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Case Reports
. 2022 Mar 27;21(1):15.
doi: 10.1186/s12941-022-00506-6.

An unexpected case of Borrelia garinii liver infection

Pierre Duffau  1   2 , Skander Korbi  3 , Vivien Guillotin  1 , Emilie Talagrand-Reboul  4   5 , Armelle Ménard  6   7 , Olivia Peuchant  8   9
Affiliations
Case Reports

An unexpected case of Borrelia garinii liver infection

Pierre Duffau et al. Ann Clin Microbiol Antimicrob. .

Abstract

Background: Lyme borreliosis is the most prevalent arthropod-borne infection in the Northern Hemisphere. In Europe, Borrelia afzelii is predominantly involved in cutaneous manifestations, Borrelia garinii and Borrelia bavariensis in neurological manifestations, and Borrelia burgdorferi sensu stricto in articular ones. Liver impairement is not classical in Lyme borreliosis. Diagnosis is currently mainly based on serological testing, and is challenging in immunocompromised patients.

Case presentation: We report the first case of B. garinii infection revealed by liver involvement in an immunocompromised man. A 73-year-old man with marginal zone lymphoma, treated with bendamustine and rituximab, developed intermittent fever and inflammatory syndrome. Microbial investigations were all negative and FDG-PET showed complete remission of the lymphoma. Three months later, liver biopsy was performed and histology revealed spirochetes-like bacteria. Microbial diagnosis was performed by 16S rDNA sequencing, flagellin (flaB) gene sequencing and multi-locus sequence typing and identified B. garinii. The patient recovered successfully after a three weeks course of antibiotics. Diagnosis was challenging because Borrelia hepatic involvement is unusual and no erythema migrans nor tick bite were notified.

Conclusion: This case highlights that unexplained fever and inflammatory syndrome in immunocompromised patients warrants specific investigations to identify bacteria such as spirochetes.

Keywords: 16S rDNA; Borrelia garinii; Kupffer cell hyperplasia; Liver involvement; Warthin Starry stain.

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Conflict of interest statement

The authors declare that they have no competing interest.

Figures

Fig. 1
Fig. 1
Images of human liver biopsy specimens. Warthin Starry silver nitrate staining showing spirochete-like bacteria (blue arrowheads) (original magnification × 400)
Fig. 2
Fig. 2
Phylogenetic tree of Borrelia taxa based on flagellin gene (158 nt). DNA extracts from two different areas of the liver biopsy were analyzed (indicated in red). The phylogenetic analyses were generated with the BioNeighbor-Joining method. The phylogeny presented is based on the alignment of 158-bp sequences of flaB gene from 44 reference sequences representing the current known diversity in the genus Borrelia and the sequence amplified in the case report presented herein. The percentage of replicate trees in which the associated taxa clustered together in the bootstrap test (1,000 replicates) is shown next to the branches. The tree is not rooted and drawn to scale, with branch lengths in the same units as those of the evolutionary distances used to infer the phylogenetic tree. The evolutionary distances were computed using the Kimura 2-parameter method and are represented in the units of the number of base substitutions per site. Evolutionary analyses were conducted using SeaView. All sequences are labeled by species, strain name, and GenBank accession number. TS type strain
See this image and copyright information in PMC

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