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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 2  |  Issue : 2  |  Page : 121-123

Recurrent infective endocarditis involving bicuspid aortic valve presenting with a rare complication: The deadly kiss


Department of Cardiology, Sri Jayadeva Institute of Cardiovascular Sciences and Research, Bengaluru, Karnataka, India

Date of Web Publication6-Sep-2018

Correspondence Address:
Dr. Arnab Ghosh Chaudhury
122/50 Kumara Krupa JP Nagar, 3rd Main, 3rd Phase, Bengaluru - 560 078, Karnataka,
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiae.jiae_87_17

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  Abstract 

Bicuspid aortic valve, the most common congenital cardiac lesion, is frequently associated with infective endocarditis (IE), and this combination is notoriously associated with increased prevalence of periannular complications. Mitral valve involvement as a “kissing” lesion secondary to aortic valve endocarditis is a very rare entity. Only few case reports are available describing this lesion. We report a case of recurrent IE involving both cusps of bicuspid aortic valve complicated by “kissing” lesion of the anterior mitral leaflet leading to aneurysm formation. The patient presented with severe aortic regurgitation and moderate mitral regurgitation. The patient was treated successfully with surgical aortic valve reconstruction (Ozaki procedure) with mitral valve repair.

Keywords: Aneurysm, anterior mitral leaflet, bicuspid aortic valve, infective endocarditis


How to cite this article:
Chaudhury AG, Bhat P, Manjunath C N, Satvic C M. Recurrent infective endocarditis involving bicuspid aortic valve presenting with a rare complication: The deadly kiss. J Indian Acad Echocardiogr Cardiovasc Imaging 2018;2:121-3

How to cite this URL:
Chaudhury AG, Bhat P, Manjunath C N, Satvic C M. Recurrent infective endocarditis involving bicuspid aortic valve presenting with a rare complication: The deadly kiss. J Indian Acad Echocardiogr Cardiovasc Imaging [serial online] 2018 [cited 2018 Dec 13];2:121-3. Available from: http://www.jiaecho.org/text.asp?2018/2/2/121/240648


  Introduction Top


Bicuspid aortic valve is the most common congenital heart lesion; it is found in 1%8211;2% of the general population. Infective endocarditis (IE) is a well-recognized complication (prevalence 0.4/100,000) and usually presents in the fourth and fifth decades of life.[1] Rarely, it can involve the mitral valve as a kissing lesion or drop lesion. We report a case of 35-year-old male presented with recurrent IE of bicuspid aortic valve with anterior mitral leaflet (AML) aneurysm as a kissing lesion from aortic valve vegetation.


  Clinical Presentation Top


A 35-year-old male patient presented with fever for 15 days in June 2016. He consulted local hospital and was investigated for fever. Echocardiography revealed 8 mm × 3 mm single vegetation on bicuspid aortic valve with moderate aortic regurgitation. Blood culture was negative. Diagnosis of IE was made. The patient was treated medically with injection ceftriaxone and injection gentamicin for 4 weeks and his symptoms improved. On June 15, 2016, the patient developed right parietal lobe bleed and he was managed conservatively.

After almost 1 year of asymptomatic period, on June 1, 2017, the patient presented to our hospital with a history of fever and palpitation of 15-days duration. On examination, the patient had high-volume regular pulse, pulsus bisferiens, and other peripheral signs of aortic runoff. Blood pressure was 130/20/0. Jugular venous pulse was normal. Apical impulse was hyperdynamic. S1 was normal, A2 was soft, and P2 was loud. Long early diastolic murmur was present at neoaortic area. Electrocardiogram revealed sinus rhythm, left atrial enlargement, and left ventricular enlargement. Chest X-ray showed huge cardiomegaly. Trans thoracic and trans esophageal echocardiography revealed severe aortic regurgitation [Figure 1]a and bicuspid aortic valve [Figure 1]c. Two vegetations (9 mm × 4 mm) were seen, one on each cusp of bicuspid aortic valve [Figure 1]c. A pouch-like globular swelling was seen arising from the atrial surface of AML [Figure 1]b and [Figure 1]d. Color Doppler revealed that blood was flowing inside the structure from ventricular side, but there was no opening in the atrial side [Figure 1]d and [Video 1]. Diagnosis of AML aneurysm was made. Vegetation on aortic valve was touching the AML lesion giving rise to the “kissing” lesion [Figure 1]b and [Figure 1]d and [Video 1].
Figure 1: (a) Transthoracic echocardiography apical five-chamber view showing severe aortic regurgitation. (b)Transthoracic echocardiography parasternal short-axis zoomed view showing globular lesion arising from anterior mitral leaflet (yellow arrow) and aortic valve vegetation(white arrow) touching anterior mitral leaflet giving rise to kissing lesion.(c)Transoesophageal echocardiography short-axis view showing bicuspid aortic valve with vegetations (arrow) attached with each cusp. (d) Transesophageal echocardiography two-chamber view showing blood is flowing inside anterior mitral leaflet aneurysm from ventricular side through the narrow mouth (arrow) but not communicating with the atrial cavit

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Blood culture was positive for  Streptococcus anginosus Scientific Name Search tive to ceftriaxone. Globular lesion with central echolucent area, arising from the AML, can be a simple cyst, blood cyst, abscess, and aneurysm with narrow mouth. In our case, we have clearly demonstrated that blood is flowing inside the cavity suggestive of aneurysm. It was proved during surgery as well [Figure 2]a and [Figure 2]b. Injection ceftriaxone 2 g once daily was given for 4 weeks. Due to severe aortic regurgitation and heart failure, the patient was taken for surgery. Aortic valve reconstruction (”Ozaki” procedure) [Figure 2]c and mitral valve repair [Figure 2]d were done. The patient is doing well at present after 6 months following surgery.
Figure 2: (a) Intraoperative picture of bicuspid aortic valve with vegetations (arrow). (b) Intraoperative picture of anterior mitral leaflet aneurysm. Ventricular surface showing narrow mouth (arrow) communicating with left ventricular cavity. (c) Post mitral valve repair. Transoesophageal echocardiography two-chamber view showing no communication between the aneurysm and ventricular cavity. (d) Post aortic valve reconstruction. Transoesophageal echocardiography short-axis view showing “neoaortic valve” with three cusps

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  Discussion Top


Congenital bicuspid aortic valve is common and in most cases remains undetected until infection or calcification supervenes. It is one of the most common cardiac predispositions for native valve endocarditis (NVE).[2] As in NVE, in general, staphylococci (12% of them are coagulase negative) and viridans Streptococci accounted for nearly three-quarters of the cases affecting bicuspid aortic valves.[2] In our case, we isolated S. anginosus as the causative organism. IE is severe and tends to occur in the fourth and fifth decades of life, requiring major surgery in most cases. There is a high incidence of serious complications, specifically heart failure (72%) and aortic root abscesses (30%).[2] Mortality associated with bicuspid aortic valve IE is 28%, according to the study by Skehan et al.[3]

Mitral valve involvement secondary to IE of bicuspid aortic valve is very rare. Three mechanisms are proposed for the spread of infection:[4] First, “drop” lesion, where the jet of aortic regurgitation strikes the AML and embeds organisms causing abscess or aneurysm;[4] second, “kissing” lesion, where aortic valve vegetations prolapse into the left ventricular outflow tract touching the AML;[4] and third, “contiguous” spread of infected tissue from aortic valve to AML.[4] Piper et al.,[5] in their study of 192 cases of aortic valve endocarditis, found 19 cases of (9.9%) of mitral valve kissing vegetations. Patients with kissing lesion had larger aortic valve vegetation size, more embolic events, renal failure, and poorer outcome.[5] Our patient had moderate-sized vegetation (9 mm × 4 mm) and had embolic event in the form of intracerebral hemorrhage, but renal function was normal. Garcia et al.[6] reported a case of aortic ulcer caused by kissing vegetation of aortic valve endocarditis which was successfully treated surgically. Our case also had kissing lesion but involving AML. There was no aortic ulcer in this case. Lee and Tsai [7] described a case of mitral valve aneurysm as kissing lesion, following aortic valve endocarditis. The patient presented with aortic regurgitation, mitral regurgitation, and ischemic stroke.


  Conclusion Top


He was treated conservatively because of high surgical risk. Our patient had a history of intracerebral hemorrhage and moderate mitral regurgitation and was treated surgically with success. Drop lesion of AML caused by Klebsiella pneumoniae was reported from our institute.[8] This time we report this case of “kissing” lesion of AML from IE involving bicuspid aortic valve 8211; “a deadly kiss.” He was treated successfully by surgery (Ozaki procedure) with mitral valve repair. At 6-month follow-up, the patient is doing well.

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.

Acknowledgment

The authors would like to thank Mrs. B. V. Sumangala (senior echo technician, SJIC and R, Bangalore).

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Roberts WC. The congenitally bicuspid aortic valve. A study of 85 autopsy cases. Am J Cardiol 1970;26:72-83.  Back to cited text no. 1
    
2.
Lamas CC, Eykyn SJ. Bicuspid aortic valve 8211; A silent danger: Analysis of 50 cases of infective endocarditis. London, United Kingdom: From St. Thomas' Hospital; Clinical Infectious Diseases 2000;30:336-41.  Back to cited text no. 2
    
3.
Skehan JD, Murray M, Mills PG. Infective endocarditis: Incidence and mortality in the North East Thames region. Br Heart J 1988;59:62-8.  Back to cited text no. 3
    
4.
Kouchoukos N, Blackstone E, Hanley F, Kirklin J. Kirklin/Barratt-Boyes Cardiac Surgery E-Book. 4th ed., Ch. 15. Elsevier; 2012. p. 677.  Back to cited text no. 4
    
5.
Piper C, Hetzer R, Körfer R, Bergemann R, Horstkotte D. The importance of secondary mitral valve involvement in primary aortic valve endocarditis; the mitral kissing vegetation. Eur Heart J 2002;23:79-86.  Back to cited text no. 5
    
6.
Garcia R, Santos A, Almeida J, Pinho P. Kissing ulcer of the aortic wall in a patient with aortic valve endocarditis. J Am Soc Echocardiogr 2009;22:323.e1-2.  Back to cited text no. 6
    
7.
Lee CH, Tsai LM. Transesophageal echocardiographic recognition of mitral valve aneurysm. J Ultrasound Med 2005;24:1141-4.  Back to cited text no. 7
    
8.
Srinivas KH, Sharma R, Agrawal N, Manjunath CN. Silent destruction of aortic and mitral valve by Klebsiella pneumoniae endocarditis. BMJ Case Rep 2013;2013. pii: bcr2013200704.  Back to cited text no. 8
    


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