Abstract
Purpose: Neuroblastoma is the most common extracranial solid tumor in children, and most patients are at high risk when they are initially diagnosed. The roles of surgery and induction chemotherapy in patients with high-risk neuroblastoma have been a subject of much controversy and debate. The objective of the current study was to assess the roles of surgery in high-risk neuroblastoma.Method: The review protocol was prospectively registered (PROSPEROID: CRD42021253961). The PubMed, Embase, Cochrane, and CNKI databases were searched from inception to January 2020 with no restrictions on language or publication date. Clinical studies comparing the outcomes of different surgical ranges for the treatment of high-risk neuroblastoma were analyzed. The Mantel–Haenszel method and a random effects model was utilized to calculate the hazard ratio (95% CI).Results: Fourteen studies that assessed 1,915 subjects met the full inclusion criteria. Compared with the gross tumor resection (GTR) group, complete tumor resection (CTR) did not significantly improve the 5-year EFS [p = 1.0; HR = 0.95 (95% CI, 0.87–1.05); I2 = 0%], and the 5-year OS [p = 0.76; HR = 1.08 (95% CI, 0.80–1.46); I2 = 0%] of patients. GTR or CTR resection had significantly better 5-year OS [p = 0.45; HR = 0.56 (95% CI, 0.43–0.72); I2 = 0%] and 5-year EFS [p = 0.15; HR = 0.80 (95% CI, 0.71–0.90); I2 = 31%] than subtotal tumor resection (STR) or biopsy only; however, both CTR or GTR showed a trend for more intra and post-operative complications compared with the STR or biopsy only [p = 0.37; OR = 1.54 (95% CI, 1.08–2.20); I2 = 0%]. The EFS of the patients who underwent GTR or CTR at the time of diagnosis and after induction chemotherapy were similar [p = 0.24; HR = 1.53 (95% CI, 0.84–2.77); I2 = 29%].Conclusion: For patients with high-risk neuroblastoma, complete tumor resection and gross tumor resection of the primary tumor were related to improved survival, with very limited effects on reducing intraoperative and postoperative complications. It is necessary to design strong chemotherapy regimens to improve the survival rate of advanced patients.Systematic Review Registration: https://www.crd.york.ac.uk/PROSPERO/, PROSPEROID [CRD42021253961].
Highlights
Neuroblastoma originating from the adrenal medulla or sympathetic ganglion is the most common extracranial solid tumor in children
Compared with the gross tumor resection (GTR) group, complete tumor resection (CTR) did not significantly improve the 5-year event-free survival (EFS) [p = 1.0; Hazard ratios (HRs) = 0.95; I2 = 0%], and the 5-year overall survival (OS) [p = 0.76; HR = 1.08; I2 = 0%] of patients
GTR or CTR resection had significantly better 5-year OS [p = 0.45; HR = 0.56; I2 = 0%] and 5-year EFS [p = 0.15; HR = 0.80; I2 = 31%] than subtotal tumor resection (STR) or biopsy only; both CTR or GTR showed a trend for more intra and post-operative complications compared with the STR or biopsy only [p = 0.37; Odds ratios (ORs) = 1.54; I2 = 0%]
Summary
Neuroblastoma originating from the adrenal medulla or sympathetic ganglion is the most common extracranial solid tumor in children. It is most common in the abdomen (75%), followed by the mediastinum (20%) and the neck (5%) (1). The INRG classification system assigns neuroblastoma patients to 1 of 16 pretreatment risk groups based on INRGSS, age, histologic category, grade of tumor differentiation, MYCN amplification, and 11q aberration. Combined studies indicate that long-term survival rates of children with high-risk neuroblastoma are currently ∼40–50% (3). The prognosis of children in the high-risk group is poor, and the long-term disease-free survival rate is
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