Chronic Splenic Melioidosis in a Patient with Fever of Unknown Origin Diagnosed by Metagenomics Next-Generation Sequencing: An Emerging Cause and Literature Review

Abstract

Introduction

Human melioidosis is an emerging infectious disease in tropical areas of China, and chronic melioidosis can be a rare cause of fever of unknown origin (FUO). Timely diagnosis may improve the prognosis of melioidosis.

Case Presentation

We report a case of melioidosis with splenic abscesses caused by Burkholderia pseudomallei in a 57-year-old man, who presented with FUO. Positron emission tomography/computed tomography (PET/CT) revealed multiple hypermetabolic lesions in the spleen. The spleen biopsy was conducted and metagenomics next-generation sequencing (mNGS) of the spleen specimen identified the presence of B. pseudomallei, confirming the diagnosis of melioidosis. Antimicrobial treatment was initiated with intravenous meropenem, followed by oral faropenem. During the follow-up, the patient was in good condition except having a low-grade fever occasionally. A splenectomy was performed, and subsequent culture and mNGS of the spleen pus were both positive for B. pseudomallei. Histopathological characteristics of chronic splenic melioidosis were noted.

Conclusion

Melioidosis is a serious endemic disease, and it is critical to raise awareness about this disease.

Keywords:

• melioidosis
• Burkholderia pseudomallei
• metagenomics next-generation sequencing
• splenectomy

Abbreviations

FUO, fever of unknown origin; mNGS, metagenomics next-generation sequencing; ¹⁸F-FDG, ¹⁸F-fluorodeoxyglucose; PET/CT, positron emission tomography/computed tomography; SUVmax, the maximum standard uptake value; CRP, C-reactive protein; ESR, erythrocyte sedimentation rate; CT, computed tomography; MRI, magnetic resonance imaging; SMZ-TMP, trimethoprim-sulfamethoxazole.

Data Sharing Statement

The original contributions presented in the study are included in the report, further inquiries can be directed to the corresponding author.

The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

Ethics Approval and Consent to Participate

This study was approved by the Ethics Review Committee of Ruijin Hospital, Shanghai Jiao Tong University School of Medicine. Written informed consent has been obtained from the patient in accordance with the Declaration of Helsinki.

Consent for Publication

The patient of this report had already agreed and signed the consent form.

Acknowledgments

The authors thank Dr. Ruokun Li for his help in revising the MR images and legends.

Disclosure

The authors declare that they have no competing interests.

Additional information

Funding

This study was supported by funding from the Shanghai Municipal Key Clinical Specialty (Infectious disease, YW20190002 to Q. Xie).