Whipple’s disease with aortic regurgitation requiring aortic valve replacement

Schneider, T; Salamon-Looijen, M.; Herbay, A. von; Schwerdt, Holger; Weg‐Remers, Susanne; Stallmach, Andreas; Zeitz, Martin

doi:10.1007/bf02771847

Cited by 20 publications

(9 citation statements)

References 14 publications

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“…Since the advent of PCR testing, a total of 11 cases have been reported of T. whipplei endocarditis without (overt) intestinal Whipple's disease (6,8,17,18,23,24,27), including one case report of a xenograft infection with T. whipplei (5). Notably, the majority of patients reported did not fulfill the common criteria for a diagnosis of Whipple's disease, and it has to be questioned whether a diagnosis of isolated cardiac Whipple's disease is adequate.…”

Section: Discussionmentioning

confidence: 99%

“…Until now, a total of 11 cases of PCR-based diagnosis of T. whipplei endocarditis without intestinal Whipple's disease have been reported (5,6,8,17,18,23,24,27), suggesting a possible role of T. whipplei in cardiac disease which has not been considered adequately until now. However, certainty about the uncommon diagnosis in the cases reported to date has been limited to single-test PCR results which were yielded with heterogeneous PCR protocols.…”

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confidence: 99%

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Tropheryma whipplei Infection of an Acellular Porcine Heart Valve Bioprosthesis in a Patient Who Did Not Have Intestinal Whipple's Disease

et al. 2004

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Rare cases of culture-negative infective endocarditis are caused by Tropheryma whipplei, the uncommon bacterium of Whipple's disease. We evaluated an 80-year-old woman with valvular heart disease but without intestinal Whipple's disease. The diagnosis of aortic valve xenograft culture-negative infection with T. whipplei was established by multiple molecular assays and by electron microscopy. First, a PCR with broad-range primers identified the complete 16S ribosomal DNA of T. whipplei in bioprosthesis tissue. Novel real-time reverse transcription-PCR assays were developed to detect mRNAs encoding recently identified proteins determined from the T. whipplei genome, specifically Whipplei surface protein (TW113) and a DNA polymerase III subunit (TW727). The positive detection of mRNAs indicated the presence of metabolically active bacteria and suggested the viability of T. whipplei. The quantification of T. whipplei genome equivalents by real-time PCR indicated a high-density bacterial colonization of the valve tissue. Additionally, an ultrastructural examination revealed numerous rod-shaped bacteria consistent in size with T. whipplei in the extracellular collagen matrix of the bioprosthesis. We conclude that extracellular growth of T. whipplei can occur in the microenvironment of biological prosthetic valve tissue and that T. whipplei endocarditis can occur in the absence of intestinal Whipple's disease.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

Tropheryma whipplei Infection of an Acellular Porcine Heart Valve Bioprosthesis in a Patient Who Did Not Have Intestinal Whipple's Disease

et al. 2004

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“…Clinical manifestations of heart disease are less evident (46,124). Nevertheless, the occurrence of cardiovascular involvement not accompanied by other symptoms is usually considered quite rare (20,27,40,57,79,115,131,166,174):…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Whipple's Disease and “Tropheryma whippelii”

2001

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SUMMARY Whipple's disease is a rare bacterial infection that may involve any organ system in the body. It occurs primarily in Caucasian males older than 40 years. The gastrointestinal tract is the most frequently involved organ, with manifestations such as abdominal pain, malabsorption syndrome with diarrhea, and weight loss. Other signs include low-grade fever, lymphadenopathy, skin hyperpigmentation, endocarditis, pleuritis, seronegative arthritis, uveitis, spondylodiscitis, and neurological manifestations, and these signs may occur in the absence of gastrointestinal manifestations. Due to the wide variability of manifestations, clinical diagnosis is very difficult and is often made only years or even decades after the initial symptoms have appeared. Trimethoprim-sulfamethoxazole for at least 1 year is usually considered adequate to eradicate the infection. The microbiological diagnosis of this insidious disease is rendered difficult by the virtual lack of culture and serodiagnostic methods. It is usually based on the demonstration of periodic acid-Schiff-positive particles in infected tissues and/or the presence of bacteria with an unusual trilaminar cell wall ultrastructure by electron microscopy. Recently, the Whipple bacteria have been characterized at the molecular level by amplification of their 16S rRNA gene(s). Phylogenetic analysis of these sequences revealed a new bacterial species related to the actinomycete branch which was named “Tropheryma whippelli.” Based on its unique 16S ribosomal DNA (rDNA) sequence, species-specific primers were selected for the detection of the organism in clinical specimens by PCR. This technique is currently used as one of the standard methods for establishing the diagnosis of Whipple's disease. Specific and broad-spectrum PCR amplifications mainly but not exclusively from extraintestinal specimens have significantly improved diagnosis, being more sensitive than histopathologic analysis. However, “T. whippelii” DNA has also been found in persons without clinical and histological evidence of Whipple's disease. It is unclear whether these patients are true asymptomatic carriers or whether differences in virulence exist among strains of “T. whippelii” that might account for the variable clinical manifestations. So far, six different “T. whippelii” subtypes have been found by analysis of their 16S-23S rDNA spacer region. Further studies of the pathogen “T. whippelii” as well as the host immune response are needed to fully understand this fascinating disease. The recent cultivation of the organisms is a promising major step in this direction.

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“…Different tissues are bast suited to verify the bacterial infection: (a) duodenal tissue obtained by endoscopy in patients with gastrointestinal symptoms; (b) synovial fluid in patients with an arthropathy [36]; (c) cerebrospinal fluid in newly diagnosed patients or when cerebral manifestation is suspected [37]; (d) cardiac valve tissue when the heart is affected [30,38]. Other non-specific laboratory tests can show malabsorption with vitamin deficiency, low albumin, and cholesterol or elevated erythrocyte sedimentation rate and acute-phase proteins.…”

Section: Diagnosismentioning

confidence: 99%

Progress, problems, and perspectives in diagnosis and treatment of Whipple’s disease

Mahnel

Marth

2004

Clin Exp Med

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Whipple's disease is a rare chronic infectious disorder first described in 1907 by G.H. Whipple. The disorder is caused by the newly identified bacterium Tropheryma whipplei and there is evidence that altered immune functions play a role in the manifestation of the disease. The organ systems mostly affected are the joints and the gut, and in the further course often also the heart, lung, brain, and eyes. The intestinal involvement occurs with abdominal pain and diarrhea, which leads to weight loss, malnutrition, and anemia. In some cases the infection spreads to the central nervous system, which may lead to loss of memory, confusion, or disturbances in gait. In the last few years, several steps towards an improved diagnosis of the disease and characterization of the causative bacterium have been made. While untreated disease may be lethal, treatment is often able to eradicate the organism. At present, therapy is based on observations in small patient groups and personal experience. There are different antibiotic therapy regimens often starting with intravenous application for 2 weeks followed by oral medication for 1 year. The first clinical therapy study is ongoing.

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Whipple’s disease with aortic regurgitation requiring aortic valve replacement

Cited by 20 publications

References 14 publications

Tropheryma whipplei Infection of an Acellular Porcine Heart Valve Bioprosthesis in a Patient Who Did Not Have Intestinal Whipple's Disease

Tropheryma whipplei Infection of an Acellular Porcine Heart Valve Bioprosthesis in a Patient Who Did Not Have Intestinal Whipple's Disease

Whipple's Disease and “Tropheryma whippelii”

Progress, problems, and perspectives in diagnosis and treatment of Whipple’s disease

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