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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 10  |  Issue : 4  |  Page : 147-149

Brucella endocarditis of bicuspid aortic valve


1 Department of Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
2 Department of Cardiology, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

Date of Submission02-May-2021
Date of Decision01-Sep-2021
Date of Acceptance06-Oct-2021
Date of Web Publication24-Dec-2021

Correspondence Address:
Dr. Debasish Das
Department of Cardiology, All India Institute of Medical Sciences, Bhubaneswar - 751 019, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcpc.jcpc_29_21

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  Abstract 


We present a rare case of Brucella endocarditis in a case of the bicuspid aortic valve in a 51-year-old male presenting with pyrexia of unknown origin for the last 2 months. Infective endocarditis caused by Brucella melitensis is rare to encounter in routine clinical practice. Although we routinely think of Mycoplasma, Legionella, and Coxiella in the cases of blood culture-negative endocarditis, one should think of Brucella melitensis and Scrub typhus as the last arrow to reach the final etiology of infective endocarditis in the subset of diseased aortic or mitral valve. We successfully treated the patient with a combined regimen of rifampicin, gentamicin, and doxycycline therapy; during follow-up, the vegetation of the aortic valve was shrunken and calcified and the patient was asymptomatic.

Keywords: Aortic, bicuspid, Brucella, endocarditis, valve


How to cite this article:
Devi S, Kar N, Sahoo D, Das T, Acharya D, Das D. Brucella endocarditis of bicuspid aortic valve. J Clin Prev Cardiol 2021;10:147-9

How to cite this URL:
Devi S, Kar N, Sahoo D, Das T, Acharya D, Das D. Brucella endocarditis of bicuspid aortic valve. J Clin Prev Cardiol [serial online] 2021 [cited 2022 Jan 19];10:147-9. Available from: https://www.jcpconline.org/text.asp?2021/10/4/147/333702




  Introduction Top


Brucella endocarditis accounts for 2% of brucellosis with a high mortality rate. Heart failure is the leading cause of death in Brucella endocarditis. The bacteria get implanted on the valve with congenital abnormality like bicuspid aortic valve as in our case or the damaged valve of rheumatic heart disease. Brucella endocarditis predominantly involves the aortic valve. Brucellosis predominates in the Middle East and Mediterranean regions.[1] This report suggests that the combination antibiotic therapy recovers Brucella endocarditis with a high success rate.


  Case Report Top


A 51-year-old male came to the medicine outpatient department with intermittent low-to-moderate grade fever for 2 months. It was associated with low backache, breathlessness on exertion with occasional dry cough, and generalized weakness. He had empirically taken the antitubercular regimen for 1 month and showed partial response for few days. On presentation, his pulse rate was 76 per minute, regular, high volume. His blood pressure was 126/58 mmHg. The general survey showed mild pallor, Grade I clubbing, bipedal pitting edema, engorged, and pulsatile jugular venous pressure (11 cm of water). Cardiovascular system examination revealed apex shifted to THE left sixth intercostal space along the midclavicular line, hyperdynamic. Ejection systolic murmur was heard at the aortic area radiating to the apex, associated with carotid thrill. Grade 4 early diastolic murmur was heard over the neoaortic area. Respiratory, abdominal, and neurological system examinations were normal.

Blood investigations revealed hemoglobin 9 g/dl with mixed microcytic hypochromic and normocytic normochromic picture on the peripheral smear. Erythrocyte Sedimentation Rate (ESR) was 20 mm/h. The renal function test was within normal range. The liver function test revealed that hypoalbuminemia (2.4 g/dl) with altered albumin globulin ratio (0.53) and mild elevated alkaline phosphatase (206 U/l) were observed. Twelve-lead Electrocardiogram (ECG) demonstrated normal sinus rhythm. Routine urine microscopy was normal. Contrast Enhanced Computed Tomography(CECT) abdomen revealed mild splenomegaly with wedge-shaped splenic infarct in the upper and mid pole. Non Contrast Computed Tomography (NCCT) thorax delineated fibrobronchiectatic changes in right middle lobe, subsegmental atelectasis at the left upper lobe, and centrilobular nodules with the tree in bud pattern in the right lower lobe with few mediastinal lymph nodes. Transthoracic two-dimensional (2D) echocardiography revealed the presence of bicuspid aortic valve with moderate-to-severe aortic regurgitation (AR) with moderate aortic stenosis. Two sets of blood cultures did not show any growth. Escherichia coli (105 CFU) appeared in urine culture, but the fever did not subside despite optimal antibiotics as per sensitivity pattern.

Because of high clinical suspicion, transesophageal echocardiography (TEE) was performed. It demonstrated a bicuspid aortic valve [Figure 1] with large vegetation of 9 mm × 7 mm in size attached to the right coronary cusp [Figure 2] and [Figure 3] causing moderate AR with two eccentric jets [Figure 4]. 3D TEE also revealed the presence of large vegetation attached to the right coronary cusp [Figure 5]. Repeat sets of blood cultures also did not yield any growth. High-grade fever persisted even with the injection of empirical antibiotics: vancomycin, gentamicin, and meropenem. Considering the possibility of culture-negative organisms in the background of protracted fever with low backache and partial response with antitubercular therapy, IgM Brucella antibody was sent which came strongly positive. The patient was started on gentamicin, doxycycline, and rifampicin combination. The patient improved with treatment, became afebrile, and ultimately discharged on request to continue treatment on a domiciliary basis.
Figure 1: Transesophageal echocardiography showing bicuspid aortic valve with raphe

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Figure 2: Transesophageal echocardiography showing large vegetation attached to right coronary cusp

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Figure 3: Vegetation of 9 mm × 7 mm attached to right coronary cusp

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Figure 4: Two regurgitant jets secondary to leaflet endocarditis

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Figure 5: Three-dimensional transesophageal echocardiography showing large vegetation in right coronary cusp

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  Discussion Top


Brucellae are the most common cause of zoonotic infection across the world.[2] They are Gram-negative Coccobacilli with three species: Brucella melitensis, Brucella abortus, and Brucella suis which cause human infection. Brucellosis is also known as Mediterranean fever, Malta fever, gastric remittent fever, and undulant fever. Brucellosis is transmitted to humans through secretions, aerosol, or intake of unpasteurized dairy products. Brucellosis is endemic in the Middle East, Mediterranean, and South Asia including India and South America.

Brucellosis often manifests with nonspecific symptoms for which many often diagnosis becomes difficult. Brucella may involve the gastrointestinal system, hepatobiliary system, nervous, musculoskeletal, genitourinary, skin, and hematological system. Cardiovascular involvement accounts for 80% of mortality in Brucella infection.[3] Median duration of symptoms in brucellosis is for 3–5 months. The mean age of presentation of brucellosis is 55 years and is predominant in males.[4] The most common presenting symptom in brucellosis is fever which is seen in 60% of cases. The aortic valve is most commonly affected in 75% of cases. Multivalvular Brucella endocarditis affecting both the mitral and aortic valve is seen in 8.3% of cases. If Brucella endocarditis of diseased valve in the form of bicuspid valve or rheumatic valve is considered, it has a predilection of aortic valve involvement than mitral valve, but native valve Brucella endocarditis occurs more over the mitral valve. The myocardial abscess occurs in up to 43% of cases of brucellosis.[5] The myocardial abscess is more common in Brucella endocarditis than due to any other causative organism.[6] Infective endocarditis and heart failure account for the highest cause of mortality in brucellosis, which may be up to 30% in endemic and untreated or maltreated cases.[7]

Diagnostic methods for Brucella endocarditis include blood culture, serology, immunohistology, and PCR of infected material. Although blood culture is the gold standard, it has a low diagnostic yield of 22.2% as described by Esmailpour et al.[8] Serological tests in the form of Brucella IgG and IgM have a higher sensitivity; Wrights agglutination titer of 1:160 is very specific and sensitive.

European Society of Cardiology guideline recommends the combined use of doxycycline, cotrimoxazole, and rifampicin for 3 months with optimal aminoglycosides for 2 months.[9] There is no role of antithrombotic medications in the acute phase of endocarditis unless coexistent left atrial thrombus, atrial fibrillation, or venous thromboembolism is there. Most people recommend an early surgical approach in Brucella endocarditis due to a high degree of tissue destruction with a high rate of recurrence. Keshtkar-Jahromi et al.[10] described the beneficial effect of combined medical and surgical approach in Brucella endocarditis substantially reducing the mortality of 32.7% in medical treatment alone group to 6.7% in combined medical and surgical arm across 308 cases of Brucella endocarditis.


  Conclusion Top


Our case is an extremely rare presentation of Brucella endocarditis in a bicuspid aortic valve causing moderate AR where vegetation was well picked up by TEE and was successfully treated with doxycycline, rifampicin, and gentamycin combination therapy. Blood culture-negative endocarditis is not always due to conventional Mycoplasma, Legionella, or Coxiella; Brucella endocarditis can be a possibility even.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sahin N, Kabukcu HK, Salman C, Celikbilek G, Titiz TA, Turkay C. Brucella endocarditis of the aortic valve. Eur J Anaesthesiol 2007;24:37.  Back to cited text no. 1
    
2.
Pappas G, Papadimitriou P, Akritidis N, Christou L, Tsianos EV. The new global map of human brucellosis. Lancet Infect Dis 2006;6:91-9.  Back to cited text no. 2
    
3.
Memish Z, Mah MW, Al Mahmoud S, Al Shaalan M, Khan MY. Brucella bacteremia: Clinical and laboratory observations in 160 patients. J Infect 2000;40:59-63.  Back to cited text no. 3
    
4.
Şimşek-Yavuz S, Şensoy A, Kaşıkçıoğlu H, Çeken S, Deniz D, Yavuz A, et al. Infective endocarditis in Turkey: Aetiology, clinical features, and analysis of risk factors for mortality in 325 cases. Int J Infect Dis 2015;30:106-14.  Back to cited text no. 4
    
5.
Dean AS, Crump L, Greter H, Hattendorf J, Schelling E, Zinsstag J. Clinical manifestations of human brucellosis: A systematic review and meta-analysis. PLoS Negl Trop Dis 2012;6:e1929.  Back to cited text no. 5
    
6.
Peery TM, Belter LF. Brucellosis and heart disease. II. Fatal brucellosis: A review of the literature and report of new cases. Am J Pathol 1960;36:673-97.  Back to cited text no. 6
    
7.
al-Harthi SS. The morbidity and mortality pattern of Brucella endocarditis. Int J Cardiol 1989;25:321-4.  Back to cited text no. 7
    
8.
Esmailpour N, Borna S, Nejad MR, Badie SM, Badie BM, Hadadi A. Brucella endocarditis: A report from Iran. Trop Doct 2010;40:47-9.  Back to cited text no. 8
    
9.
Habib G, Lancellotti P, Antunes MJ, Bongiorni MG, Casalta JP, Zotti FD, et al. 2015 ESC Guidelines for the management of infective endocarditis: The task force for the management of infective endocarditis of the European Society of Cardiology (ESC) Endorsed by: European Association for Cardio-Thoracic Surgery (EACTS), the European Association of Nuclear Medicine (EANM). Eur Heart J 2015;36:3075-128.  Back to cited text no. 9
    
10.
Keshtkar-Jahromi M, Razavi SM, Gholamin S, Keshtkar-Jahromi M, Hossain M, Sajadi MM. Medical versus medical and surgical treatment for Brucella endocarditis. Ann Thorac Surg 2012;94:2141-6.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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