Infective endocarditis due to Bartonella bacilliformis

associated with systemic vasculitis: a case report

Item Type info:eu-repo/semantics/article

Authors Peñafiel-Sam, Joshua; Alarcón-Guevara, Samuel; Chang-

Cabanillas, Sergio; Perez-Medina, Wilkerson; Mendo-Urbina,
Fernando; Ordaya-Espinoza, Eloy

Citation Infective endocarditis due to Bartonella bacilliformis associated

with systemic vasculitis: a case report 2017, 50 (5):706 Revista da
Sociedade Brasileira de Medicina Tropical

DOI 10.1590/0037-8682-0042-2017

Publisher Sociedade Brasileira de Medicina Tropical - SBMT

Journal Revista da Sociedade Brasileira de Medicina Tropical

Rights info:eu-repo/semantics/openAccess

Download date 11/04/2020 22:18:24

Link to Item http://hdl.handle.net/10757/622419

Rev Soc Bras Med Trop 50(5):706-708, September-October, 2017
doi: 10.1590/0037-8682-0042-2017

Case Report

Infective endocarditis due to Bartonella bacilliformis

associated with systemic vasculitis: a case report
Joshua Peñafiel-Sam[1], Samuel Alarcón-Guevara[1], Sergio Chang-Cabanillas[1],
Wilkerson Perez-Medina[2], Fernando Mendo-Urbina[1],[3] and Eloy Ordaya-Espinoza[1],[4]

[1]. Escuela de Medicina, Universidad Peruana de Ciencias Aplicadas, Lima, Perú. [2]. Servicio de Reumatología,
Hospital Nacional Edgardo Rebagliati Martins, Lima, Perú. [3]. Servicio de Infectología, Hospital Nacional Edgardo Rebagliati Martins,
Lima, Perú. [4]. Department of Medicine, University of Minnesota, Minneapolis, MN, USA.

Abstract
Infective endocarditis due to Bartonella bacilliformis is rare. A 64-year-old woman, without previous heart disease, presented
with 6 weeks of fever, myalgias, and arthralgias. A systolic murmur was heard on the tricuspid area upon examination, and an
echocardiogram showed endocardial lesions in the right atrium. Bartonella bacilliformis was isolated in blood cultures, defining
the diagnosis of infective endocarditis using Duke’s criteria. Subsequently, the patient developed clinical and laboratory features
compatible with antineutrophil cytoplasmic antibody-associated vasculitis. This case presents an uncommon complication of
B. bacilliformis infection associated with the development of systemic vasculitis.
Keywords: Infective endocarditis. Bartonella bacilliformis. Systemic vasculitis.

INTRODUCTION polyangiitis (MPA), granulomatosis with polyangiitis [(GPA),

previously Wegener’s granulomatosis], and eosinophilic
Infective endocarditis (IE) is an infection of the endocardial
granulomatosis with polyangiitis [(EGPA), previously Churg-
surface that usually occurs in patients with previous anatomical
Strauss syndrome]. Several microorganisms that trigger systemic
abnormalities, such as rheumatic heart disease, congenital
malformations, prosthetic valves, and intracardiac devices. vasculitis have been described, including Staphylococcus aureus
Staphylococcus and Streptococcus spp. are the most commonly nasal carriage, which has been related to GPA and B. henselae
identified microorganisms in blood cultures. However, in with Henoch-Schönlein purpura3,4. Here, we present the case of
some cases, conventional cultures are negative due to previous a woman diagnosed with B. bacilliformis endocarditis associated
antibiotic use or because some fastidious microorganisms with systemic vasculitis.
require isolation in special media, such as infections by the
CASE REPORT
HACEK group (Haemophilus spp., Aggregatibacter spp.,
Cardiobacterium hominis, Eikenella corrodens, and Kingella A 64-year-old woman, with a past medical history of
spp.) and Brucella spp. Additionally, a few Bartonella spp. idiopathic pulmonary fibrosis and hypothyroidism, presented
have been identified as causes of endocarditis, with Bartonella with 6 weeks of intermittent fever, myalgias, and arthralgias.
quintana and Bartonella henselae being the most common1. She reported a previous course of antibiotics for an unspecified
Bartonella bacilliformis, the etiologic agent of Carrion’s disease infection without presenting any improvement. On physical
(CD), has also been reported as a cause of endocarditis in a examination, she was febrile at 39°C and had hypothyroid
pediatric patient with a history of right ventricular-coronary facies and mesosystolic murmur on the tricuspid area. The
artery fistula2. Infections can trigger vasculitis through various rest of the examination was unremarkable. Ancillary testing
direct (e.g., endothelium infection) or indirect (e.g., molecular revealed normocytic, normochromic anemia (11.1g/dl), 9.26 ×
mimicry) pathways. Antineutrophil cytoplasmic antibody 103 leukocytes/μl, lymphopenia (0.84 × 103 lymphocytes/μl),
(ANCA)-associated vasculitis (AAV) is a group of vasculitides neutrophilia (7.72 × 103 neutrophils/μl), 0.09 × 103 eosinophils/
that predominantly affect small vessels and cause systemic μl, thyroid-stimulating hormone level of 22.7μUI/ml, total
manifestations. AAV has three major variants: microscopic bilirubin of 0.30mg/dl, and creatinine of 0.82mg/dl. Initial
blood and urine cultures were negative. An electrocardiogram
revealed no abnormalities, but the transthoracic echocardiogram
Corresponding author: Dr. Eloy Ordaya Espinoza.
showed a perivalvular lesion in the right atrium, suggestive
e-mail: eeordaya@umn.edu
Received 10 February 2017 of vegetation. The patient was admitted to the Cardiology
Accepted 5 May 2017 ward with the probable diagnosis of atrial myxoma vs IE.

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 Peñafiel-Sam J et al. - Endocarditis due to B. bacilliformis

Transesophageal echocardiography (TEE) revealed two masses,

10 × 18mm and 5 × 10mm, on the lateral wall of the right atrium
(Figure 1). The patient was transferred to the Infectious Diseases
ward, where tests were performed, including repeated aerobic
and anaerobic blood cultures, peripheral smear for malaria,
serology testing for Brucella and Salmonella, bone marrow
culture, and enzyme-linked immunosorbent assay (ELISA)
test for human immunodeficiency virus, all of which resulting
negative. The erythrocyte sedimentation rate, rheumatoid factor,
and C-reactive protein were elevated (94mm/h, 24.2IU/ml,
and 8.31mg/dl, respectively), and the complement values were
normal (C3, 157mg/dl and C4, 18mg/dl). Due to persistent fever
of 39°C, worsening anemia (8.9g/dl), and a history of a recent
travel to Tarapoto, a jungle region in San Martín, Peru, where
FIGURE 1 - Transesophageal echocardiogram: masses on the lateral wall of
CD cases were recently reported, blood samples were sent to the the right atrium, measuring (a) 10 × 18mm and (b) 5 × 10mm on admission.
Institute de Medicina Tropical Daniel Alcides Carrión in Lima AI: left atrium; AD: right atrium.
to rule out CD. The samples revealed negative blood smears
(Giemsa stain) but positive blood cultures for B. bacilliformis in
Columbia agar (growth at 15 days of incubation). The definitive
diagnosis of IE due to B. bacilliformis was made using modified
Duke’s criteria. Treatment with intravenous (IV) ciprofloxacin
(400mg/12h) was initiated, but changed to IV ceftriaxone
(2g/24h) shortly after, due to lack of clinical improvement.
Despite this therapeutic modification, the patient had
persistent anemia and intermittent fever and presented progressive
weight loss, paresthesia in extremities, muscular weakness, livedo
reticularis, and macular exanthema in the abdomen, dorsum, and
lower limbs. Moreover, acute kidney injury (AKI) with creatinine
of 1.69mg/dl, proteinuria (0.5g/24h), and the absence of hematuria
were noticed. For this reason, Rheumatology evaluated the case
and recommended electromyography, which was conclusive for
mononeuritis multiplex; immunological markers were positive FIGURE 2 - Transesophageal echocardiogram: longitudinal mass located
for p-ANCA (by ELISA). Renal biopsy revealed tubulointerstitial on the lateral wall of the right atrium, measuring 32 × 12mm at 3 weeks of
inflammation to predominance of mononuclear cells and the initial antibiotic regimen administered to the patient. AD: right atrium;
moderate-severe thickening of juxtaglomerular and interlobular MASA: atrial mass.
vessels. Due to the persistence of symptoms, two Bartonella
blood cultures were repeated and, again, resulted positive (growth DISCUSSION
at 20 days and 17 days of incubation). A few coccobacilli were
Carrion’s disease is a metaxenic disease endemic in
also reported in the blood smear. Furthermore, a follow-up TEE
showed growth of the intracardiac masses (Figure 2), prompting a Colombia, Ecuador, and Peru, caused by B. bacilliformis and
change from ceftriaxone monotherapy to a combination therapy of transmitted by the bite of female Lutzomyia sand flies. This
IV ciprofloxacin (200mg/12h), gentamicin (4mg/kg per 24h), and biphasic disease consists of an acute form, known as Oroya
oral (PO) rifampicin (10mg/kg per 24h). The aminoglycoside was fever, and a chronic phase, denominated Peruvian wart 5.
changed later to PO azithromycin (500mg/24h) due to worsening Infectious and noninfectious complications occur in 70% of
renal function. Additionally, surgical treatment was considered, patients in the acute phase, such as neurological, hematologic,
but the patient did not consent to the procedure. The fever, anemia, and cardiovascular alterations, including cardiac failure,
and lymphopenia progressively resolved, but the weakness and pericarditis, and myocarditis6. To our knowledge, only one
paresthesias persisted. A multidisciplinary team decided to initiate case of endocarditis caused by this bacterium was reported in a
PO prednisone (20mg/24h) for AAV, based on the symptomatology pediatric patient with underlying cardiac disease2. We diagnosed
and immunological and electromyographic findings. The patient B. bacilliformis endocarditis based on modified Duke’s criteria1,
was discharged afebrile, with progressive clinical improvement considering as major criteria the persistence of positive cultures
and negative blood cultures for B. bacilliformis after 10 weeks for B. bacilliformis and the presence of intracardiac lesions
of antibiotic treatment. On outpatient evaluation, a follow-up on TEE in the absence of any other anatomical explanation.
TEE showed absence of vegetations, prednisone dose was Persistent bacteremia due to B. bacilliformis has been described
reduced to 5mg/day, and PO azathioprine (50mg/24h) was in up to 54% of patients with Peruvian wart living in endemic
added, resulting in resolution of myalgias and paresthesias. areas, from where there are no reports of endocarditis5. Our

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Rev Soc Bras Med Trop 50(5):706-708, September-October, 2017

patient had a recent and short epidemiological exposure to Conflict of interest

a new endemic area in the Peruvian jungle, which could The authors declare that there is no conflict of interest.
explain her different outcome7. The patient initially received
a regimen based on our current guidelines for treating acute
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Financial support
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This project was self-financed. Clin Exp Nephrol. 2009;13(6):605-13.

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