Figure 1

Figure 1. Large erythema chronicum migrans rash on a 60-year-old woman in France that was later determined to be caused by Borrelia spielmanii. A) Edges of linear erythema band on...

A 60-year-old woman sought treatment on November 15, 2017, for 2 erythematous linear asymptomatic and noninfiltrated bands on her left knee and the left side of her thorax. In August 2017, the woman had noticed an annular erythema initially in the middle and on the left side of her back. The annular erythema had gradually extended to the upper left side of her back to form a single erythematous band (Figure 1, panel A) and progressed to the left knee, also forming an erythematous band (Figure 1, panel B). Both general and skin exams were otherwise unremarkable. We hypothesized that the rash might constitute an unusually large ECM. Because Lyme borreliosis is unknown in that area of southeastern France, we questioned her about her recent travel history. She had spent the week of June 14–20, 2017, in the county of Oise, north of Paris, where Lyme borreliosis is endemic. We performed a punch biopsy on the thoracic erythema band and prescribed oral amoxicillin (1 g 3×/d for 15 d), which resolved the ECM.

We screened a serum sample from the patient using an enzyme-linked immunoassay (Liaison Borrelia burgdorferi; DiaSorin, https://www.diasorin.com), which revealed presence of IgG and absence of IgM for B. burgdorferi sensu lato. Western blotting using LymeCheck Optima IgG & IgM (Biosynex, https://www.biosynex.com) revealed presence of Borrelia spp.–specific IgG for p100, VlsE, p58, and p41 antigens and a faint band for B. spielmanii–specific ospC antigen (Appendix Figure). Two weak bands showed IgM for B. spielmanii p100 and p41 antigens (Appendix Figure).

Figure 2

Figure 2. Maximum-likelihood phylogenetic tree of the 16s rRNA gene (rrs) of Borrelia genus bacteria showing the position of the B. spielmaniisequence obtained from the patient (large bold...

We amplified portions of 16S rRNA (13) and ospA (5) borrelial genes from the biopsy sample. BLAST search (https://blast.ncbi.nlm.nih.gov/Blast.cgi) of sequenced amplicons showed 100% identity of the biopsy sequences with strain A14S of B. spielmanii for both the 910 bp–long portion of 16S rRNA gene (Genbank accession no. AF102056) and 260 bps-long amplicon of ospA (Genbank accession no. CP001469). We deposited our sequences into Genbank (accession nos. OR192893 and OR234396). The phylogenic tree (Figure 2) showed that the sequence obtained from the patient clusters with other B. spielmanii strains.

Previously, only B. azfelii, B. garinii, and B. burgdoferi sensu stricto had been identified from patients in France (14). Our patient manifested a rare clinical form of ECM, with a large erythema migrans across her body, extending from her upper back to her knee. However, the unusually large size might have resulted from delays in seeking treatment and diagnosis, so that particular clinical manifestation might not be specific to B. spielmanii.

We reviewed available literature on clinical descriptions of human cases of B. spielmanii infection and found only 2 published case reports. A 69-year-old woman from Slovenia showed skin manifestations described as redness, mild local itching, burning, and pain on the left knee and later a 24 × 20 cm ring-like lesion on the left thigh (8), but she had no identified tick bite. In a second case, a 42-year-old woman from Hungary exhibited an ECM 10 cm in diameter on her knee (6). Clinical manifestations were missing in other case reports (3,9), in which only human isolates were described. In 1 study, B. spielmanii was detected in isolates from 4/242 patients with ECM from Germany and Slovenia (9). However, in that study, 3 of the 4 patients infected with B. spielmanii lived in Munich where a higher proportion of ticks were positive for that pathogen. This finding argues for sporadic occurrences of the infection in other locations.

In summary, our study has contributed more data on Borrelia spp. as potential causes of Lyme disease, prompting need for broader surveillance. However, additional well-documented reports on ECM as a manifestation of B. spielmanii are needed to provide new information about the epidemiology of this Borrelia in Europe.