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Orthopedic Infections and Treatments · Journal article
Romanian Journal of Cardiology · August 7, 2026
Early or partial results. Treat as a signal, not a conclusion.
This is a case report of Coxiella burnetii endocarditis in an 11-year-old with complex congenital heart disease, diagnosed by fluorescence in situ hybridization of explanted prosthetic conduit material after initial serology and blood PCR were negative. The patient showed clinical improvement after conduit replacement and doxycycline-hydroxychloroquine therapy, with subsequent seroconversion. The case illustrates that tissue-based diagnostics may detect infection when conventional blood-based tests fail, particularly in pediatric immunocompromised hosts.
Case report. An 11-year-old boy with complex congenital heart disease (specific diagnoses not detailed), history of multiple prior cardiac surgical interventions, splenectomy for severe unexplained splenomegaly with hypersplenism, and immunological abnormalities including hypogammaglobulinemia.. Intervention: Hybrid surgical replacement of stenotic prosthetic conduit; initiation of doxycycline-hydroxychloroquine combination therapy.. Single center (location not specified in abstract)..
11-year-old boy with two-year history of low-grade fever, hepatomegaly, hypoalbuminemia, hypogammaglobulinemia, peripheral edema, and progressive right-sided heart failure prior to diagnosis Coxiella burnetii detected by fluorescence in situ hybridization of explanted Contegra conduit; blood PCR and serology initially negative Following conduit replacement and doxycycline-hydroxychloroquine therapy, patient showed rapid clinical improvement with resolution of systemic manifestations
Safety was not reported in the material analysed. Check the source before drawing any conclusion about harm.
This case suggests that in pediatric patients with congenital heart disease and prosthetic material who present with prolonged unexplained systemic manifestations, Coxiella burnetii should be considered even when initial serology is negative, and tissue-based molecular diagnostics from explanted material may establish diagnosis. The case demonstrates utility of tissue PCR when blood-based testing fails and supports early empirical doxycycline-hydroxychloroquine therapy pending confirmatory serology.
A single pediatric case report describing diagnosis and management of an uncommon infection in congenital heart disease; raises awareness of diagnostic approach but does not establish treatment efficacy or generalizable outcomes.
As stated by the source record.
Quoted from the source exactly as published.
This case suggests that in pediatric patients with congenital heart disease and prosthetic material who present with prolonged unexplained systemic manifestations, Coxiella burnetii should be considered even when initial serology is negative, and tissue-based molecular diagnostics from explanted material may establish diagnosis. The case demonstrates utility of tissue PCR when blood-based testing fails and supports early empirical doxycycline-hydroxychloroquine therapy pending confirmatory serology.
Graded across the dimensions that decide whether you should act, each from what the source actually supports. There is no single score, and where a dimension was not assessed it says so.
Abstract Background Coxiella burnetii is a well-recognized cause of culture-negative endocarditis in patients with prosthetic cardiac material. In pediatric populations, diagnosis is uncommon and frequently delayed, particularly in the presence of immunological abnormalities that may impair serological responses. Case presentation We present a case of Coxiella burnetii endocarditis in an 11-year-old boy with complex congenital heart disease and multiple prior cardiac surgical interventions who developed chronic systemic manifestations associated with prosthetic conduit infection. He was first evaluated at our center at nine years of age, presenting with a two-year history of low-grade fever, hepatomegaly, hypoalbuminemia, hypogammaglobulinemia, peripheral edema, and progressive right-sided heart failure. His medical history was notable for splenectomy performed for severe unexplained splenomegaly with hypersplenism. Extensive investigations failed to identify an infectious etiology. Echocardiography demonstrated severe conduit stenosis without vegetations. Two years later, during hybrid surgical replacement of the stenotic conduit, fluorescence in situ hybridization of the explanted Contegra conduit detected Coxiella burnetii, while blood PCR and serology were initially negative. Following conduit replacement and initiation of doxycycline-hydroxychloroquine therapy, the patient showed rapid clinical improvement, with resolution of systemic manifestations and normalization of protein and immunoglobulin levels. Seroconversion with high phase I and II IgG titers occurred after three months of therapy. Conclusions In pediatric patients with congenital heart disease and prosthetic cardiac material, Coxiella burnetii infection may present with prolonged systemic manifestations. Despite negative initial serology, tissue-based detection from explanted material can establish the diagnosis when conventional investigations fail, and may support the initiation of targeted long-term antimicrobial therapy.
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