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A Prospective Study of Expectant Observation as Primary Therapy for Neuroblastoma in Young Infants, a Children’s Oncology Group Study
Jed G. Nuchtern*1, Wendy B. London*2, Carol E. Barnewolt*2, Arlene Naranjo*3, James D. Geiger4, Susan L. Cohn*5, Robert C. Shamberger2
1Baylor College of Medicine, Houston, TX;2Harvard Medical School, Boston, MA;3University of Florida, Gainesville, FL;4University of Michigan, Ann Arbor, MI;5University of Chicago, Chicago, IL

OBJECTIVE(S): Neuroblastoma is the most common malignant tumor in infants. The objective of this study was to demonstrate that expectant observation of young infants with adrenal masses would result in excellent overall and event-free survival (OS and EFS) while avoiding surgical resection in the majority of cases.
METHODS: A prospective study of infants less than 6 months of age with small adrenal masses and no evidence of spread beyond the primary tumor was performed at all participating Children’s Oncology Group institutions. Parents could choose observation or immediate surgical resection. Serial abdominal sonograms and urinary VMA and HVA measurements were performed over a ninety-week interval. Infants experiencing a 50% increase in the volume of the mass or urine catecholamine values, or an increase in the HVA/VMA ratio >2 were referred for surgical resection.
RESULTS: 88 patients were enrolled, 84 elected observation and 4 chose immediate surgery. 16 observation patients ultimately had surgery; 9 had INSS stage 1 neuroblastoma, 2 had higher stage neuroblastoma (2B and 4S), 2 had low grade adrenocortical neoplasm, 2 had adrenal hemorrhage and one had extralobar sequestration. The two patients with adrenocortical tumors were resected for >50% increase in tumor volume. The 3-year EFS for a neuroblastoma event was 97.7±2.3%. The overall survival was 100% with median follow-up of 2.9 years. 81% of patients on the observation arm were spared resection.
CONCLUSIONS: Expectant observation of infants with small adrenal masses led to excellent EFS and OS while avoiding surgical intervention in a large majority of the patients.


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