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ScienceDirect www.sciencedirect.com Médecine et maladies infectieuses 45 (2015) 95–97

Case report

Late Stenotrophomonas maltophilia pacemaker infective endocarditis Endocardite infectieuse sur pacemaker à Stenotrophomonas maltophilia Q. Reynaud a , E. Weber a , A. Gagneux-Brunon a,b , F. Suy a , C. Romeyer-Bouchard c , F. Lucht a,b , E. Botelho-Nevers a,b,∗ b

a Infectious diseases department, Saint-Etienne university hospital, 42055 Saint-Etienne cedex 2, France GIMAP EA 3064 (groupe immunité des muqueuses et agents pathogènes), university of Lyon, 42023 Saint-Etienne, France c Cardiology department, Saint-Etienne university hospital, 42055 Saint-Etienne cedex 2, France

Received 30 December 2014; received in revised form 13 January 2015; accepted 29 January 2015 Available online 24 February 2015

Keywords: Pacemaker; Endocarditis; Stenotrophomonas maltophilia; Antibiotic resistance Mots clés : Pacemaker ; Endocardite infectieuse ; Stenotrophomonas maltophilia ; Résistance aux antibiotiques

1. Introduction

2. Case presentation

Stenotrophomonas maltophilia is a Gram-negative aerobic bacillus, distributed in natural and human environments, considered as an opportunistic pathogen, and responsible for device infections and other nosocomial infections [1]. S. maltophilia has intrinsic or acquired resistance to many antibiotics (betalactams, aminoglycosides, carbapenems, and fluoroquinolones) [2]. Pneumonia and bacteremia are the most common manifestations of S. maltophilia infection. Risk factors associated with S. maltophilia infection usually include admission to an intensive care unit, HIV infection, malignancy, cystic fibrosis, neutropenia, mechanical ventilation, central venous catheters, recent surgery, trauma, and previous therapy with broad spectrum antibiotics [1]. S. maltophilia infections have been associated with high morbidity and mortality rates [3]. Little documentation is available on infective endocarditis due to this bacterium. Only 40 cases of S. maltophilia infective endocarditis (IE) have been reported to date [4]; half of them were hospitalacquired and occurred most frequently on prosthetic valves. We present a rare case of S. maltophilia pacemaker endocarditis in an 81 year-old female patient.

An 81 year-old female patient was admitted to our hospital infectious diseases unit for a suspected pacemaker infection. She had a history of arterial hypertension, atrial fibrillation treated with Fluindione, rhizomelic pseudo-polyarthritis treated with Prednisone (5 mg per day), and chronic kidney failure related to a congenital single kidney. She was allergic to Penicillin. Seventeen months earlier, she had undergone a triple-chamber permanent pacemaker implantation to treat dilated cardiomyopathy. Twelve months after the implantation, she presented with erythema around the pacemaker pocket, with calor and tenderness. She was treated by local care without systemic antibiotics and no sampling was performed. Five months later, i.e. 17 months after implantation, she presented with a purulent discharge of the pacemaker pocket and externalization of one of the leads. At admission, her body temperature was 36.6 ◦ C, no heart murmur and no peripheral sign of infective endocarditis were observed. The initial laboratory examinations revealed a normal white blood cell count (WBC) and C-reactive protein at 20 mg/L. S. maltophilia was identified from swabbing of the pacemaker pocket. The pacemaker and leads were removed. Culture of the pacemaker leads revealed S. maltophilia, and repeated blood cultures remained sterile. The susceptibility testing revealed resistance to ticarcillinclavulanic acid, cephalosporins, carbapenems, ofloxacin, and aminoglycosides. Intravenous trimethoprim-sulfamethoxazole

∗ Corresponding author. Service des maladies infectieuses et tropicales, hôpital Nord, 42055 Saint-Etienne cedex 2, France. E-mail address: [email protected] (E. Botelho-Nevers).

http://dx.doi.org/10.1016/j.medmal.2015.01.016 0399-077X/© 2015 Elsevier Masson SAS. All rights reserved.

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Q. Reynaud et al. / Médecine et maladies infectieuses 45 (2015) 95–97

(TMP-SMX) 800 mg 4 times a day plus moxifloxacin 400 mg bid were initiated. A transthoracic echocardiography showed 3 mobile vegetations (< 10 mm each) in the right atrium, compatible with S. maltophilia endocarditis of the tricuspid native valve. No trans-esophageal echocardiography was performed due to the patient’s bad condition. No septic pulmonary embolism was observed with lung Technetium 99 m scintigraphy. The patient remained apyretic and her laboratory values remained stable. A control transthoracic echocardiography at 7 days revealed an increasing size of the vegetations in the right atrium with septal fixation (13 × 10 mm, 11 × 10 mm, 27 × 27 mm). The patient died suddenly 2 days later, despite appropriate antibiotic treatment, probably because of a massive pulmonary embolism. No autopsy was performed.

Table 1 Review of Stenotrophomonas maltophilia infective endocarditis and pacemaker infective endocarditis characteristics. Comparaison des caractéristiques des cas d’endocardites infectieuses sur pacemaker et non sur pacemaker à Stenotrophomonas maltophilia.

IE risk factors S. maltophilia risk factors Prosthetic valve Complications (infections, thromboembolic events) Case fatality rate

S. maltophilia IE n = 40 [4]

S. maltophilia pacemaker IE n=3

92% NA 65% 81%

33% 33% 0 0

37%

66%a

IE: infective endocarditis; NA: not available. a Within 15 days.

3. Discussion We report a fatal case of S. maltophilia pacemaker endocarditis, reported only twice before [5,6]. The clinical characteristics of infective endocarditis (IE) on implantable cardiac devices have been frequently described [7]. We consider that our patient presented with pacemaker endocarditis, as defined by Athan et al. [7], and not just pacemaker infection despite the negative blood cultures, because tricuspid valve vegetations were observed [7]. However, when vegetation are not detected, positive lead culture alone may be explained by intra-operative contamination and reflect local device infection and not cardiac device-related endocarditis [8]. The specific risk factors for pacemaker endocarditis are repeated surgical procedures associated with the pulse generator or leads, the presence of postsurgical hematoma, surgical drainage of a pocket hematoma, oral corticosteroids, anticoagulant therapy, diabetes mellitus, age, intravenous catheters, neoplasms, thin skin, temporary pacing, and skin diseases [8]. These risk factors are added to those of S. maltophilia infections, usually those of opportunistic infections (admission to an intensive care unit, neutropenia, mechanical ventilation, central venous catheters, recent surgery, and previous therapy with broad spectrum antibiotics). Our patient had several risk factors for pacemaker IE, such as old age, treatment with anticoagulant therapy, and oral corticosteroid intake. No specific risk factor for S. maltophilia was identified but her husband underwent chronic dialysis and could have been a healthy S. maltophilia carrier. In the 2 reported cases, as in our case, S. maltophilia IE occurred a long time after pacemaker implantation, respectively 8 months [5], 17 years [6], and 15 months in our case. This suggested an infection secondary to a transient bacteremia rather an inoculation at the time of implantation [9]. Moreover, the usual risks factors for S. maltophilia infection were lacking in the 3 cases. There was no specific risk for pacemaker endocarditis and S. maltophilia infection in the first reported case of S. maltophilia IE [5]. The second case was a female patient with a history of intermittent chronic antibiotic therapy [6]. None of the 3 patients carried a prosthetic valve, whereas S. maltophilia IE was reported to be associated with prosthetic valves in 65% of cases (26/40 cases reported [4], Table 1). The benefit of combination therapy has not been documented. The 3 patients received

different treatments: 1 was given TMP-SMX plus Amikacin [5], the second was treated with TMP-SMX and minomycin and pazufloxacin [6], and our patient was treated with TMP-SMX and Moxifloxacin. The 3 patients underwent surgical cardiac device removal and 2 of them died within 15 days [5]. Fifteen out of 40 (37.5%) reported patients of S. maltophilia IE died [4] (see Table 1). S. maltophilia pacemaker IE seems to be associated with high rate of morbidity and mortality, and infection control is hard to achieve. The treatment of S. maltophilia pacemaker IE can be challenging because of S. maltophilia’s intrinsic resistance to many antibiotics (beta-lactams, aminoglycosides, carbapenems, and fluoroquinolones). The clinical data is limited, but trimethoprimsulfamethoxazole (TMP-SMX) is the standard of care for S. maltophilia infections. There are no guidelines for this rare infection but we suggest 4 to 6 weeks of effective antibiotherapy, associated with cardiac device removal, as usually suggested for pacemaker IE [9]. Ticarcillin-clavulanic acid has been suggested as an alternative therapeutic agent, although in vitro resistance may reach 54.7% [10]. Other antibiotics, usually effective in vitro against S. maltophilia, include ceftazidime, levofloxacin, moxifloxacin, minocycline, tigecycline, polymyxins, and rifampicin [10]. In the 2 reported cases, the patients were treated with TMP-SMX plus amikacin [5], and TMP-SMX and minomycin plus pazufloxacin [6]. The infection control was achieved only when TMP/SMX was used and the patient underwent surgery [4,5].

4. Conclusion S. maltophilia pacemaker infective endocarditis is rare and associated with a high mortality rate because of S. maltophilia’s broad resistance to antimicrobials and the patient’s underlying conditions. A prolonged treatment with TMP-SMX is the standard of care. Infection control may be achieved only after complete pacemaker removal. Patients presenting with S. maltophilia infection risks should be promptly identified, and managed rapidly and adequately to decrease the mortality rate.

Q. Reynaud et al. / Médecine et maladies infectieuses 45 (2015) 95–97

Contribution of authors All the authors participated in the patient’s management. QR, EW, FS reviewed the literature. QR wrote the first version of the article, AGB, FL, and EBN proofread the article. Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. References [1] Looney WJ, Narita M, Mühlemann K. Stenotrophomonas maltophilia: an emerging opportunist human pathogen. Lancet Infect Dis 2009;9:312–23. [2] Nicodemo AC, Paez JIG. Antimicrobial therapy for Stenotrophomonas maltophilia infections. Eur J Clin Microbiol Infect Dis 2007;26:229–37. [3] Paez JIG, Costa SF. Risk factors associated with mortality of infections caused by Stenotrophomonas maltophilia: a systematic review. J Hosp Infect 2008;70:101–8. [4] Carrillo-Córdova JR, Amezcua-Guerra LM. Autoimmunity as a possible predisposing factor for Stenotrophomonas maltophilia endocarditis. Arch Cardiol México 2012;82:204–7.

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[5] Rostoff P, Paradowski A, Gackowski A, et al. Stenotrophomonas maltophilia pacemaker endocarditis in a patient with d-transposition of the great arteries after atrial switch procedure. Int J Cardiol 2010;145: e92–5. [6] Takigawa M, Noda T, Kurita T, et al. Extremely late pacemakerinfective endocarditis due to Stenotrophomonas maltophilia. Cardiology 2008;110:226–9. [7] Athan E, Chu VH, Tattevin P, et al. Clinical characteristics and outcome of infective endocarditis involving implantable cardiac devices. JAMA 2012;307:1727–35. [8] Habib G, Hoen B, Tornos P, et al. Guidelines on the prevention, diagnosis, and treatment of infective endocarditis (new version 2009): the Task Force on the Prevention, Diagnosis, and Treatment of Infective Endocarditis of the European Society of Cardiology (ESC). Endorsed by the European Society of Clinical Microbiology and Infectious Diseases (ESCMID) and the International Society of Chemotherapy (ISC) for Infection and Cancer. Eur Heart J 2009;30:2369–413. [9] Baddour LM, Epstein AE, Erickson CC, et al. Update on cardiovascular implantable electronic device infections and their management: a scientific statement from the American Heart Association. Circulation 2010;121:458–77. [10] Falagas ME, Valkimadi P-E, Huang Y-T, et al. Therapeutic options for Stenotrophomonas maltophilia infections beyond co-trimoxazole: a systematic review. J Antimicrob Chemother 2008;62:889–94.

Late Stenotrophomonas maltophilia pacemaker infective endocarditis.

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