Description
A 25-year-old woman presented with a 3-week history of fever and weight loss on return from a trip to Thailand. Clinical examination revealed inflammatory tumefaction of the right knee and of the right wrist. Positive blood cultures identified Burkholderia pseudomallei. MRI confirmed right palmar tenosynovitis and osteoarthritis of the carpus (figure 1A, B) as well as osteomyelitis of the right femur's lower extremity (figure 1C, D). In addition, a positron emission tomography CT (PET-CT) scan (figure 1E) showed multifocal melioidosis involving the liver, lung, lymph nodes and multiple cutaneous and muscular nodules. Intravenous ceftazidime and oral cotrimoxazole were started. Her general condition improved but surgical procedures were required for the bone and joint foci: (1) palmar tenosynovitis on month 2; (2) aggressive sequential femoral intramedullary lytic ‘Brodie-like’ abscess curettage on months 2 and 3 on account of persistent productive fistula. Ceftazidime was switched to oral levofloxacin on month 5. A PET-CT scan on month 7 demonstrated significant decline in hypermetabolism of the right femur's lower extremity and resolution of other hypermetabolic foci. Levofloxacin–cotrimoxazole was switched to doxycycline on month 10. Antimicrobial therapy was stopped on month 12 as the PET-CT scan showed that the patient had normalised. The patient was followed up for 2 years without evidence of relapse.
Figure 1.
Multifocal melioidosis with osteoarthritis and palmar tenosynovitis of the right hand evidenced by T1-weighted MRI (without (A) and with (B) gadolinium), osteomyelitis of the right femur's lower extremity evidenced by T1-weighted MRI (without (C) and with (D) gadolinium), liver, lung and lymph node involvement, multiple cutaneous and muscular nodules as featured by positron emission tomography-CT (E).
Melioidosis, an endemic disease in Southeast Asia, is increasingly reported in travellers. Typical presentation includes pneumonia and recurrent abscesses, with or without bacteraemia. Bone and joint involvement has been exceptionally reported in travellers without comorbidities. In a prospective study, the incidence of osteomyelitis and/or septic arthritis during melioidosis in endemic areas was 7.6%.1 In our case, osteotomy and several femoral curettages were required to improve femoral infection, which has never been described. Otherwise, a PET-CT scan has been suggested to evaluate dissemination at diagnosis and follow-up, which greatly facilitates the diagnosis of secondary location (figure 1, panel E).2 Treatment of osteomyelitis during melioidosis relies on repeated surgical debridement of collections and prolonged intravenous antimicrobial therapy by ceftazidime or carbapenem for ≥6 weeks, followed by oral cotrimoxazole for ≥6 months.3
Learning points.
Burkholderia pseudomallei should be considered as a possible aetiological agent of sepsis and bone and joint infections in travellers returning from endemic regions.
Treatment of osteomyelitis consists of antimicrobial therapy for ≥6 months and surgical drainage of bone collections.
Positron emission tomography-CT is a valuable tool for initial evaluation of dissemination of melioidosis and follow-up under appropriate treatment.
Acknowledgments
The authors would like to thank Lyon Bone and Joint Infection Study Group. Coordinator: TF; infectious diseases specialists—TF, FV, Thomas Perpoint, André Boibieux, François Biron, Patrick Miailhes, FA, Julien Saison, Sandrine Roux, Claire Philit, Fatiha Daoud, Johanna Lippman, Evelyne Braun, Christian Chidiac, Yves Gillet, Laure Hees; surgeons—Sébastien Lustig, Philippe Neyret, Olivier Reynaud, Adrien Peltier, Olivier Cantin, Michel-Henry Fessy, Anthony Viste, Philippe Chaudier, Romain Desmarchelier, Thibault Vermersch, Cédric Barrey, Francesco Signorelli, Emmanuel Jouanneau, Timothée Jacquesson, Ali Mojallal, Fabien Boucher, Hristo Shipkov, Mehdi Ismail, Joseph Chateau; microbiologists—Frederic Laurent, François Vandenesch, Jean-Philippe Rasigade, Céline Dupieux, Sophie Trouillet-Assant; nuclear medicine—Isabelle Morelec, Marc Janier, Francesco Giammarile; PK/PD specialists—Michel Tod, Marie-Claude Gagnieu, Sylvain Goutelle; Clinical Research Assistant—Eugénie Mabrut.
Footnotes
Contributors: AC, FV, TF and FA participated to the patient care, the writing and the revision of the manuscript.
Competing interests: None declared.
Patient consent: Obtained.
Provenance and peer review: Not commissioned; externally peer reviewed.
References
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