Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival

Isabelle S A de Vries; Carlo Morosi; Gianni Bisogno; Veronique Minard-Colin; Beatrice Coppadoro; Ilaria Zanetti; Andrea Ferrari; Daniel Orbach; Salma Moalla; Myriam Ben-Arush; Christine Devalck; Roelof van Ewijk; Kieran McHugh; Meriel Jenney; Julia Chisholm; Gabriela Guillen; Henry Mandeville; Johannes H M Merks; Rick R Van Rijn

doi:10.1007/s00247-025-06359-3

Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival

Isabelle S A de Vries, Carlo Morosi, Gianni Bisogno, Veronique Minard-Colin, Beatrice Coppadoro, Ilaria Zanetti, Andrea Ferrari, Daniel Orbach, Salma Moalla, Myriam Ben-Arush, Christine Devalck, Roelof van Ewijk, Kieran McHugh, Meriel Jenney, Julia Chisholm, Gabriela Guillen, Henry Mandeville, Johannes H M Merks, Rick R Van Rijn

Onderzoeksoutput: Bijdrage aan tijdschrift › Artikel › peer review

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BACKGROUND: There is an urgent need for early radiological markers predicting survival in paediatric rhabdomyosarcoma patients.

OBJECTIVE: To analyse the predictive value of early radiological tumour volume response to chemotherapy in non-metastatic rhabdomyosarcoma patients.

METHODS: The European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study prospectively included non-metastatic rhabdomyosarcoma patients. Patients with Intergroup Rhabdomyosarcoma Studies (IRS) postsurgical Group III (i.e. measurable disease) at diagnosis, with three tumour diameters available at diagnosis and following three cycles of chemotherapy, were included. Tumour response was categorized as complete response, partial response, minor partial response, stable disease, or progressive disease. Patients with progressive disease were excluded from analyses. Failure-free survival and overall survival were estimated with Kaplan-Meier using a landmark analysis. Prognostic factors were assessed using Cox proportional hazards analyses.

RESULTS: Of 1,674 patients aged < 18 years at diagnosis included in the EpSSG RMS 2005 study, 613 patients (median age 4.2 years, interquartile range 2.2-8.0, 346 males) were eligible for analysis. After three cycles of chemotherapy, 48 patients reached complete response, 371 partial response, 139 minor partial response, 48 stable disease, and seven patients progressive disease. Univariate Cox proportional hazards model showed no significant difference between response groups for failure-free survival or overall survival. In multivariate Cox regression analysis, tumour volume response was not an independent prognostic factor for failure-free survival or overall survival.

CONCLUSION: Early radiological tumour volume response after the third cycle of neoadjuvant chemotherapy, with exclusion of progressive disease, does not predict survival in paediatric rhabdomyosarcoma patients.

Originele taal-2	Engels
Tijdschrift	Pediatric Radiology
Vroegere onlinedatum	14 aug. 2025
DOI's	https://doi.org/10.1007/s00247-025-06359-3
Status	Gepubliceerd - 2025
Extern gepubliceerd	Ja

Trefwoorden

Paediatric oncology
Radiological response assessment
Rhabdomyosarcoma
Tumour volume

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10.1007/s00247-025-06359-3

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de Vries, I. S. A., Morosi, C., Bisogno, G., Minard-Colin, V., Coppadoro, B., Zanetti, I., Ferrari, A., Orbach, D., Moalla, S., Ben-Arush, M., Devalck, C., van Ewijk, R., McHugh, K., Jenney, M., Chisholm, J., Guillen, G., Mandeville, H., Merks, J. H. M., & Van Rijn, R. R. (2025). Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival. Pediatric Radiology. https://doi.org/10.1007/s00247-025-06359-3

@article{9738319d33244dfcb6e88813d22625cb,

title = "Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival",

abstract = "BACKGROUND: There is an urgent need for early radiological markers predicting survival in paediatric rhabdomyosarcoma patients.OBJECTIVE: To analyse the predictive value of early radiological tumour volume response to chemotherapy in non-metastatic rhabdomyosarcoma patients.METHODS: The European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study prospectively included non-metastatic rhabdomyosarcoma patients. Patients with Intergroup Rhabdomyosarcoma Studies (IRS) postsurgical Group III (i.e. measurable disease) at diagnosis, with three tumour diameters available at diagnosis and following three cycles of chemotherapy, were included. Tumour response was categorized as complete response, partial response, minor partial response, stable disease, or progressive disease. Patients with progressive disease were excluded from analyses. Failure-free survival and overall survival were estimated with Kaplan-Meier using a landmark analysis. Prognostic factors were assessed using Cox proportional hazards analyses.RESULTS: Of 1,674 patients aged < 18 years at diagnosis included in the EpSSG RMS 2005 study, 613 patients (median age 4.2 years, interquartile range 2.2-8.0, 346 males) were eligible for analysis. After three cycles of chemotherapy, 48 patients reached complete response, 371 partial response, 139 minor partial response, 48 stable disease, and seven patients progressive disease. Univariate Cox proportional hazards model showed no significant difference between response groups for failure-free survival or overall survival. In multivariate Cox regression analysis, tumour volume response was not an independent prognostic factor for failure-free survival or overall survival.CONCLUSION: Early radiological tumour volume response after the third cycle of neoadjuvant chemotherapy, with exclusion of progressive disease, does not predict survival in paediatric rhabdomyosarcoma patients.",

keywords = "Paediatric oncology, Radiological response assessment, Rhabdomyosarcoma, Tumour volume",

author = "\{de Vries\}, \{Isabelle S A\} and Carlo Morosi and Gianni Bisogno and Veronique Minard-Colin and Beatrice Coppadoro and Ilaria Zanetti and Andrea Ferrari and Daniel Orbach and Salma Moalla and Myriam Ben-Arush and Christine Devalck and \{van Ewijk\}, Roelof and Kieran McHugh and Meriel Jenney and Julia Chisholm and Gabriela Guillen and Henry Mandeville and Merks, \{Johannes H M\} and \{Van Rijn\}, \{Rick R\}",

note = "{\textcopyright} 2025. The Author(s).",

year = "2025",

doi = "10.1007/s00247-025-06359-3",

language = "English",

journal = "Pediatric Radiology",

issn = "0301-0449",

publisher = "Springer Nature",

}

de Vries, ISA, Morosi, C, Bisogno, G, Minard-Colin, V, Coppadoro, B, Zanetti, I, Ferrari, A, Orbach, D, Moalla, S, Ben-Arush, M, Devalck, C, van Ewijk, R, McHugh, K, Jenney, M, Chisholm, J, Guillen, G, Mandeville, H, Merks, JHM & Van Rijn, RR 2025, 'Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival', Pediatric Radiology. https://doi.org/10.1007/s00247-025-06359-3

TY - JOUR

T1 - Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival

AU - de Vries, Isabelle S A

AU - Morosi, Carlo

AU - Bisogno, Gianni

AU - Minard-Colin, Veronique

AU - Coppadoro, Beatrice

AU - Zanetti, Ilaria

AU - Ferrari, Andrea

AU - Orbach, Daniel

AU - Moalla, Salma

AU - Ben-Arush, Myriam

AU - Devalck, Christine

AU - van Ewijk, Roelof

AU - McHugh, Kieran

AU - Jenney, Meriel

AU - Chisholm, Julia

AU - Guillen, Gabriela

AU - Mandeville, Henry

AU - Merks, Johannes H M

AU - Van Rijn, Rick R

PY - 2025

Y1 - 2025

N2 - BACKGROUND: There is an urgent need for early radiological markers predicting survival in paediatric rhabdomyosarcoma patients.OBJECTIVE: To analyse the predictive value of early radiological tumour volume response to chemotherapy in non-metastatic rhabdomyosarcoma patients.METHODS: The European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study prospectively included non-metastatic rhabdomyosarcoma patients. Patients with Intergroup Rhabdomyosarcoma Studies (IRS) postsurgical Group III (i.e. measurable disease) at diagnosis, with three tumour diameters available at diagnosis and following three cycles of chemotherapy, were included. Tumour response was categorized as complete response, partial response, minor partial response, stable disease, or progressive disease. Patients with progressive disease were excluded from analyses. Failure-free survival and overall survival were estimated with Kaplan-Meier using a landmark analysis. Prognostic factors were assessed using Cox proportional hazards analyses.RESULTS: Of 1,674 patients aged < 18 years at diagnosis included in the EpSSG RMS 2005 study, 613 patients (median age 4.2 years, interquartile range 2.2-8.0, 346 males) were eligible for analysis. After three cycles of chemotherapy, 48 patients reached complete response, 371 partial response, 139 minor partial response, 48 stable disease, and seven patients progressive disease. Univariate Cox proportional hazards model showed no significant difference between response groups for failure-free survival or overall survival. In multivariate Cox regression analysis, tumour volume response was not an independent prognostic factor for failure-free survival or overall survival.CONCLUSION: Early radiological tumour volume response after the third cycle of neoadjuvant chemotherapy, with exclusion of progressive disease, does not predict survival in paediatric rhabdomyosarcoma patients.

AB - BACKGROUND: There is an urgent need for early radiological markers predicting survival in paediatric rhabdomyosarcoma patients.OBJECTIVE: To analyse the predictive value of early radiological tumour volume response to chemotherapy in non-metastatic rhabdomyosarcoma patients.METHODS: The European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study prospectively included non-metastatic rhabdomyosarcoma patients. Patients with Intergroup Rhabdomyosarcoma Studies (IRS) postsurgical Group III (i.e. measurable disease) at diagnosis, with three tumour diameters available at diagnosis and following three cycles of chemotherapy, were included. Tumour response was categorized as complete response, partial response, minor partial response, stable disease, or progressive disease. Patients with progressive disease were excluded from analyses. Failure-free survival and overall survival were estimated with Kaplan-Meier using a landmark analysis. Prognostic factors were assessed using Cox proportional hazards analyses.RESULTS: Of 1,674 patients aged < 18 years at diagnosis included in the EpSSG RMS 2005 study, 613 patients (median age 4.2 years, interquartile range 2.2-8.0, 346 males) were eligible for analysis. After three cycles of chemotherapy, 48 patients reached complete response, 371 partial response, 139 minor partial response, 48 stable disease, and seven patients progressive disease. Univariate Cox proportional hazards model showed no significant difference between response groups for failure-free survival or overall survival. In multivariate Cox regression analysis, tumour volume response was not an independent prognostic factor for failure-free survival or overall survival.CONCLUSION: Early radiological tumour volume response after the third cycle of neoadjuvant chemotherapy, with exclusion of progressive disease, does not predict survival in paediatric rhabdomyosarcoma patients.

KW - Paediatric oncology

KW - Radiological response assessment

KW - Rhabdomyosarcoma

KW - Tumour volume

UR - https://www.mendeley.com/catalogue/5095061b-7023-3075-a145-fb2fcd1789c6/

U2 - 10.1007/s00247-025-06359-3

DO - 10.1007/s00247-025-06359-3

M3 - Article

C2 - 40810863

SN - 0301-0449

JO - Pediatric Radiology

JF - Pediatric Radiology

ER -

Early radiologic tumour volume response in non-metastatic rhabdomyosarcoma is not predictive for survival

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