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Burkholderia cepacia infective endocarditis of a double-layer transcatheter pulmonary valve

Published online by Cambridge University Press:  13 October 2025

Kenan ÖztürkerOpen the ORCID record for Kenan Öztürker[Opens in a new window],
Eylem Tunçer,
Fatih Tomrukçu and
Mehmet Emirhan Işık
Kenan Öztürker*
Affiliation:
Department of Pediatric Cardiovascular Surgery, Ministry of Health Koşuyolu High Specialisation Training and Research Hospital, Istanbul, Turkiye
Eylem Tunçer
Affiliation:
Department of Pediatric Cardiovascular Surgery, Ministry of Health Koşuyolu High Specialisation Training and Research Hospital, Istanbul, Turkiye
Fatih Tomrukçu
Affiliation:
Department of Pediatric Cardiovascular Surgery, Ministry of Health Koşuyolu High Specialisation Training and Research Hospital, Istanbul, Turkiye
Mehmet Emirhan Işık
Affiliation:
Deparment of Infectious Diseases, Kartal Kosuyolu Training and Research Hospital, Istanbul, Turkiye
*
Corresponding author: Kenan Öztürker; Email: kenanozturker@gmail.com
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Abstract

Burkholderia cepacia is a rare cause of prosthetic valve endocarditis. We report an 18-year-old male with Tetralogy of Fallot and two sequential transcatheter pulmonary valves (Melody and Myval), presenting with persistent bacteraemia unresponsive to antibiotics. Surgical explantation of both valves with homograft replacement and tricuspid repair achieved complete recovery, emphasising surgery in refractory infections.

Keywords

Burkholderia cepaciapulmonary valve endocarditisprosthetic valvehomograftMelody valveMyvalCHD

Information

Type
Case Report
Information
Cardiology in the Young , Volume 35 , Issue 11 , November 2025, pp. 2373 - 2375
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Copyright
© The Author(s), 2025. Published by Cambridge University Press

Introduction

Burkholderia cepacia is an opportunistic, multidrug-resistant Gram-negative bacillus that rarely causes infective endocarditis. Reference Green, McKenzie and Wirth1 When involved, it is typically seen in immunocompromised individuals, intravenous drug users, or patients with indwelling catheters or prosthetic cardiac devices. Prosthetic pulmonary valve endocarditis is particularly rare, and there is a limited literature describing B. cepacia as the causative organism. Reference Haddad, Andraws and Gonzalez2–Reference Singh, Kumar and Arora4 This report presents a unique case of B. cepacia prosthetic valve endocarditis involving both Melody and Myval transcatheter pulmonary valves, successfully managed with surgical resection and homograft implantation.

Case presentation

An 18-year-old male with a history of Tetralogy of Fallot, initially operated at 10 months of age with ventricular septal defect closure and transannular patch repair, underwent Melody valve implantation at age 14 for free pulmonary regurgitation. At age 16, due to progressive stenosis, a Myval valve was implanted within the Melody valve. Antibiotic prophylaxis was administered prior to both catheter interventions according to institutional protocol. During four months of persistent fever, the patient was initially followed as an outpatient with oral antibiotic therapy for approximately one month. As blood cultures continued to yield B. cepacia, he was subsequently hospitalised in another centre for three months and treated with intravenous antibiotics. Despite this prolonged course, bacteraemia persisted until referral to our institution. Upon referral to our centre, transthoracic echocardiography revealed a 61 mmHg maximum gradient across the pulmonary valve, a 7 × 9 mm vegetation on the posterior leaflet of the Myval valve (Figure 1), and moderate-to-severe tricuspid regurgitation. Ceftazidime-avibactam therapy was initiated. After two weeks of targeted antibiotic treatment, the patient underwent surgical intervention. Intraoperatively, the two nested prosthetic valves (Figure 2) together with the adjacent pulmonary artery segment were removed en bloc. A 21-mm pulmonary homograft was implanted, with the proximal anastomosis constructed onto the right ventricle and the distal end sutured to the pulmonary artery bifurcation. During four months of persistent fever, the patient was initially followed as an outpatient with oral antibiotic therapy for approximately one month. As blood cultures continued to yield B. cepacia, he was subsequently hospitalised in another centre for three months and treated with intravenous antibiotics. Despite this prolonged course, bacteraemia persisted until referral to our institution. Upon referral to our centre, transthoracic echocardiography revealed a 61 mmHg maximum gradient across the pulmonary valve, a 7 × 9 mm vegetation on the posterior leaflet of the Myval valve, and moderate-to-severe tricuspid regurgitation. Ceftazidime-avibactam therapy was initiated. After two weeks of targeted antibiotic treatment, the patient underwent surgical intervention. Intraoperatively, the two nested prosthetic valves together with the adjacent pulmonary artery segment were removed en bloc. A 21-mm pulmonary homograft was implanted, with the proximal anastomosis constructed onto the right ventricle and the distal end sutured to the pulmonary artery bifurcation.

Figure 1.

Transthoracic echocardiographic image demonstrating a large mobile vegetation (7 × 9 mm) attached to the posterior leaflet of the Myval transcatheter pulmonary valve.

Figure 2.

Intraoperative photograph showing the explanted transcatheter Melody and Myval valves removed en bloc, with visible thrombotic and infected material on the prostheses.

Postoperatively, ceftazidime-avibactam was continued for four weeks. Blood cultures became sterile, and the patient was discharged in good health. At 3-month follow-up, the patient remained asymptomatic with sterile blood cultures and normal homograft function. No long-term suppressive antibiotic therapy was required. At 6-month and 1-year follow-up, the patient remained in good health with sterile blood cultures and normal homograft function.

Discussion

B. cepacia is a non-fermenting, aerobic Gram-negative bacillus commonly found in soil and water, notorious for its intrinsic resistance to multiple antibiotics. While it rarely causes infective endocarditis, when it does, it often involves prosthetic valves or indwelling cardiac devices, and its management poses significant challenges due to limited antimicrobial options and a high risk of treatment failure.Reference Mylonakis and Calderwood5 In our case, the patient had a history of complex CHD with two sequential transcatheter pulmonary valve implantations: a Melody valve followed by a Myval valve-in-valve procedure. The occurrence of infective endocarditis involving a double-layered transcatheter valve is extremely rare, and, to our knowledge, no prior reports have described B. cepacia endocarditis in this context. The persistent bacteraemia despite prolonged antibiotic therapy highlights the pathogen’s resilience and the inadequacy of medical management alone in such infections. Although ceftazidime-avibactam has shown promising activity against multidrug-resistant Gram-negative bacteria, surgical intervention remains crucial in eradicating the infection source, especially in the setting of prosthetic valve involvement. The surgical approach in our patient— en bloc removal of the two nested prostheses with implantation of a 21-mm homograft and tricuspid valve repair—proved to be life-saving. The homograft remained well-functioning at 1-year follow-up. In our case, the patient had a history of complex CHD with two sequential transcatheter pulmonary valve implantations: a Melody valve followed by a Myval valve-in-valve procedure. The occurrence of infective endocarditis involving a double-layered transcatheter valve is extremely rare, and, to our knowledge, no prior reports have described B. cepacia endocarditis in this context. The persistent bacteraemia despite prolonged antibiotic therapy highlights the pathogen’s resilience and the inadequacy of medical management alone in such infections. Although ceftazidime-avibactam has shown promising activity against multidrug-resistant Gram-negative bacteria, surgical intervention remains crucial in eradicating the infection source, especially in the setting of prosthetic valve involvement. The surgical approach in our patient—en bloc removal of the two nested prostheses with the implantation of a 21-mm homograft and tricuspid valve repair—proved to be life-saving. The homograft remained well-functioning at 1-year follow-up.

This case underlines the importance of early surgical consultation in patients with persistent bacteraemia and prosthetic valve involvement, even when the pathogen is rarely associated with endocarditis. Reference Baddour, Wilson and Bayer6 Furthermore, this report contributes to the growing but still limited body of literature regarding B. cepacia endocarditis and reinforces the need for heightened suspicion in patients with indwelling prosthetic material who fail to respond to antibiotics. Furthermore, this report contributes to the growing but still limited body of literature regarding B. cepacia endocarditis and reinforces the need for heightened suspicion in patients with indwelling prosthetic material who fail to respond to antibiotics.

Conclusion

This case illustrates a rare but severe instance of B. cepacia prosthetic valve endocarditis involving sequentially implanted transcatheter pulmonary valves. The persistence of multidrug-resistant bacteraemia despite prolonged antibiotic therapy underscores the limitations of medical treatment alone in such complex infections. Surgical removal of infected prosthetic material, even in high-risk CHD patients, remains essential for definitive management. Early recognition and a multidisciplinary approach are critical to improving outcomes in similar cases.

References

Green, SL, McKenzie, WR, Wirth, DF. Burkholderia cepacia complex: emerging pathogen in patients with cystic fibrosis and other chronic diseases. Clin Microbiol Rev 2021; 34: e00044–e00019.Google Scholar
Haddad, F, Andraws, R, Gonzalez, MD et al. Burkholderia cepacia prosthetic valve endocarditis: case report and review of literature. Clin Infect Dis 2000; 31: 1014–1015.Google Scholar
Aggarwal, N, Gupta, D, Kumar, A. Fatal early prosthetic valve endocarditis due to Burkholderia cepacia. J Heart Valve Dis 2005; 14: 519–521.Google ScholarPubMed
Singh, M, Kumar, S, Arora, V et al. Successful treatment of early prosthetic valve endocarditis due to Burkholderia cepacia with combined surgical and antimicrobial therapy. J Med Sci Res 2013; 4: 180–183.Google Scholar
Mylonakis, E, Calderwood, SB. Infective endocarditis in adults. N Engl J Med 2001; 345: 1318–1330.CrossRefGoogle ScholarPubMed
Baddour, LM, Wilson, WR, Bayer, AS et al. Infective endocarditis in adults: diagnosis, antimicrobial therapy, and management of complications. Circulation 2015; 132: 1435–1486.CrossRefGoogle ScholarPubMed
Figure 0

Figure 1.Transthoracic echocardiographic image demonstrating a large mobile vegetation (7 × 9 mm) attached to the posterior leaflet of the Myval transcatheter pulmonary valve.

Figure 1

Figure 2.Intraoperative photograph showing the explanted transcatheter Melody and Myval valves removed en bloc, with visible thrombotic and infected material on the prostheses.