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ScienceDirect www.sciencedirect.com Médecine et maladies infectieuses 45 (2015) 47–49

Case report

The first autochthonous case of human melioidosis in Reunion Island夽 Premier cas autochtone de melioïdose à la Réunion G. Borgherini a,∗ , G. Camuset a , A. Foucher a , J.C. Maiza b , F.M. Thibault c , S. Picot d , P. Poubeau a a

Service de maladies infectieuses, centre hospitalier universitaire de Saint-Pierre, BP 350, 97448 Saint-Pierre, Reunion b Endocrinologie, centre hospitalier universitaire de Saint-Pierre, 97448 Saint-Pierre, Reunion c Institut de recherche biomédicale des Armées, 91220 Brétigny-sur-Orge, France d Microbiologie, centre hospitalier universitaire de Saint-Pierre, 97448 Saint-Pierre, Reunion Received 5 October 2014; received in revised form 13 November 2014; accepted 30 November 2014 Available online 14 January 2015

Keywords: Melioidosis; Burkholderia pseudomallei; Reunion island; Prostatitis Mots clés : Mélioïdose ; Burkholderia pseudomallei ; Réunion ; Prostatite

1. Introduction Little is known about the true distribution of melioidosis in Africa. Sporadic cases have been observed in several countries [1], including some islands such as Madagascar [2–4], Mauritius [5], and the Seychelles [6] in the South-West Indian Ocean. Imported melioidosis has already been documented in Reunion Island [2,3,7], a French overseas district, but no case of locally acquired melioidosis has ever been recorded. We report the case of a 57-year-old male patient presenting with prostatic melioidosis. This patient, beside some short travels to Mauritius, had never left Reunion Island and it seems very likely that the infection was acquired locally. 2. Case presentation A 57-year-old male patient presenting with type 2 diabetes mellitus (requiring insulin and metformin treatment) and hypertension was admitted to the hospital on March 16, 2012, with a 4-day history of fever and dysuria. The patient, a technical manager, has always lived in Reunion Island except for 3 travels to Mauritius, the last one in 2007. None of these stays had lasted more than 1 week: the patient, who was on vacation, spent most of his time in a beach resort.

夽 Presented as a poster at the 7th World Melioidosis Congress, Bangkok, Thailand, September 18 to 20, 2013. ∗ Corresponding author. E-mail address: [email protected] (G. Borgherini).

http://dx.doi.org/10.1016/j.medmal.2014.11.008 0399-077X/© 2014 Elsevier Masson SAS. All rights reserved.

He had no pets, no garden but owned an orchard where he used to go once a week. The patient and his family sometimes used a storage tank, where tap water had been collected in the previous months. On admission, he was febrile (39 ◦ C) and in acute urinary retention, requiring the placement of a urinary catheter. His blood pressure was normal (130/70 mmHg). The physical examination yielded only a tender and enlarged prostate gland without any other physical abnormalities. The blood test results showed an elevated C-reactive protein (248 mg/L) and fasting hyperglycemia (17 mmol/L); the other results, including the leukocyte count, were normal. The patient was admitted with a diagnosis of acute bacterial prostatitis and an empirical ceftriaxone was initiated. The urine culture and 3 blood cultures were positive for a Gram-negative rod identified as Burkholderia pseudomallei by the Vitek Compact biochemical identification (bioMérieux La Balme-Les-Grottes, France). The antibiotic susceptibility was tested with the Vitek Compact apparatus (bioMérieux La BalmeLes-Grottes, France) and with antimicrobial susceptibility disks (BIORAD Marnes-la-Coquette, France). The bacterium was sensitive to ceftazidime, imipenem, doxycycline, and resistant to all aminosides and colistin, according to the European Committee on Antimicrobial Susceptibility Testing (EUCAST). Trimethoprim-sulfamethoxazole was at the threshold of sensitivity. The identification and antimicrobial susceptibility were confirmed by a referral center, the Institute for biomedical research from the French military medical service (Centre de recherche du service de santé des Armées) in La Tronche, France.


G. Borgherini et al. / Médecine et maladies infectieuses 45 (2015) 47–49

B. pseudomallei identification was confirmed by Polymerase Chain Reaction identification targeting type III secretion system genes [8] performed on GeneXpert (Cepheid Europe S.A.S.,Maurens-Scopont, France), MALDI-TOF mass spectrometry performed on a MALDI Biotyper system (Bruker Daltonique, Wissembourg, France) and 16S rDNA sequencing. A CT-scan revealed an enlarged, heterogeneous prostate with multiple fluid septations, the largest one was 2.5 cm in diameter; no other focus of infection was detected in the thorax and in the abdomen. The therapy was switched to intravenous ceftazidime (2 g every 8 hours) and doxycycline (100 mg every 12 hours). The fever subsided with this treatment and the CRP came back to normal. The patient’s blood glucose level came back to normal once the sepsis was controlled. The patient was discharged. He carried a peripheral catheter to complete a 4-week course of ceftazidime. Doxycycline was also continued and then associated to oral trimethoprim-sulfamethoxazole for a 5-month eradication treatment. A control CT-scan was performed after 3 months of treatment showing an almost complete resolution of the prostatic abscesses. The patient was asymptomatic at 12 months of follow-up. 3. Discussion B. pseudomallei, the causative agent of melioidosis, is found in the natural environment (ground and surface water) in tropical and subtropical regions. Melioidosis is endemic in South-East Asia and Northern Australia but its true global distribution remains to be determined. The infection results from a percutaneous inoculation, inhalation, or ingestion. The disease is often observed in patients with an impaired host immune response, and diabetes is a well-known risk factor. Melioidosis features protean clinical presentations, ranging from acute, fulminant, septic illness to chronic infection. A case of animal melioidosis, related to an outbreak in France (the so-called “Affaire de Jardin des Plantes”) occurred in 1979 in a horse recently imported in Reunion Island [7]. Two cases of human melioidosis acquired in Madagascar were diagnosed in Reunion Island in 2004 [2] and 2005 [3]. The first case was the index for 2 secondary cases at the hospital, infected by a contaminated bronchoscope [2]. No autochthonous case of melioidosis had ever been reported in Reunion Island. It seems very unlikely that our patient acquired the infection out of the island. The only foreign place the patient had visited was Mauritius, a non-endemic country, where only 1 case of melioidosis has been documented so far [5]. The patient did not report any risk activity during his stays in Mauritius, which never lasted more than 1 week. Considering the acute presentation of this case, an incubation period of 5 years (his latest visit to Mauritius in 2007) seemed improbable. The disease was acquired during the Reunion Island rainy season, as in 80% of melioidosis cases. The patient had reported no risk activity in the weeks before the onset of symptoms. The domestic use of water from a

storage tank had already been described as a possible source of contamination [9] and was most likely the source of infection. Unfortunately, it was not possible to analyze environmental samples and to identify precisely how the patient was infected. The patient presented with diabetes mellitus, a known risk factor for melioidosis, which could explain that nobody else using the same water tank was infected. Prostatic involvement is a known presentation of melioidosis. A routine CT abdomen and pelvis scan is performed in every case of melioidosis in Northern Australia and prostatic abscesses were diagnosed in 21% of male patients [10]. The prevalence of prostatic infection is much lower among Thai patients. The surgical treatment of prostatic melioidosis is debated. An ultrasound guided needle drainage combined to antibiotic treatment is recommended for prostatic abscesses in Australia [10]. Melioidosis resolved on antibiotic treatment alone in our case. 4. Conclusion The high prevalence of diabetes, the tropical climate, and a population often involved in agricultural activities, make Reunion Island an ideal setting for melioidosis. The health care system is similar to that of continental France and blood cultures are systematically performed for febrile patients admitted to the hospital: it thus seems unlikely that melioidosis is under diagnosed on the island. Further research is required to prove the presence of B. pseudomallei in Reunion Island as suggested by our case. Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. Acknowledgements G. Borgherini, G. Camuset, A. Foucher, J.C. Maiza and P. Poubeau were involved in the clinical care of the patient. S. Picot and F. Thibault’s help was necessary for the microbiological diagnosis. References [1] Katangwe T, Purcell J, Bar-Zeev N, Denis B, Montgomery J, Alaerts M, et al. Human melioidosis, Malawi, 2011. Emerg Infect Dis 2013;19:981–4. [2] Martinet O, Pac Soo AM, Knezynski M. Melioidosis: regarding a case acquired in Madagascar and two nosocomial cases. Bull Soc Pathol Exot 2004;97:369 [in French]. [3] Garin B, Djaomazala I, Dubois-Cauwelaert N, Mahafaly Raharimanga V, Ralison F, Herindrainy P, et al. Autochthonous melioidosis in humans, Madagascar, 2012 and 2013. Emerg Infect Dis 2014;20:1735–7. [4] Borgherini G, Poubeau P, Paganin F, Picot S, Michault A, Thibault F, et al. Melioidosis: an imported case from Madagascar. J Travel Med 2006;13:318–20. [5] Issack MI, Bundhun CD, Gokhool H. Melioidosis in Mauritius. Emerg Infect Dis 2005;11:139–40. [6] Bibi J, Biscornet L, Bermingham A, von Gottberg A. First identification of Burkholderia pseudomallei in Seychelles. In: Proceeding of the 1st International Forum Public Health Surveillance and Response in Island

G. Borgherini et al. / Médecine et maladies infectieuses 45 (2015) 47–49 Territories (The S.E.G.A. Network [Surveillance Épidémiologique et Gestion des Alertes]); 2013 [Reunion Island]. [7] Galimand M, Dodin A. Focus on melioidosis throughout the world [in French]. Bull Soc Pathol Exot 1982;75:375–83. [8] Thibault FM, Valade E, Vidal DR. Identification and discrimination of Burkholderia pseudomallei, B. mallei, and B. thailandensis by realtime PCR targeting type III secretion system genes. J Clin Microbiol 2004;42:5871–4.


[9] Currie BJ, Mayo M, Anstey NM, Donohoe P, Haase A, Kemp DJ. A cluster of melioidosis cases from an endemic region is clonal and is linked to the water supply using molecular typing of Burkholderia pseudomallei isolates. Am J Trop Med Hyg 2001;65:177–9. [10] Morse LP, Moller CC, Harvey E, Ward L, Cheng AC, Carson PJ, et al. Prostatic abscess due to Burkholderia pseudomallei: 81 cases from a 19year prospective melioidosis study. J Urol 2009;182:542–7.

The first autochthonous case of human melioidosis in Reunion Island.

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