sarcoma were randomized to receive standard chemotherapy with or without bevacizumab, with radiation therapy to all disease sites recommended after chemotherapy cycle 6. We retrospectively evaluated the impact of radiation therapy on survival in the mRMS cohort. METHODS AND MATERIALS: Patients were grouped according to the radiation therapy they received: radical, partial, or none. Radical irradiation was defined as radiation therapy delivered to all disease sites, unless a site was completely surgically resected. Partial irradiation was defined as radiation therapy to ≥1, but not all, disease sites. Landmark analysis excluded patients with an event before day 221. Overall survival (OS) and event-free survival (EFS) were modeled using Cox proportional hazards models. RESULTS: Of 102 patients with mRMS, 97 were included in the analysis for OS and 85 for EFS. Overall, 27 patients received radical irradiation, 46 partial irradiation, and 24 no irradiation. EFS was not significantly different among patient groups after adjustment for prognostic factors (hazard ratio [HR] = 0.520; P = .054 for any vs no irradiation). Radiation therapy was associated with improved OS compared with no radiation therapy (adjusted HR = 0.249; P = .00025), with OS being greater for radical versus partial irradiation (HR = 0.245; P = .039). The 3-year OS rate was 84%, 54%, and 23% for patients receiving radical, partial, and no irradiation, respectively. Radical treatment (surgery, irradiation, or both) of the primary site improved EFS and OS compared with no treatment. CONCLUSIONS: These findings demonstrate variability in the application of radiation therapy for mRMS and support the routine use of radical treatment to the primary site. Radical irradiation to metastatic sites may further improve OS. The burden of such treatment should be balanced against prognosis; further studies are needed.
PURPOSE: There is limited evidence to define the role of radiation therapy in children with metastatic rhabdomyosarcoma (mRMS). In the international BERNIE study, children with mRMS or non-RMS soft tissue sarcoma were randomized to receive standard chemotherapy with or without bevacizumab, with radiation therapy to all disease sites recommended after chemotherapy cycle 6. We retrospectively evaluated the impact of radiation therapy on survival in the mRMS cohort. METHODS AND MATERIALS: Patients were grouped according to the radiation therapy they received: radical, partial, or none. Radical irradiation was defined as radiation therapy delivered to all disease sites, unless a site was completely surgically resected. Partial irradiation was defined as radiation therapy to ≥1, but not all, disease sites. Landmark analysis excluded patients with an event before day 221. Overall survival (OS) and event-free survival (EFS) were modeled using Cox proportional hazards models. RESULTS: Of 102 patients with mRMS, 97 were included in the analysis for OS and 85 for EFS. Overall, 27 patients received radical irradiation, 46 partial irradiation, and 24 no irradiation. EFS was not significantly different among patient groups after adjustment for prognostic factors (hazard ratio [HR] = 0.520; P = .054 for any vs no irradiation). Radiation therapy was associated with improved OS compared with no radiation therapy (adjusted HR = 0.249; P = .00025), with OS being greater for radical versus partial irradiation (HR = 0.245; P = .039). The 3-year OS rate was 84%, 54%, and 23% for patients receiving radical, partial, and no irradiation, respectively. Radical treatment (surgery, irradiation, or both) of the primary site improved EFS and OS compared with no treatment. CONCLUSIONS: These findings demonstrate variability in the application of radiation therapy for mRMS and support the routine use of radical treatment to the primary site. Radical irradiation to metastatic sites may further improve OS. The burden of such treatment should be balanced against prognosis; further studies are needed.
Authors: Meerim Park; Jun Ah Lee; Hye Young Jin; Joo-Young Kim; Jong Woong Park; June Hyuk Kim; Hyun Guy Kang; Seog Yun Park; Eun Young Park; Hyeon Jin Park; Byung Kiu Park Journal: J Cancer Res Clin Oncol Date: 2022-07-23 Impact factor: 4.322
Authors: Si-Qin Liu; Fang-Hua Ye; Chen-Ying Fan; Min Peng; Jia-Jia Dong; Wen-Jun Deng; Hui Zhang; Yan Yu; Liang-Chun Yang Journal: Zhongguo Dang Dai Er Ke Za Zhi Date: 2022 Sept 15