2022 Volume 86 Issue 11 Pages 1789-
A 25-year-old woman was prescribed antibiotic treatment for possible infective endocarditis (IE), but her symptoms of chest discomfort and intermittent headache were refractory during the 2 years preceding the current admission. Based on clinical symptoms of renal hypertension, multiple lacunar infarcts and purpura, and positive lupus anticoagulant testing, a diagnosis of primary antiphospholipid syndrome (APS) was made. Blood culture suggested Corynebacterium positive.
Transesophageal echocardiography (TEE) demonstrated vegetations located at the coaptation point of the mitral valve, and those associated with the anterior mitral leaflet (AML) were hyperreflectant (Figure A; white arrow), which cardiac magnetic resonance confirmed as fibrosis (Figure E). The vegetations associated with the posterior mitral leaflet (PML) appeared cystic and hypoechoic (Figure A; blue arrow) with blood flow through the ruptured cystic structure (Figure B). Three-dimensional TEE demonstrated vegetations during diastole (Figure C) and a prolapsed PML during systole (Figure D).
Two-dimensional (A), color Doppler (B) and three-dimensional (C,D) transesophageal echocardiography. Cardiac magnetic resonance at T2-mapping and late gadolinium enhancement (LGE) (E). Intraoperative findings of the mitral valve (F). Pathology of the anterior mitral leaflet (AML) (G, H&E ×40; H, H&E ×400) and posterior mitral leaflet (PML) (I, H&E ×400). AO, aorta; LA, left atrium; LV, left ventricle.
Thus, the cardiac manifestation was considered as longstanding Libman-Sacks endocarditis (LSE) superimposed with recent IE. Mitral mechanical prosthetic valve replacement surgery was performed and confirmed the diagnosis (Figure F, Supplementary Movie). Histopathology revealed small remnants on the fibrotic AML with mononuclear cell infiltration (Figure G,H; blue arrows) and without inflammation, consistent with longstanding Libman-Sacks vegetations; the vegetations on the PML were infiltrated by neutrophils and microorganisms (Figure I), which accorded with recent infection.
To our knowledge, this is the first case of LSE and IE vegetations coexisting in a patient. Ignoring secondary infection of LSE developing into IE may affect treatment choice and outcome.
None.
Supplementary Movie. Intraoperative view of the mitral valve.
Please find supplementary file(s);
http://dx.doi.org/10.1253/circj.CJ-22-0326
