The Impact of Radiation Therapy in Children and Adolescents With Metastatic Rhabdomyosarcoma

Cameron, Alison; Elze, Markus C.; Casanova, Michela; Geoerger, Birgit; Gaze, Mark; Minard‐Colin, Véronique; McHugh, Kieran; Rijn, Rick R. van; Kelsey, Anna; Martelli, Hélène; Mandeville, Henry; Bisogno, Gianni; Lowis, Stephen P.; Ronghe, Milind; Orbach, Daniel; Guizani, Cecile; Fürst‐Recktenwald, Sabine; Chisholm, Julia; Merks, Johannes H. M.

doi:10.1016/j.ijrobp.2021.06.031

Cited by 17 publications

(40 citation statements)

References 14 publications

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“…We also considered the following prognostic (d) number of metastatic sites less than or equal to two versus greater than or equal to three. 10 To examine the impact of radiotherapy on survival, we used a landmark analysis 25 as done in the study by Cameron et al, 20 the aim being to reduce the risk of immortality bias in patients given radiotherapy, and adjust for the fact that some patients' disease progressed before they received radiotherapy. The analysis only included patients who were alive without progression at day 221, which was the planned end of cycle 9 plus a 1-month grace period.…”

Section: Discussionmentioning

confidence: 99%

“…A standard approach is still lacking, and there is a marked variability in how radiotherapy has reportedly been used, even in clinical trials. 20 As most studies over the years have focused on intensifying systemic treatments to improve the outcome of patients with metastatic RMS, [12][13][14][15][16][17][18] the potential role of radiotherapy may have sometimes been underestimated.…”

Section: -Year Os (%) Os P-value (Lor-rank)mentioning

confidence: 99%

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Metastatic rhabdomyosarcoma: Evidence of the impact of radiotherapy on survival. A retrospective single‐center experience

Ferrari

Bergamaschi

Chiaravalli

et al. 2022

Pediatric Blood & Cancer

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Background The prognosis for patients with metastatic rhabdomyosarcoma (RMS) remains largely unsatisfactory despite the adoption of intensive multimodal therapy. To assess the role of different treatments adopted over the years, we retrospectively analyzed a cohort of patients <21 years old with metastatic RMS, treated from 1990 to 2020 at a referral center for pediatric sarcomas. Methods Patients were treated using a multimodal approach that included surgery, radiotherapy, and chemotherapy (both high‐dose chemotherapy and maintenance therapy in some cases). The type of radiotherapy administered was categorized as radical (to all sites of disease); partial (to at least one, but not all sites of disease); or none. A landmark analysis was used to examine the impact of radiotherapy on survival, that is, patients who had an event before day 221 were excluded from the analysis. Results The series included 80 patients. Event‐free survival (EFS) and overall survival (OS) rates at 5 years were 17.3% and 21.3%, respectively. Survival was significantly associated with radiotherapy to metastatic sites, and with the radiotherapy category. In particular, 5‐year EFS and OS rates were 70.6% and 76.0% for patients given radical radiotherapy, and 4.8% and 10.7%, respectively, for those given partial radiotherapy or none. Using the Cox multivariable analysis, OS correlated significantly with radiotherapy category. Conclusions While confirming the poor overall outcome of patients with metastatic RMS, this study identified radiotherapy—when given to all sites of disease (including metastases)—as the main variable influencing survival.

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Section: Discussionmentioning

confidence: 99%

Section: -Year Os (%) Os P-value (Lor-rank)mentioning

confidence: 99%

Metastatic rhabdomyosarcoma: Evidence of the impact of radiotherapy on survival. A retrospective single‐center experience

Ferrari

Bergamaschi

Chiaravalli

et al. 2022

Pediatric Blood & Cancer

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“…In the analysis of the BERNIE study, a cutoff of day 221 was used to define radical radiotherapy to take this issue into account. 21 Cases with extensive metastatic disease may require metastatic radiotherapy delivered in sequential phases, treated in succession to limit the associated bone marrow and other acute toxicities. Delayed radiotherapy for metastatic sites is also recommended in COG studies, with metastatic site radiotherapy to now occur at approximately week 40 after completion of primary chemotherapy on ARST2031 (previously, on the prior high-risk studies, ARST0431 and ARST08P1, there was the option to deliver metastatic site radiotherapy either with primary site radiation or after completion of primary chemotherapy).…”

Section: Evidencementioning

confidence: 99%

“…Yet, it is apparent that these guidelines were not always adhered to, particularly with the challenges of treating metastatic rhabdomyosarcoma and multiple disease sites leading to delays in radiotherapy. In the analysis of the BERNIE study, a cutoff of day 221 was used to define radical radiotherapy to take this issue into account 21 . Cases with extensive metastatic disease may require metastatic radiotherapy delivered in sequential phases, treated in succession to limit the associated bone marrow and other acute toxicities.…”

Section: Consensus Guidelinesmentioning

confidence: 99%

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

Casey

Mandeville

Bradley

et al. 2022

Pediatric Blood & Cancer

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The International Soft Tissue Sarcoma Database Consortium (INSTRuCT) consists of a collaboration between the Children's Oncology Group (COG) Soft Tissue Sarcoma Committee, the European pediatric Soft Tissue Sarcoma Study Group (EpSSG), and the Cooperative Weichteilsarkom Studiengruppe (CWS). As part of the larger initiative of INSTRuCT to provide consensus expert opinions for clinical treatment of pediatric soft tissue sarcoma, we sought to provide updated, evidenced-based consensus guidelines for local treatment of parameningeal rhabdomyosarcoma using both existing literature as well as recommendations from the relevant cooperative group clinical trials. Overall, parameningeal rhabdomyosarcoma represents a distinctly challenging disease to treat, given its location near many critical structures in the head and neck, frequently advanced local presentation, and predilection for local failure. Definitive chemoradiation remains the standard treatment approach for parameningeal rhabdomyosarcoma, with surgery often limited to biopsy or salvage therapy for recurrent disease. In this consensus paper, we specifically discuss consensus guidelines and evidence for definitive local management with radiotherapy, with a focus on imaging for radiotherapy planning, dose and timing of radiation, approach for nodal irradiation, various radiation techniques, including proton therapy, and the limited role of surgical resection.

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“…In the analysis of the BERNIE study, a cut off of day 221 was used to define radical radiotherapy to take this issue into account. 21 Cases with extensive metastatic disease may require metastatic radiotherapy delivered in sequential phases, treated in succession to limit the associated bone marrow and other acute toxicities. Delayed radiotherapy for metastatic sites is also recommended in COG studies, with metastatic site radiotherapy to now occur at approximately week 40 after completion of primary chemotherapy on ARST2031 (previously, on the prior high-risk study studies, ARST0431 and ARST08P1, there was the option to deliver metastatic site radiotherapy either with primary site radiation or after completion of primary chemotherapy).2.3 Definitive radiation dosing and volumes…”

Section: Evidencementioning

confidence: 99%

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

Casey

Mandeville

Bradley³

et al. 2022

Preprint

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The International Soft-Tissue Sarcoma Database Consortium (INSTRuCT) consists of a collaboration between the Children’s Oncology Group (COG) Soft Tissue Sarcoma Committee, the European pediatric Soft-Tissue Sarcoma Study Group (EpSSG), and the Cooperative Weichteilsarkom Studiengruppe (CWS). As part of the larger initiative of INSTRuCT to provide consensus expert opinions for clinical treatment of pediatric soft tissue sarcoma, we sought to provide updated, evidenced-based consensus guidelines for local treatment of parameningeal rhabdomyosarcoma using both existing literature as well as recommendations from the relevant cooperative group clinical trials. Overall, parameningeal rhabdomyosarcoma represents a distinctly challenging disease to treat given its location near many critical structures in the head and neck, frequently advanced local presentation, and predilection for local failure. Definitive chemoradiation remains the standard treatment approach for parameningeal rhabdomyosarcoma, with surgery often limited to biopsy or salvage therapy for recurrent disease. In this consensus paper, we specifically discuss consensus guidelines and evidence for definitive local management with radiotherapy, with a focus on imaging for radiotherapy planning, dose and timing of radiation, approach for nodal irradiation, various radiation techniques including proton therapy, and the limited role of surgical resection.

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The Impact of Radiation Therapy in Children and Adolescents With Metastatic Rhabdomyosarcoma

Cited by 17 publications

References 14 publications

Metastatic rhabdomyosarcoma: Evidence of the impact of radiotherapy on survival. A retrospective single‐center experience

Metastatic rhabdomyosarcoma: Evidence of the impact of radiotherapy on survival. A retrospective single‐center experience

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

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