Dyda Dao1, Anna J Najor1,2, Philip Y Sun3, Forough Farrokhyar4,5, Christopher R Moir6, Michael B Ishitani7. 1. Mayo Clinic Alix School of Medicine, Mayo Clinic, Rochester, MN, USA. 2. Johns Hopkins Bloomberg School of Public Health, Baltimore, MD, USA. 3. Department of Neurology, Los Angeles County + USC Medical Center, Los Angeles, CA, USA. 4. Department of Surgery, McMaster University, Hamilton, ON, Canada. 5. Department of Health Research Methods, Evidence, and Impact, McMaster University, Hamilton, ON, Canada. 6. Division of Pediatric Surgery, Mayo Clinic's Children Center, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA. 7. Division of Pediatric Surgery, Mayo Clinic's Children Center, Mayo Clinic, 200 1st St SW, Rochester, MN, 55902, USA. Ishitani.Michael@mayo.edu.
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.
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.
Authors: Nguyen S Pham; Brian Poirier; Scott C Fuller; Arthur B Dublin; Travis T Tollefson Journal: Int J Pediatr Otorhinolaryngol Date: 2010-05-15 Impact factor: 1.675
Authors: Allison L Speer; Deborah E Schofield; Kasper S Wang; Cathy E Shin; James E Stein; Donald B Shaul; G Hossein Mahour; Henri R Ford Journal: J Pediatr Surg Date: 2008-07 Impact factor: 2.545
Authors: Sara C Fallon; Mary L Brandt; J Ruben Rodriguez; Sanjeev A Vasudevan; Monica E Lopez; M John Hicks; Eugene S Kim Journal: J Surg Res Date: 2013-05-25 Impact factor: 2.192
Authors: Karen Fritchie; Lu Wang; Zhaohong Yin; Joy Nakitandwe; Dale Hedges; Andrew Horvai; Jorge Torres Mora; Andrew L Folpe; Armita Bahrami Journal: Mod Pathol Date: 2020-10-23 Impact factor: 7.842