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Multicenter Study
. 2024 Jul;71(7):e31026.
doi: 10.1002/pbc.31026. Epub 2024 Apr 28.

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

Audra J Reiter  1   2 , Lynn Huang  2 , Brian T Craig  3 , Andrew M Davidoff  4 , Lindsay J Talbot  4 , Jordan Coggins  4 , Jasmine Smith  4 , Jennifer H Aldrink  5 , Katherine C Bergus  5 , Taleen A MacArthur  6 , Stephanie F Polites  6 , Chloe Boehmer  7 , Joseph Brungardt  7 , Marcus M Malek  8 , Hannah N Rinehardt  8 , Zachary J Kastenberg  9 , Cameron M Arkin  9 , Antoine Gourmel  10 , Nelson Piche  10 , Marshall Wallace  11 , Jiancong Liang  11 , Harold N Lovvorn 3rd  11 , Robin T Petroze  12 , Gwendolyn Gillies  12 , John P Marquart  3 , Kerri Becktell  3 , Hau D Le  13 , Juan Favela  13 , Barrie S Rich  14 , Richard D Glick  14 , Natashia M Seemann  15 , Jacob Davidson  15 , Claire A Wilson  15 , Jonathan Roach  16 , Erin G Brown  17 , Kathleen E Doyle  17 , Brian A Coakley  18 , Pamela Emengo  18 , Pamela Merola  19 , Christa N Grant  20 , Anuritha Tirumani  20 , Elisabeth T Tracy  21 , Mary E Moya-Mendez  21 , Roshni Dasgupta  7 , Timothy B Lautz  1 ; Pediatric Surgical Oncology Research Collaborative
Affiliations
Multicenter Study

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

Audra J Reiter et al. Pediatr Blood Cancer. 2024 Jul.

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.

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Conflict of interest statement

Disclosures/Conflicts of Interest: The authors have no related conflicts of interest to declare.

Figures

FIGURE 1
FIGURE 1
Kaplan-Meier curves for overall survival in (A) children who achieved rapid complete response (RCR) versus those who did not and (B) patients with and without RCR by whole lung irradiation (WLI)
FIGURE 2
FIGURE 2
Kaplan-Meier curves for (A) overall survival in children who had attempted complete surgical clearance of pulmonary disease (B) pulmonary relapse in children who had attempted complete surgical clearance of pulmonary disease
FIGURE 3
FIGURE 3
Kaplan-Meier curves for pulmonary relapse in (A) children who achieved rapid complete response (RCR) versus those who did not and (B) patients with and without RCR by whole lung irradiation (WLI)
See this image and copyright information in PMC

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