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Table of Contents
CASE REPORT
Year : 2018  |  Volume : 19  |  Issue : 4  |  Page : 150-151  

Tropheryma Whipplei endocarditis: Case report and literature review


1 Department of Cardiology, The Canberra Hospital; College of Medicine, Biology and Environment, Australian National University, Canberra, Australian Capital Territory, Australia
2 Department of Infectious Diseases, The Canberra Hospital, Canberra, Australia
3 Department of Infectious Diseases, The Canberra Hospital; College of Medicine, Biology and Environment, Australian National University, Canberra, Australian Capital Territory, Australia
4 Department of Neurology, The Canberra Hospital, Canberra, Australia

Date of Web Publication15-Apr-2019

Correspondence Address:
Dr. Mohammad Paymard
Department of Cardiology, The Canberra Hospital, Canberra, Australian Capital Territory
Australia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/HEARTVIEWS.HEARTVIEWS_112_18

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   Abstract 


We describe a young man who initially presented with stroke and febrile illness. He was eventually diagnosed with Tropheryma whipplei endocarditis. This is a very rare condition and to the best of our knowledge, this is the first documented case of T. whipplei endocarditis in Australia and New Zealand regions. This report aims to increase awareness of clinicians of this very rare but potentially treatable condition. It is reasonable to exclude T. whipplei endocarditis when dealing with high-risk patients who are suspected for “culture-negative” endocarditis.

Keywords: Culture-negative endocarditis, infective endocarditis, Tropheryma whipplei endocarditis


How to cite this article:
Paymard M, Sukumaran V, Senanayake S, Watson A, Das C, Abhayaratna WP. Tropheryma Whipplei endocarditis: Case report and literature review. Heart Views 2018;19:150-1

How to cite this URL:
Paymard M, Sukumaran V, Senanayake S, Watson A, Das C, Abhayaratna WP. Tropheryma Whipplei endocarditis: Case report and literature review. Heart Views [serial online] 2018 [cited 2019 Jun 24];19:150-1. Available from: http://www.heartviews.org/text.asp?2018/19/4/150/256232




   Introduction Top


Whipple's disease (WD) is a rare chronic multisystemic infectious disease that very rarely causes infective endocarditis (IE).[1],[2] The presenting symptoms are usually nonspecific, and therefore, the accurate diagnosis is often delayed.


   Case Presentation Top


A 35-year-old roof plumber presented with 1-day history of malaise, vomiting, confusion, and dysphasia with mild right-sided weakness and numbness. One-week prior, he had an infected tooth. Four months earlier, he experienced transient right-sided sensorimotor deficits. He had intermittent bilateral wrist arthralgia for 2 years. He denied intravenous illicit drug use.

On admission, he was febrile (38.2°C), in sinus rhythm, and had a diastolic murmur on auscultation. There were no stigmata of IE. Blood tests showed raised white cell count 12.9 × 109/L (4.0–11.0 × 109/L) and C-reactive protein 56 mg/L (<5 mg/L). Magnetic resonance imaging brain confirmed left parietal ischemic stroke.

A large lesion on the right coronary cusp (RCC) of the aortic valve associated with severe aortic regurgitation was documented on transthoracic and subsequent transoesophageal echocardiography. The working diagnosis was ischemic stroke due to IE, and he was treated with intravenous ceftriaxone and vancomycin. Two weeks later, he underwent bioprosthetic aortic valve replacement and mitral valve debridement. Large vegetation on the RCC of the aortic valve (30 mm × 20 mm × 2 mm) and two small vegetations on the anterior leaflet of the mitral valve were found intraoperatively.

Blood cultures, serology for hepatitis B and C and HIV, and screening for blood culture-negative endocarditis (BCNE) and vasculitis were negative. Five weeks postadmission, the result of his 16S rRNA polymerase chain reaction (PCR) from explanted valves confirmed Whipple's endocarditis. Valve histopathology showed macrophages with periodic acid-Schiff-positive organisms [Figure 1]. Electron microscopy findings also confirmed the diagnosis. Vancomycin was ceased as soon as Whipple's endocarditis was confirmed by 16S rRNA polymerase chain reaction (PCR). However, the patient continued with intravenous ceftriaxone therapy for another 2 weeks to complete a total of 7-week course before it was ceased and replaced by Bactrim DS one tablet twice daily, which was continued for 12 months. He remains well with excellent speech recovery and no residual weakness.
Figure 1: Foamy macrophages containing characteristic inclusion Tropheryma whipplei particle (periodic acid–Schiff stain)

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   Discussion Top


This is the first documented case of IE due to WD in Australia. WD is a rare chronic multisystemic infectious disease with a prevalence of less than 1:1,000,000 that preferentially affects middle-aged men.[1] The classic manifestations of WD are weight loss, abdominal discomfort, diarrhea, malabsorption, and arthralgia.[1] WD is caused by T. whipplei which is found in the general environment and sewage water as well as healthy individuals' stool.[1],[3] The mode of transmission has been postulated to be fecal-oral route.[1] Our patient possibly had occupational exposure to the organism while working for years as a plumber.

T. whipplei endocarditis is a very rare condition with unknown incidence.[2] The aortic valve is most commonly involved.[4] Approximately 26% of patients are complicated by ischemic stroke and up to 75% report arthralgia.[4] The diagnosis is made based on tissue 16S rRNA PCR, histopathology, immunohistochemistry, and electron microscopy. The recommended antibiotic regimen includes 2–4 weeks of intravenous ceftriaxone, penicillin, or meropenem followed by 1 year of oral trimethoprim-sulfamethoxazole or doxycycline.[5] It has been proposed that patients be monitored for early relapse or reinfection with PCR on saliva and stool 3 and 6 months after commencement of treatment and annually thereafter.[6]


   Conclusion Top


T. whipplei endocarditis is a rare but potentially treatable condition. Given the specific diagnostic and therapeutic approach, clinicians should have a low threshold to investigate for T. whipplei in high-risk patients with BCNE.

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.
Fenollar F, Puéchal X, Raoult D. Whipple's disease. N Engl J Med 2007;356:55-66.  Back to cited text no. 1
    
2.
Geissdörfer W, Moos V, Moter A, Loddenkemper C, Jansen A, Tandler R, et al. High frequency of Tropheryma whipplei in culture-negative endocarditis. J Clin Microbiol 2012;50:216-22.  Back to cited text no. 2
    
3.
Schöniger-Hekele M, Petermann D, Weber B, Müller C. Tropheryma whipplei in the environment: Survey of sewage plant influxes and sewage plant workers. Appl Environ Microbiol 2007;73:2033-5.  Back to cited text no. 3
    
4.
Fenollar F, Célard M, Lagier JC, Lepidi H, Fournier PE, Raoult D, et al. Tropheryma whipplei endocarditis. Emerg Infect Dis 2013;19:1721-30.  Back to cited text no. 4
    
5.
Feurle GE, Junga NS, Marth T. Efficacy of ceftriaxone or meropenem as initial therapies in Whipple's disease. Gastroenterology 2010;138:478-86.  Back to cited text no. 5
    
6.
Gruber JR, Sarro R, Delaloye J, Surmely JF, Siniscalchi G, Tozzi P, et al. Tropheryma whipplei bivalvular endocarditis and polyarthralgia: A case report. J Med Case Rep 2015;9:259.  Back to cited text no. 6
    


    Figures

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