Systematic Reviews
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Pediatr. Dec 9, 2023; 12(5): 350-358
Published online Dec 9, 2023. doi: 10.5409/wjcp.v12.i5.350
Warburg effect mimicking inborn errors of metabolism in childhood hematologic malignancies: A case-based systematic review
Khanittha Permtawee, Maliwan Tengsujaritkul, Chane Choed-Amphai, Supapitch Chanthong, Kanittha Mankhemthong, Lalita Sathitsamitphong, Rungrote Natesirinilkul, Pimlak Charoenkwan
Khanittha Permtawee, Maliwan Tengsujaritkul, Chane Choed-Amphai, Supapitch Chanthong, Lalita Sathitsamitphong, Rungrote Natesirinilkul, Pimlak Charoenkwan, Department of Pediatrics, Faculty of Medicine, Chiang Mai University, Chiang Mai 50200, Thailand
Kanittha Mankhemthong, Department of Pediatrics, Nakornping Hospital, Chiang Mai 50180, Thailand
Author contributions: Permtawee K, Tengsujaritkul M and Choed-Amphai C drafted the manuscript; Permtawee K, Tengsujaritkul M, Choed-Amphai C, Chanthong S, Mankhemthong K, Sathitsamitphong L, Natesirinilkul R and Charoenkwan P participated in patient management and data collection; Permtawee K, Choed-Amphai C and Chanthong S contributed to the systematic review; All authors contributed to the article and approved the submitted version.
Conflict-of-interest statement: All the authors declare that they have no competing interests.
PRISMA 2009 Checklist statement: The authors have read the PRISMA 2009 Checklist, and the manuscript was prepared and revised according to the PRISMA 2009 Checklist.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Chane Choed-Amphai, MD, Doctor, Department of Pediatrics, Faculty of Medicine, Chiang Mai University, No. 110 Intawarorot road, Sriphum, Muang, Chiang Mai 50200, Thailand. chane.c@cmu.ac.th
Received: July 22, 2023
Peer-review started: July 22, 2023
First decision: September 4, 2023
Revised: September 9, 2023
Accepted: September 26, 2023
Article in press: September 26, 2023
Published online: December 9, 2023
ARTICLE HIGHLIGHTS
Research background

Type B lactic acidosis is a rare metabolic complication in children with hematologic malignancies which can mimic inborn errors of metabolism (IEM). There have been few pediatric case series and case reports published on this specific condition. Moreover, the most recent review was conducted over two decades ago, in 2001.

Research motivation

The illustrative case of a 10-year-old boy with Burkitt leukemia who exhibited Warburg effect mimicking IEM was the basis of our systematic review. The previous review of this metabolic complication showed an extremely poor outcome. In recent years, however, advancements in cancer treatments may have led to the overall improvement in the clinical course.

Research objectives

To identify the clinical course, treatment strategies, and outcomes of childhood hematologic malignancies with type B lactic acidosis.

Research methods

We performed a comprehensive search of the PubMed, Scopus, and Cochrane databases without any time restrictions to identify children with leukemia/lymphoma and type B lactic acidosis. The publications considered for inclusion were limited to English language articles.

Research results

Lactic acidosis initially subsided in 80% of patients receiving chemotherapy compared to 60% in the contrast group. The mortality rate of newly diagnosed cases was 45.5%, while the relapsed cases had a 100% mortality rate. All 8 cases reported before 2001 died from disease-related complications, while cases reported between 2003 and 2023 showed a 54.5% rate of complete remission.

Research conclusions

Historically, this complication has led to fatal outcome; however, patients who received chemotherapy showed a more favorable response. Therefore, it is crucial to promptly initiate specific treatment in this context.

Research perspectives

This systematic review has revealed an improvement in the clinical course and outcomes compared to the past. Future studies in this context might include a larger scale of cases involving multicenter research. Retrospective study on prognostic factors or therapeutic research in the era of immunotherapy and targeted therapy could also be performed in this population.