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Ewing sarcoma among children 5 years of age or younger: Is it a different disease?
Chitrakshi Nagpal1, Shuvadeep Ganguly2, Archana Sasi1
1Department of Medical Oncology, Dr. B.R.A. IRCH, All India Institute of Medical Sciences, New Delhi, India.
Insights
Young children with Ewing sarcoma (ES) present with a distinct, favorable clinical profile. However, age is not an independent prognostic factor for survival outcomes in pediatric ES.
Area of Science:
- Pediatric Oncology
- Sarcoma Research
- Clinical Epidemiology
Background:
- Ewing sarcoma (ES) in children aged ≤5 years may exhibit unique biological characteristics.
- Limited data exists on the specific clinical features and outcomes of this younger demographic.
- This study addresses the scarcity of information regarding ES in very young children.
Purpose of the Study:
- To evaluate the clinical presentation, treatment, and survival outcomes of ES in children aged ≤5 years.
- To identify prognostic factors influencing outcomes in this age group.
- To compare the characteristics and outcomes of younger children with older children diagnosed with ES.
Main Methods:
- Retrospective analysis of children with ES diagnosed between 2003 and 2019.
- Multivariable Cox regression was used to identify prognostic factors.
- Propensity score-matched (PSM) analysis compared outcomes between age groups, including metastatic and localized disease subgroups.
Main Results:
- Out of 859 patients, 86 (10%) were ≤5 years old, with a median age of 4 years.
- Younger children presented with more head and neck/thorax primaries and fewer pelvic tumors.
- Metastatic disease and symptom duration ≤3 months were associated with inferior overall survival (OS) and event-free survival (EFS).
- PSM analysis indicated age was not a significant independent prognostic factor for OS in either metastatic or localized disease.
Conclusions:
- Children with ES aged ≤5 years demonstrate a distinct and more favorable clinical presentation.
- Despite favorable presentation, age itself is not an independent predictor of survival when other factors are considered.
- Findings suggest a need for tailored analysis of ES in very young children.
Introduction:
Children ≤5 years of age with Ewing's sarcoma (ES) possibly have a distinct disease biology, data on which are scarce. We evaluated clinical features, outcomes, and prognostic factors of ES among children with age ≤5 years.
Methods:
Children with ES registered between 2003 and 2019 were included. Baseline clinical and treatment details were retrieved from medical records. Prognostic factors were identified using multivariable Cox regression. Clinical features and outcomes of children ≤5 years were compared with those greater than 5 years by chi-square and log-rank tests. Propensity score-matched (PSM) analysis was done to evaluate the impact of age on survival in the metastatic and localized subgroups.
Results:
Out of the 859 patients, 86 (10%) were ≤5 years of age (median age 4 years, 60 males [69.8%]). The most common location was the extremities (37.2%), followed by thorax (27.9%) and head and neck (H&N) (22.1%); baseline metastases were seen in 25 patients (29.8%). The median event-free-survival (EFS) and overall survival (OS) were 25.6 and 68.7 months, respectively. Metastatic disease predicted inferior OS (hazard ratio [HR] = 2.54, p = .018) and EFS (HR = 2.47, p = .007], symptom duration ≤3 months predicted an inferior OS (HR = 2.17, p = .048). Compared to age greater than 5 years, younger children had more H&N and less pelvic primaries (p < .001) and lesser baseline metastases (p = .037). PSM analysis did not reveal any significant impact of age on OS in the metastatic (HR = 1.59, p = .29) or localized cohort (HR = 1.77, p = .09).
Conclusions:
Children with ES ≤5 years of age have a distinct favorable clinical presentation. However, age is not an independent prognostic factor for survival outcomes when adjusted for confounders.
