Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases

Audra J. Reiter; Lynn Huang; Brian T. Craig; Andrew M. Davidoff; Lindsay J. Talbot; Jordan Coggins; Jasmine Smith; Jennifer H. Aldrink; Katherine C. Bergus; Taleen A. MacArthur; Stephanie F. Polites; Chloe Boehmer; Joseph Brungardt; Marcus M. Malek; Hannah N. Rinehardt; Zachary J. Kastenberg; Cameron M. Arkin; Antoine Gourmel; Nelson Piche; Marshall Wallace; Jiancong Liang; Harold N. Lovvorn; Robin T. Petroze; Gwendolyn Gillies; John P. Marquart; Kerri Becktell; Hau D. Le; Juan Favela; Barrie S. Rich; Richard D. Glick; Natashia M. Seemann; Jacob Davidson; Claire A. Wilson; Jonathan Roach; Erin G. Brown; Kathleen E. Doyle; Brian A. Coakley; Pamela Emengo; Pamela Merola; Christa N. Grant; Anuritha Tirumani; Elisabeth T. Tracy; Mary E. Moya-Mendez; Roshni Dasgupta; Timothy B. Lautz

doi:10.1002/pbc.31026

Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases

Audra J. Reiter, Lynn Huang, Brian T. Craig, Andrew M. Davidoff, Lindsay J. Talbot, Jordan Coggins, Jasmine Smith, Jennifer H. Aldrink, Katherine C. Bergus, Taleen A. MacArthur, Stephanie F. Polites, Chloe Boehmer, Joseph Brungardt, Marcus M. Malek, Hannah N. Rinehardt, Zachary J. Kastenberg, Cameron M. Arkin, Antoine Gourmel, Nelson Piche, Marshall WallaceJiancong Liang, Harold N. Lovvorn, Robin T. Petroze, Gwendolyn Gillies, John P. Marquart, Kerri Becktell, Hau D. Le, Juan Favela, Barrie S. Rich, Richard D. Glick, Natashia M. Seemann, Jacob Davidson, Claire A. Wilson, Jonathan Roach, Erin G. Brown, Kathleen E. Doyle, Brian A. Coakley, Pamela Emengo, Pamela Merola, Christa N. Grant, Anuritha Tirumani, Elisabeth T. Tracy, Mary E. Moya-Mendez, Roshni Dasgupta, Timothy B. Lautz

Research output: Contribution to journal › Article › peer-review

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Abstract

Purpose: Our objectives were to compare overall survival (OS) and pulmonary relapse between patients with metastatic Ewing sarcoma (EWS) at diagnosis who achieve rapid complete response (RCR) and those with residual pulmonary nodules after induction chemotherapy (non-RCR). Patients and methods: This retrospective cohort study included children under 20 years with metastatic EWS treated from 2007 to 2020 at 19 institutions in the Pediatric Surgical Oncology Research Collaborative. Chi-square tests were conducted for differences among groups. Kaplan–Meier curves were generated for OS and pulmonary relapse. Results: Among 148 patients with metastatic EWS at diagnosis, 61 (41.2%) achieved RCR. Five-year OS was 71.2% for patients who achieved RCR, and 50.2% for those without RCR (p =.04), and in multivariable regression among patients with isolated pulmonary metastases, RCR (hazards ratio [HR] 0.42; 95% confidence interval [CI]: 0.17–0.99) and whole lung irradiation (WLI) (HR 0.35; 95% CI: 0.16–0.77) were associated with improved survival. Pulmonary relapse occurred in 57 (37%) patients, including 18 (29%) in the RCR and 36 (41%) in the non-RCR groups (p =.14). Five-year pulmonary relapse rates did not significantly differ based on RCR (33.0%) versus non-RCR (47.0%, p =.13), or WLI (38.8%) versus no WLI (46.0%, p =.32). Discussion: Patients with EWS who had isolated pulmonary metastases at diagnosis had improved OS if they achieved RCR and received WLI, despite having no significant differences in rates of pulmonary relapse.

Original language	English
Article number	e31026
Journal	Pediatric Blood and Cancer
Volume	71
Issue number	7
DOIs	https://doi.org/10.1002/pbc.31026
State	Published - Jul 2024

Keywords

Ewing sarcoma
pediatric surgery
pulmonary metastasectomy

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10.1002/pbc.31026

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Reiter, A. J., Huang, L., Craig, B. T., Davidoff, A. M., Talbot, L. J., Coggins, J., Smith, J., Aldrink, J. H., Bergus, K. C., MacArthur, T. A., Polites, S. F., Boehmer, C., Brungardt, J., Malek, M. M., Rinehardt, H. N., Kastenberg, Z. J., Arkin, C. M., Gourmel, A., Piche, N., ... Lautz, T. B. (2024). Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases. Pediatric Blood and Cancer, 71(7), Article e31026. https://doi.org/10.1002/pbc.31026

@article{3bad37c88bb7475190165289dc63a619,

title = "Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases",

abstract = "Purpose: Our objectives were to compare overall survival (OS) and pulmonary relapse between patients with metastatic Ewing sarcoma (EWS) at diagnosis who achieve rapid complete response (RCR) and those with residual pulmonary nodules after induction chemotherapy (non-RCR). Patients and methods: This retrospective cohort study included children under 20 years with metastatic EWS treated from 2007 to 2020 at 19 institutions in the Pediatric Surgical Oncology Research Collaborative. Chi-square tests were conducted for differences among groups. Kaplan–Meier curves were generated for OS and pulmonary relapse. Results: Among 148 patients with metastatic EWS at diagnosis, 61 (41.2%) achieved RCR. Five-year OS was 71.2% for patients who achieved RCR, and 50.2% for those without RCR (p =.04), and in multivariable regression among patients with isolated pulmonary metastases, RCR (hazards ratio [HR] 0.42; 95% confidence interval [CI]: 0.17–0.99) and whole lung irradiation (WLI) (HR 0.35; 95% CI: 0.16–0.77) were associated with improved survival. Pulmonary relapse occurred in 57 (37%) patients, including 18 (29%) in the RCR and 36 (41%) in the non-RCR groups (p =.14). Five-year pulmonary relapse rates did not significantly differ based on RCR (33.0%) versus non-RCR (47.0%, p =.13), or WLI (38.8%) versus no WLI (46.0%, p =.32). Discussion: Patients with EWS who had isolated pulmonary metastases at diagnosis had improved OS if they achieved RCR and received WLI, despite having no significant differences in rates of pulmonary relapse.",

keywords = "Ewing sarcoma, pediatric surgery, pulmonary metastasectomy",

author = "Reiter, {Audra J.} and Lynn Huang and Craig, {Brian T.} and Davidoff, {Andrew M.} and Talbot, {Lindsay J.} and Jordan Coggins and Jasmine Smith and Aldrink, {Jennifer H.} and Bergus, {Katherine C.} and MacArthur, {Taleen A.} and Polites, {Stephanie F.} and Chloe Boehmer and Joseph Brungardt and Malek, {Marcus M.} and Rinehardt, {Hannah N.} and Kastenberg, {Zachary J.} and Arkin, {Cameron M.} and Antoine Gourmel and Nelson Piche and Marshall Wallace and Jiancong Liang and Lovvorn, {Harold N.} and Petroze, {Robin T.} and Gwendolyn Gillies and Marquart, {John P.} and Kerri Becktell and Le, {Hau D.} and Juan Favela and Rich, {Barrie S.} and Glick, {Richard D.} and Seemann, {Natashia M.} and Jacob Davidson and Wilson, {Claire A.} and Jonathan Roach and Brown, {Erin G.} and Doyle, {Kathleen E.} and Coakley, {Brian A.} and Pamela Emengo and Pamela Merola and Grant, {Christa N.} and Anuritha Tirumani and Tracy, {Elisabeth T.} and Moya-Mendez, {Mary E.} and Roshni Dasgupta and Lautz, {Timothy B.}",

note = "Publisher Copyright: {\textcopyright} 2024 Wiley Periodicals LLC.",

year = "2024",

month = jul,

doi = "10.1002/pbc.31026",

language = "English",

volume = "71",

journal = "Pediatric Blood and Cancer",

issn = "1545-5009",

publisher = "Wiley-Liss Inc.",

number = "7",

}

Reiter, AJ, Huang, L, Craig, BT, Davidoff, AM, Talbot, LJ, Coggins, J, Smith, J, Aldrink, JH, Bergus, KC, MacArthur, TA, Polites, SF, Boehmer, C, Brungardt, J, Malek, MM, Rinehardt, HN, Kastenberg, ZJ, Arkin, CM, Gourmel, A, Piche, N, Wallace, M, Liang, J, Lovvorn, HN, Petroze, RT, Gillies, G, Marquart, JP, Becktell, K, Le, HD, Favela, J, Rich, BS, Glick, RD, Seemann, NM, Davidson, J, Wilson, CA, Roach, J, Brown, EG, Doyle, KE, Coakley, BA, Emengo, P, Merola, P, Grant, CN, Tirumani, A, Tracy, ET, Moya-Mendez, ME, Dasgupta, R & Lautz, TB 2024, 'Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases', Pediatric Blood and Cancer, vol. 71, no. 7, e31026. https://doi.org/10.1002/pbc.31026

TY - JOUR

T1 - Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases

AU - Reiter, Audra J.

AU - Huang, Lynn

AU - Craig, Brian T.

AU - Davidoff, Andrew M.

AU - Talbot, Lindsay J.

AU - Coggins, Jordan

AU - Smith, Jasmine

AU - Aldrink, Jennifer H.

AU - Bergus, Katherine C.

AU - MacArthur, Taleen A.

AU - Polites, Stephanie F.

AU - Boehmer, Chloe

AU - Brungardt, Joseph

AU - Malek, Marcus M.

AU - Rinehardt, Hannah N.

AU - Kastenberg, Zachary J.

AU - Arkin, Cameron M.

AU - Gourmel, Antoine

AU - Piche, Nelson

AU - Wallace, Marshall

AU - Liang, Jiancong

AU - Lovvorn, Harold N.

AU - Petroze, Robin T.

AU - Gillies, Gwendolyn

AU - Marquart, John P.

AU - Becktell, Kerri

AU - Le, Hau D.

AU - Favela, Juan

AU - Rich, Barrie S.

AU - Glick, Richard D.

AU - Seemann, Natashia M.

AU - Davidson, Jacob

AU - Wilson, Claire A.

AU - Roach, Jonathan

AU - Brown, Erin G.

AU - Doyle, Kathleen E.

AU - Coakley, Brian A.

AU - Emengo, Pamela

AU - Merola, Pamela

AU - Grant, Christa N.

AU - Tirumani, Anuritha

AU - Tracy, Elisabeth T.

AU - Moya-Mendez, Mary E.

AU - Dasgupta, Roshni

AU - Lautz, Timothy B.

PY - 2024/7

Y1 - 2024/7

N2 - Purpose: Our objectives were to compare overall survival (OS) and pulmonary relapse between patients with metastatic Ewing sarcoma (EWS) at diagnosis who achieve rapid complete response (RCR) and those with residual pulmonary nodules after induction chemotherapy (non-RCR). Patients and methods: This retrospective cohort study included children under 20 years with metastatic EWS treated from 2007 to 2020 at 19 institutions in the Pediatric Surgical Oncology Research Collaborative. Chi-square tests were conducted for differences among groups. Kaplan–Meier curves were generated for OS and pulmonary relapse. Results: Among 148 patients with metastatic EWS at diagnosis, 61 (41.2%) achieved RCR. Five-year OS was 71.2% for patients who achieved RCR, and 50.2% for those without RCR (p =.04), and in multivariable regression among patients with isolated pulmonary metastases, RCR (hazards ratio [HR] 0.42; 95% confidence interval [CI]: 0.17–0.99) and whole lung irradiation (WLI) (HR 0.35; 95% CI: 0.16–0.77) were associated with improved survival. Pulmonary relapse occurred in 57 (37%) patients, including 18 (29%) in the RCR and 36 (41%) in the non-RCR groups (p =.14). Five-year pulmonary relapse rates did not significantly differ based on RCR (33.0%) versus non-RCR (47.0%, p =.13), or WLI (38.8%) versus no WLI (46.0%, p =.32). Discussion: Patients with EWS who had isolated pulmonary metastases at diagnosis had improved OS if they achieved RCR and received WLI, despite having no significant differences in rates of pulmonary relapse.

AB - Purpose: Our objectives were to compare overall survival (OS) and pulmonary relapse between patients with metastatic Ewing sarcoma (EWS) at diagnosis who achieve rapid complete response (RCR) and those with residual pulmonary nodules after induction chemotherapy (non-RCR). Patients and methods: This retrospective cohort study included children under 20 years with metastatic EWS treated from 2007 to 2020 at 19 institutions in the Pediatric Surgical Oncology Research Collaborative. Chi-square tests were conducted for differences among groups. Kaplan–Meier curves were generated for OS and pulmonary relapse. Results: Among 148 patients with metastatic EWS at diagnosis, 61 (41.2%) achieved RCR. Five-year OS was 71.2% for patients who achieved RCR, and 50.2% for those without RCR (p =.04), and in multivariable regression among patients with isolated pulmonary metastases, RCR (hazards ratio [HR] 0.42; 95% confidence interval [CI]: 0.17–0.99) and whole lung irradiation (WLI) (HR 0.35; 95% CI: 0.16–0.77) were associated with improved survival. Pulmonary relapse occurred in 57 (37%) patients, including 18 (29%) in the RCR and 36 (41%) in the non-RCR groups (p =.14). Five-year pulmonary relapse rates did not significantly differ based on RCR (33.0%) versus non-RCR (47.0%, p =.13), or WLI (38.8%) versus no WLI (46.0%, p =.32). Discussion: Patients with EWS who had isolated pulmonary metastases at diagnosis had improved OS if they achieved RCR and received WLI, despite having no significant differences in rates of pulmonary relapse.

KW - Ewing sarcoma

KW - pediatric surgery

KW - pulmonary metastasectomy

UR - http://www.scopus.com/inward/record.url?scp=85191778083&partnerID=8YFLogxK

U2 - 10.1002/pbc.31026

DO - 10.1002/pbc.31026

M3 - Article

C2 - 38679864

AN - SCOPUS:85191778083

SN - 1545-5009

VL - 71

JO - Pediatric Blood and Cancer

JF - Pediatric Blood and Cancer

IS - 7

M1 - e31026

ER -

Survival outcomes in pediatric patients with metastatic Ewing sarcoma who achieve a rapid complete response of pulmonary metastases

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Keywords

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