International Journal of Infectious Diseases 30 (2015) e133–e134

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Case Report

First case of osteomyelitis due to Erysipelothrix rhusiopathiae: pubic osteomyelitis in a gored farmer E. Denes a,*, Y. Camilleri b, F. Fiorenza c, C. Martin d a

Infectious Diseases Department, Limoges Teaching Hospital, France Radiology Department, Limoges Teaching Hospital, France c Orthopaedics Surgery Department, Limoges Teaching Hospital, France d Bacteriology Laboratory, Limoges Teaching Hospital, France b

A R T I C L E I N F O

Article history: Received 6 October 2014 Received in revised form 26 October 2014 Accepted 19 November 2014 Keywords: Erysipelothrix rhusiopathiae pubic osteomyelitis bone infection osteitis cow pubic bone osteomyelitis

A B S T R A C T

We report the first proven case of osteomyelitis due to Erysipelothrix rhusiopathiae. This infection occurred almost 20 years after traumatic inoculation of the bacterium, when the patient was gored by one of his cows. Diagnosis was made by bone biopsies, and treatment included rifampicin and levofloxacin for 3 months. ß 2014 The Authors. Published by Elsevier Ltd on behalf of International Society for Infectious Diseases. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/bync-nd/3.0/).

1. Case Report In 1992, a 38 year-old farmer with no past medical history was gored by a cow, resulting in an iliac fossa wound with an ileum evisceration but no vascular injury. During the next 19 years, he did not show any sequelae to this injury and there was no other wound in relation with his cattle. In 2011, he underwent surgery for a supposed inguinal hernia, but the surgeon found an abscess. No bacteria were isolated except for Staphylococcus aureus on a superficial swab. Chronic pus formation persisted, and a second surgery was performed in 2012,during which a fistula in contact with the pelvic bones was discovered. No bacterium was found on samples. In 2013, the patient was referred to our hospital for advice. Clinical examination showed a chronic fistula and pain in both adductor muscles and the pelvis, but no fever. A CT scan was performed, which revealed the fistula, pubic symphysis arthritis and bilateral pubic osteomyelitis (Figure 1). Several CT-scan guided biopsies were done for bacteriological analysis. The culture of 2 out of 3 biopsies revealed Erysipelothrix rhusiopathiae.

* Corresponding author. Service de Maladies Infectieuses et Tropicales, CHU Dupuytren, 2, Avenue Martin Luther King, 87000 Limoges. Tel.: +33.5.55.05.66.44; fax: +33.5.55.05.66.48. E-mail address: [email protected] (E. Denes).

Identification was done by mass spectrometry technology (Biomerieux, France) and confirmed by 16S RNA sequencing. An antibiotic combination using rifampicin (900 mg BID) and levofloxacin (500 mg BID) was prescribed for 3 months. This combination was chosen because of the bone localisation. A high dose of rifampicin was prescribed due to the weight of the patient (105 kg). Because of previous reported chronic infections with endocarditis, a cardiac echography was performed and did not reveal any endocarditis. After 2 months of antibiotics, the patient had total pain relief (in the legs and pelvis). Six months after the cessation of antibiotics, there was no relapse. E. rhusiopathiae is a Gram-positive, non-spore-forming, nonacid- fast, rod-shaped bacterium. This agent is an occupational pathogen for humans and is the cause of 3 human diseases. These pathologies are: a localized cutaneous form, also known as erysipeloid; a generalized cutaneous form; and a septicemic form that is often associated with endocarditis1. Only a few cases of arthritis have been reported. To our knowledge, osteomyelitis due to E. rhusiopathiae has never been reported before. This case is interesting for two main reasons. First, this is the first time that it is proven that E. rhusiopathiae can infect bones. In a previous report, osteolysis was found on the left pedicle of the 3rd lumbar vertebra in a patient with endocarditis due to E. rhusiopathiae. Bone infection was thought to be due to the same bacterium, however, culture of bone biopsies remained sterile2.

http://dx.doi.org/10.1016/j.ijid.2014.11.015 1201-9712/ß 2014 The Authors. Published by Elsevier Ltd on behalf of International Society for Infectious Diseases. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/3.0/).

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Figure 1. A) Fistula and inflammation in the pubic area. B) Bilateral osteolysis of the pubic bone.

Secondly, in our case, the horn accident is more than likely the source of the infection. Indeed, E. rhusiopathiae is found, among other locations, in soil1. The bacterium could have contaminated the horn, which then, transported the bacterium to or near the pubic bone during the injury. Its long survival in soil is known, but has not been reported previously in humans. It is known that other bacteria, such as Staphylococcus spp., can persist for a long time in a bone biofilm and be reactivated for an unknown reason3. This is what probably occurred in our patient, even if the period between inoculation and infection was very long (19 years). Furthermore, E. rhusiopathiae has adhesive surface proteins (RspA and RspB), which share some characteristics with those of Staphylococcus aureus4. These surface proteins are thought to initiate biofilm formation4. These characteristics can then explain the persistence of the bacteria in the pubic bone of our patient until its reactivation and the occurrence of osteomyelitis. Usually, E. rhusiopathiae is traumatically inoculated, in butchers or fish handlers for example, by contaminated bones or fish bones. This can be followed by erysipeloid, but considering this case we must now keep in mind that if the wound reaches bone, such as in fingers, a careful follow up must be carried out to be sure that no bone infection is left untreated.

In conclusion, we report the first case of a proven bone infection due to E. rhusiopathiae. It was the consequence of a direct inoculation of the bacteria by a cow horn, and proves that this bacterium can stay latent for a very long time, probably thanks to biofilm. Conflict of Interest/Funding None There is no conflict of interest, and no financial support was received for the study. References 1. Wang Q, Chang BJ, Riley TV. Erysipelothrix rhusiopathiae. Vet Microbiol 2010;140(3–4):405–17. http://dx.doi.org/10.1016/j.vetmic.2009.08.012. 2. Romney M, Cheung S, Montessori V. Erysipelothrix rhusiopathiae endocarditis and presumed osteomyelitis. Can J Infect Dis 2001;12(4):254–6. 3. Wright Ja, Nair SP. Interaction of staphylococci with bone. Int J Med Microbiol 2010;300(2–3):193–204. http://dx.doi.org/10.1016/j.ijmm.2009.10.003. 4. Shimoji Y, Ogawa Y, Osaki M, et al. Adhesive Surface Proteins of Erysipelothrix rhusiopathiae Bind to Polystyrene, Fibronectin, and Type I and IV Collagens. J Bacteriol 2003;185(9):2739–48. http://dx.doi.org/10.1128/JB. 185.9.2739.

First case of osteomyelitis due to Erysipelothrix rhusiopathiae: pubic osteomyelitis in a gored farmer.

We report the first proven case of osteomyelitis due to Erysipelothrix rhusiopathiae. This infection occurred almost 20 years after traumatic inoculat...
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