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A case of infective endocarditis caused by an uncommon agent Abiotrophia defectiva with atypical manifestations is presented. A 42-year-old woman previously had rheumatic heart disease, presented with the symptoms of fever and chills that resolved within 3 days under antibiotherapy. She was diagnosed with endocarditis due to A. defectiva. Despite culture-directed antibiotics being administered in the first admission, her symptoms and also blood culture growth relapsed 3 weeks later. She was successfully treated with antimicrobial therapy and surgical intervention including aorta and mitral valve replacement. This case demonstrates that A. defectiva should be considered as a causative organism of endocarditis particularly in the presence of atypical symptoms and should be followed up carefully in terms of relapses and complications.