Coxiella burnetti prosthetic joint infection in an immunocompromised woman: iterative surgeries, prolonged ofloxacin-rifampin treatment and complex reconstruction were needed for the cure

Abstract

Background: Q fever is a zoonotic disease caused by the bacterium Coxiella burnetii, a strictly intracellular pathogen that can cause acute and chronic infection. Chronic Q fever can occur in immunocompetent as well as in immuno-compromised hosts, as a persistent localized infection. The main localizations are endocardial, vascular and, less frequently, osteoarticular. The most frequent osteoarticular form is spondyliscitis. Recommended treatment is combined doxycycline and hydroxychloroquine for 18 months, with cotrimoxazole as another option. Coxiella burnetti infection has been implicated in rare cases of prosthetic joint infection (PJI), and the medical and surgical management and outcome in such cases have been little reported.

Case presentation: We report an unusual case of chronic Q fever involving a hip arthroplasty in an immunocompromised woman treated with tumor necrosis factor (TNF)-α blockers for rheumatoid arthritis. Numerous surgical procedures (explantation, "second look", femoral resection and revision by megaprosthesis), modification of the immunosuppressant therapy and switch from doxycycline-hydroxychloroquine to prolonged ofloxacin-rifampin combination therapy were needed to achieve reconstruction and treat the PJI, with a follow-up of 7 years.

Conclusions: Coxiella burnetti PJI is a complex infection that requires dedicated management in an experienced reference center. Combined use of ofloxacin-rifampin can be effective.

Keywords: Anti-TNF-α; Coxiella burnetii; Prosthetic joint infection; Q fever; Rheumatoid arthritis.

Fig. 1

X-ray and CT-scan of the left hip performed: A at admission; B after prosthesis explantation, debridement and set-up of a spacer; C after performance of a new debridement and spacer removal, unfortunately with recurring abscesses visible on CT-scan (C1: ilio-psoas recurrent abscess, red arrow; C2: large collection within the joint; C3: abscesses in the thigh, red arrow) despite prolonged hydroxychloroquine-doxycycline treatment; D after a subsequent surgery with debridement of the recurrent abscesses and performance of a femoral resection; E 6 years after reimplantation of a megaprosthesis, no prosthesis loosening

Fig. 2

Kinetics of anti-phase I IgG titers (all previous sera were usually but not systematically analyzed in parallel) and results of the specific coxiella PCR during antimicrobial therapy and surgical procedures according to immunofluorescence assays. The Ct of the specific PCR (Smartcycler), when positive, was respectively 33.7, 31.3 and 24.6. Notes: HCQ = Hydroxychloroquine. Surgical acts: the first was performed on the 6th, August 2013, the second the 29th, October 2013 and the third, the 15th, January 2014

Fig. 3

Positron emission tomography/computed tomography (PET-CT) performed ~ 3 months after the prosthesis explantation, showing a large ilio-psoas muscle collection (47 × 56 mm) communicating with numerous periprosthetic collections of the left hip (panel A). A second PET-CT was performed in June 2014, after the third surgical procedures and at ~ 6 month of rifampin and ofloxacin combination therapy, showing no persistent inflammatory process of the left hip and thigh (panel B)