Tropheryma whipplei bivalvular endocarditis and polyarthralgia: a case report

Abstract

Introduction: Tropheryma whipplei infection should be considered in patients with suspected infective endocarditis with negative blood cultures. The case (i) shows how previous symptoms can contribute to the diagnosis of this illness, and (ii) elucidates current recommended diagnostic and therapeutic approaches to Whipple's disease.

Case presentation: A 71-year-old Swiss man with a past history of 2 years of diffuse arthralgia was admitted for a possible endocarditis with severe aortic and mitral regurgitation. Serial blood cultures were negative. Our patient underwent replacement of his aortic and mitral valve by biological prostheses. T. whipplei was documented by polymerase chain reactions on both removed valves and on stools, as well as by valve histology. A combination of hydroxychloroquine and doxycycline was initiated as lifetime treatment followed by the complete disappearance of his arthralgia.

Conclusions: This case report underlines the importance of considering T. whipplei as a possible causal etiology of blood culture-negative endocarditis. Lifelong antibiotic treatment should be considered for this pathogen (i) due to the significant rate of relapses, and (ii) to the risk of reinfection with another strain since these patients likely have some genetic predisposition.

Fig. 1

Transoesophageal echocardiographic views of the aortic and mitral valve. a Visualization of the perforation of aortic cusp (arrow). b Visualization of the large regurgitant jet through perforated aortic cusp (arrow). c Three-dimensional transoesophageal echocardiography of mitral valve. d Three-dimensional transoesophageal echocardiography: visualization of regurgitant color Doppler jet through mitral valve (arrows)

Fig. 2

Transoesophageal echocardiographic views of the aortic and mitral valve. a Visualization of subacute vegetations on posterior leaflet and chordae tendinae of mitral valve (arrows). b Visualization of a vegetation on aortic valve (arrow)

Fig. 3

Histopathology findings: gross appearance of T. whipplei-infected native mitral valve specimen. a Unremarkable atrial side of valvular leaflet. b Transverse section of valve harboring voluminous bright yellow vegetations (arrowheads), up to 6 mm exclusively confined to the ventricular side of the valve and chordae tendinae

Fig. 4

Histopathology findings: microscopic appearance of T. whipplei-infected native mitral valve specimen. a On low-power view, dense foamy macrophage infiltrate within the valve (hematoxylin and eosin, original magnification × 200). b On high-power view, macrophages with light gray and granular cytoplasm (hematoxylin and eosin, original magnification × 400). c The cytoplasm is filled with periodic acid-Schiff-positive material (original magnification × 200). d The cytoplasmatic periodic acid-Schiff-positive material is also strongly positive with Warthin-Starry stain (original magnification × 400)