Sui MZ, Wan KC, Chen YL, Li HL, Wang SS, Chen ZF. Fatal hemophagocytic lymphohistiocytosis-induced multiorgan dysfunction secondary to Burkholderia pseudomallei sepsis: A case report. World J Clin Cases 2023; 11(30): 7372-7379 [PMID: 37969441 DOI: 10.12998/wjcc.v11.i30.7372]
Corresponding Author of This Article
Ze-Fu Chen, BMed, Attending Doctor, Department of Pediatrics, Hainan General Hospital, Xiuhua Road, Haikou 570311, Hainan Province, China. 42648804@hainmc.edu.cn
Research Domain of This Article
Pediatrics
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: http://creativecommons.org/licenses/by-nc/4.0/
Ming-Ze Sui, Department of Pulmonary and Critical Care Medicine, Kunming Children’s Hospital, Yunnan Key Laboratory of Children’s Major Disease Research, Kunming 650034, Yunnan Province, China
Ke-Cheng Wan, Yuan-Lu Chen, Huan-Long Li, Shan-Shan Wang, Ze-Fu Chen, Department of Pediatrics, Hainan General Hospital, Haikou 570311, Hainan Province, China
Author contributions: Sui MZ and Wan KC contributed equally to this work; Sui MZ wrote the original draft of the manuscript; Wan KC performed the analysis and interpretation of imaging findings; Chen YL and Li HL were responsible for the treatment of the patient and provided clinical data; Wang SS was responsible for literature retrieval; Chen ZF was responsible for the revision and editing of the manuscript; All authors approved the final version to be submitted.
Informed consent statement: The patient’s parents signed a written informed consent form.
Conflict-of-interest statement: The authors declare having no conflicts of interest.
CARE Checklist (2016) statement: The authors read the CARE Checklist (2016) and the manuscript was prepared and revised according to it Checklist (2016).
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: Ze-Fu Chen, BMed, Attending Doctor, Department of Pediatrics, Hainan General Hospital, Xiuhua Road, Haikou 570311, Hainan Province, China. 42648804@hainmc.edu.cn
Received: September 4, 2023 Peer-review started: September 4, 2023 First decision: September 13, 2023 Revised: September 24, 2023 Accepted: October 8, 2023 Article in press: October 8, 2023 Published online: October 26, 2023 Processing time: 51 Days and 7.4 Hours
Abstract
BACKGROUND
Burkholderia pseudomallei (B. pseudomallei) is a short, straight, medium-sized Gram-negative bacterium that mostly exists alone, without a capsule or spores, has more than three flagella at one end, and actively moves. B. pseudomallei confers high morbidity and mortality, with frequent granulocytopenia in B. pseudomallei sepsis-related deaths. However, mortality may be related to hemophagocytic lymphohistiocytosis (HLH) secondary to B. pseudomallei infection.
CASE SUMMARY
A 12-year-old female was referred from a local hospital to the pediatric intensive care unit with suspected septic shock and fever, cough, dyspnea, and malaise. After admission, supportive symptomatic treatments including fluid resuscitation, anti-infective therapy, mechanical ventilation, and a vasoactive drug maintenance cycle were carefully initiated. The patient became unconscious, her blood pressure could not be maintained even under the exposure of vasoactive drugs, and she experienced cardiorespiratory arrest. The patient died due to ineffective high-quality in-hospital cardiopulmonary resuscitation. A subsequent bone marrow smear examination revealed extensive phagocytosis, and the blood culture was positive for B. pseudomallei. Family history revealed a sibling death from B. pseudomallei sepsis 5 years earlier.
CONCLUSION
The higher mortality rate in patients with B. pseudomallei sepsis may be related to secondary HLH after infection, wherein multiorgan dysfunction syndrome may be directly related to infection or immune damage caused by secondary HLH. Patients with B. pseudomallei can be asymptomatic and can become an infective source.
Core Tip: Given the high mortality rate associated with Burkholderia pseudomallei (B. pseudomallei), it is particularly important to fully understand the pathogenesis. This report presents the clinical characteristics of a case of B. pseudomallei infection and some clinical data of the patient’s brother, who also died from B. pseudomallei infection. The chronic carrier status of B. pseudomallei and secondary hemophagocytic lymphohistiocytosis warrants attention in research on the pathogenesis and treatment of B. pseudomallei sepsis.
