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Follow-up outcomes of pediatric patients who underwent surgical resection for lipoblastomas or lipoblastomatosis: a single-institution experience with a systematic review and meta-analysis

Abstract

Purpose

The objective of this study was to examine the long-term outcomes of pediatric patients who underwent surgical resection for lipoblastoma and lipoblastomatosis (LB/LBM).

Methods

A single-center retrospective study of pediatric patients with LB/LBMs seen between 1991 and 2015 was conducted. A systematic review, including studies published prior to late August 2018, was performed. Using a random effect meta-analysis, pooled weighted proportions and unadjusted odds ratios (OR) with 95% confidence intervals (CI) were calculated.

Results

The retrospective study included 16 patients, while the systematic review included 19 published studies consisting of 381 patients. Among 329 (82%) patients with follow-up information, the pooled recurrence rate was 16.8% (95% CI 10.9–23.5%; I2 = 59%). The reported time to recurrence ranged from < 1 to 8 years. Recurrence risk was greater for incomplete (n = 34) than complete resection (n = 150): OR 11.4 (95% CI 3.0–43.6; I2 = 43%). LBMs (n = 35) had a greater recurrence risk than LBs (n = 116): OR 5.5 (95% CI 1.9–15.9; I2 = 0%). Recurrences were higher for studies with approximately ≥ 3 years of follow-up versus studies with < 3 years of follow-up.

Conclusion

Recurrences are more likely to occur with LBMs and/or incomplete resection. Follow-up beyond 3–5 years should be considered given that the recurrence risk appears to be greater in the long-term.

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Acknowledgements

We would like to thank Cynthia J. Beeler, MLIS for her assistance with developing the electronic search strategy. We would also like to thank Dr. Suhail Zeineddin, MD for reviewing an early abstract of the manuscript.

Funding

None.

Author information

Idea/conceptualization: D.D., P.Y.S., M.B.I.; methodology: D.D., A.J.N., P.Y.S., M.B.I.; literature search: D.D.; data analysis: D.D., F.F.; writing—original draft preparation: D.D.; writing—review and editing (critical revision): D.D., A.J.N., F.F., P.Y.S., C.R.M., M.B.I.; resources: C.R.M., M.B.I.; supervision: M.B.I.

Correspondence to Michael B. Ishitani.

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Conflict of interest

The authors have no conflicts of interest, financial or otherwise, to disclose. No grant funding was used to support this research. The study was presented at the Minnesota Surgical Society 2019 Fall Conference (Minneapolis, MN, October 4, 2019) as an oral presentation.

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Dao, D., Najor, A.J., Sun, P.Y. et al. Follow-up outcomes of pediatric patients who underwent surgical resection for lipoblastomas or lipoblastomatosis: a single-institution experience with a systematic review and meta-analysis. Pediatr Surg Int (2020) doi:10.1007/s00383-019-04612-z

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Keywords

  • Lipoblastoma
  • Lipoblastomatosis
  • Recurrence
  • Pediatric surgery
  • Systematic review
  • Meta-analysis