Lemierre Syndrome: Report of a Case with an Innovative Diagnostic Method and Literature Review

Abstract

Objective

To understand the clinical features, diagnosis and treatment of Lemierre syndrome (LS), a high-risk and low-prevalence infectious disease.

Methods

We present the severe LS case that was diagnosed using metagenomic next-generation sequencing (mNGS) in our hospital, and systematically summarized the diagnosis and treatment strategies of patients that reported LS from 2006 to 2022.

Results

The 24-year-old patient in our hospital suffered from cranial nerve paralysis, a neurological complication rarely seen in LS cases. The causative agent (Fusobacterium necrophorum, Fn) of this patient was only detected by mNGS tests, and the reads number of Fn detected by plasma mNGS tests was decrease as the patients gradually improved, indicating plasma mNGS is valuable in monitoring treatment efficacy. Although most of the cases retrieved from the literature showed typical symptoms, such as a history of sore throat, septic emboli, and internal jugular vein thrombosis, clinical manifestations were still relatively heterogeneous (eg, diversity of predisposing factors and pathogens, differences in pulmonary imaging features).

Conclusion

We summarized the clinical presentation, diagnosis, treatment, and regression of 17 symptomatic cases reported LS to provide clinicians with knowledge about this rare but fatal disease. mNGS assays should be considered as early as possible to identify the responsible pathogens for acute and critically ill patients with suspected infections in order to implement accurate and effective treatment.

Keywords:

• lemierre syndrome
• Fusobacterium necrophorum
• metagenomic next-generation sequencing
• thrombophlebitis
• sepsis

Ethical Approval Statement

This study was approved by the First Affiliated Hospital, Zhejiang University School of Medicine (FAHZU) institutional review board (IIT20220714A). The informed consent was obtained from the patient for the publication of all images, clinical data and other data.

Acknowledgments

We thank all clinicians who provided detailed diagnostic and treatment data of the patient for our study, as well as all infectious disease (ID) physicians and clinical microbiologists who received our infectious disease consultations.

Author Contributions

All authors made a significant contribution to the work reported, whether that is in the conception, study design, execution, acquisition of data, analysis and interpretation, or in all these areas; took part in drafting, revising or critically reviewing the article; gave final approval of the version to be published; have agreed on the journal to which the article has been submitted; and agree to be accountable for all aspects of the work.

Disclosure

The authors declare no competing interests.

Additional information

Funding

This study was supported by the Zhejiang Provincial Natural Science Foundation (grant number LY23H200001 by D.H.) and China International Medical Foundation (grant numbers Z-2017-24-2202 by D.H.).