Audra J. Reiter, Lynn Huang, Jennifer H. Aldrink, Brian T. Craig, Andrew M. Davidoff, Lindsay J. Talbot, Jordan Coggins, Jasmine Smith, Katherine C. Bergus, Taleen A. MacArthur, Stephanie F. Polites, Roshni Dasgupta, Chloe Boehmer, Joseph Brungardt, Marcus M. Malek, Hannah N. Rinehardt, Zachary J. Kastenberg, Cameron M. Arkin, Antoine Gourmel, Nelson Piche, Timothy B. Lautz
Background
Relapse occurs in 30–40 % of patients with localized Ewing sarcoma (EWS). Our objective was to describe the current management and outcomes of patients with initially localized EWS who experience first pulmonary relapse.
Methods
This multi-center retrospective cohort study included patients ≤22 years old with initially localized EWS treated from 2007 to 2020 at 19 Pediatric Surgical Oncology Research Collaborative institutions, who developed pulmonary relapse. Kaplan-Meier analysis was performed.
Results
Thirty-three patients with initially localized EWS developed pulmonary relapse at a median age of 17 (IQR 14; 20) years. Eleven (33 %) patients also had extra-pulmonary metastases (EPM) at relapse. Among the 22 (67 %) patients with pulmonary-only relapse, 10 (45 %) had solitary pulmonary nodules. Pulmonary metastasectomy was performed in 8/10 (80 %) patients with solitary pulmonary-only metastases, 5/12 (42 %) patients with multiple pulmonary-only metastases, and 2/11 (18 %) patients who also had EPM. Whole lung irradiation was administered in 7/10 (70 %) with solitary pulmonary-only metastases, 7/12 (58 %) with multiple pulmonary-only metastases, and 2/11 (18 %) with EPM. Rates of further pulmonary relapse/progression were similar between groups (p = 0.97). In Kaplan-Meier analysis, 3-year overall survival was 73 % with solitary pulmonary-only metastases, 40 % with multiple pulmonary-only metastases, and 23 % with EPM (p = 0.097).
Conclusions
While survival for patients with relapsed EWS is poor, the subset of patients with solitary relapse confined to the lung are often good candidates for pulmonary metastasectomy and have a non-statistically significant trend towards improved survival outcomes.
Intended audience: Healthcare professionals and clinicians.
Hi, I'm Sophia Schermerhorn, a research fellow at Cincinnati Children's, and here's an article that you should know about. In 2024, Writer etal published a multi-center retrospective study in the Journal of Pediatric Surgery through PeaceSOC, the Pediatric Surgical Oncology Research Collaborative, a network of over 50 centers in North America that work together to improve outcomes in pediatric surgical oncology. This study looked at patients less than 22 years old with initially localized Ewing sarcoma who. Developed the first pulmonary relapse between 2007 and 2020 across 19 different centers. Here's what they found. Among 33 patients, about 2/3 had relapse limited to the lungs, and nearly half of those patients had just a solitary pulmonary nodule. In the group with the solitary pulmonary nodule, these patients were more likely to undergo metacystectomy and whole lung radiation, and had the highest overall three-year survival at 73%. Compared to 40% for those with multiple nodules and just 23% for those with extra pulmonary disease. Why does this matter? This pattern suggests that solitary pulmonary relapse may represent a biologically more favorable type of recurrence. One that's more amenable to achieving meaningful local control, with metacystectomy playing an important role as part of multimodal therapy. So with that in mind, would you offer a metacystectomy for a solitary Ewing relapse at your institution?
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