Myocarditis due to Coxiella burnetti

Rodríguez Arboleya, Sara; García González, Paula; Santos Seoane, Sagrario

doi:10.1016/j.medcle.2023.04.014

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We report the case of a young male who was admitted for bilateral pneumonia with a good course, and who after months developed a condition compatible with severe myocarditis with positive serology for C. burnetti. The interesting aspect of this case lies in the importance of history-taking to establish clinical suspicion and reach a diagnosis, with the aim of initiating early treatment and avoiding complications.A 32-year-old, healthy male presented to our clinic with cough and fever. Chest X-ray showed bilobar pneumonia. Laboratory tests showed an increase in high-sensitivity troponin I (hsTnI) up to 507 ng/l (0–26 ng/l) with a subsequent decrease. This finding was not taken into account as he had no symptoms and a normal electrocardiogram. Blood cultures, pneumococcal and Legionella urinary antigen test, as well as the study of viruses in respiratory samples were negative and serology for microorganisms related to atypical pneumonias was not requested. After 7 days of treatment with levofloxacin, the patient experienced clinical and radiological improvement.Three months later the patient presented with dyspnoea and orthopnoea. Laboratory tests showed NT-proBNP 4102 pg/ml (0–1800 pg/ml) and hsTnI 585 ng/l (0–26 ng/l). Electrocardiogram was normal and echocardiogram showed severe biventricular dysfunction (left ventricular ejection fraction [LVEF] 20%). Myocarditis was suspected and confirmed by magnetic resonance imaging (MRI), which showed subendocardial delayed enhancement. Serology was performed and was positive for C. burnetti, with an IgG phase II titre of 1/3200 and phase I of 1/800. The patient, who is a goat farmer, was re-interviewed and reported that he had helped with the birth of his animals a few weeks before the first admission. Treatment was started with doxycycline 200 mg per day, with spironolactone, beta-blocker and sacubitril–valsartan, with recovery of ventricular function (LVEF 55%) at 12 months and symptom resolution.Q fever is a zoonosis caused by C. burnetti, an intracellular gram-negative bacillus with a worldwide distribution. The reservoir is goats, sheep and cattle, although cases have also been reported in dogs and cats.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a> The most common transmission mechanism is inhalation of pseudospores. Transmission also occurs through ingestion of contaminated products, such as unpasteurised milk, or occupational exposure to the products of livestock births.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>The acute form is usually self-limiting within 2 weeks. Chronic infection manifests 12–24 months after contact and is rare. Infection is usually asymptomatic (54%–60%), followed by acute (40%) and chronic (1%–5%) forms.Cardiac involvement is usually seen in the chronic form of endocarditis, aortitis, infection of aneurysms or vascular prostheses. Pericarditis and myocarditis are less common forms, with endocarditis being the most common form of cardiac damage.<a class="elsevierStyleCrossRef" href="#bib0005">1</a>Coxiella myocarditis is exceptional, with an incidence of less than 1%, and fewer than 30–35 cases have been reported.<a class="elsevierStyleCrossRef" href="#bib0015">3</a> Myocarditis occurs in the acute presentation, but there is one reported case of chronic presentation.<a class="elsevierStyleCrossRef" href="#bib0020">4</a>Coxiella has antigenic variation: it expresses antigens according to the stage of infection. In acute forms, IgG > 1/128 and IgM ≥ 1/32 for phase II antigen are detected. In chronic forms, IgG ≥ 1/800 for phase I antigen is considered diagnostic.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>The clinical presentation of myocarditis is highly variable. Elevation of hsTnI is less common. Unspecific ST-segment abnormalities are seen on the electrocardiogram.<a class="elsevierStyleCrossRef" href="#bib0025">5</a> MRI is useful. Three sequences are recommended: T2 for oedema, early enhancement for hyperaemia and late enhancement for irreversible damage. The presence of 2 out of 3 predicts myocarditis in 78%.<a class="elsevierStyleCrossRef" href="#bib0025">5</a>Treatment consists of 200 mg doxycycline daily for 2 weeks. If valvular disease is present, hydroxychloroquine 600 mg daily for 3–6 months is added to prevent endocarditis.<a class="elsevierStyleCrossRef" href="#bib0020">4</a> In our case, since the contact had been months ago and the chronic phase antigens were borderline, doubts arose as to how long to maintain treatment, so we opted for doxycycline 6 months until the IgG against phase I antigen was less than 1/200, as if it were a chronic manifestation.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>Myocarditis mortality is 25% and overall mortality is 1%–2%.<a class="elsevierStyleCrossRef" href="#bib0015">3</a> In our case the diagnosis was delayed for several months, and the development was severe, although the patient’s progress was good.Coxiella myocarditis is a rare entity. Mild cases are subclinical and should be included in the differential diagnosis of patients with febrile syndrome presenting with troponin elevations, chest pain, rhythm disturbances or heart failure. In this entity, a good history-taking on epidemiological factors is essential. Diagnosis is based on clinical findings, serology and non-invasive imaging tests such as MRI.Ethical considerationsIn this study we followed the ethical guidelines established by the Ethics Committee of our institution, as well as international guidelines for medical research in humans. All patients included were treated confidentially and protected in accordance with local legislation and international standards.FundingThis research has not received specific support from public, private or non-profit organisations.Conflict of interestThe authors declare that they have no conflicts of interest." 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Tello" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/s0300-8932(02)76719-5" "Revista" => array:6 [ "tituloSerie" => "Rev Esp Cardiol" "fecha" => "2002" "volumen" => "55" "paginaInicial" => "875" "paginaFinal" => "877" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/12199986" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Infección por Coxiella burnetti (fiebre Q)" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "M.T. Fraile" 1 => "C. Muñoz" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Enferm Infecc Micrbiol Clin" "fecha" => "2010" "volumen" => "8" "paginaInicial" => "29" "paginaFinal" => "32" "itemHostRev" => array:3 [ "pii" => "S0002934313002210" "estado" => "S300" "issn" => "00029343" ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0015" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Q fever presenting as myocarditis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R. Hammami" 1 => "A. Bahloul" 2 => "S. Charfeddine" 3 => "W. Feki" 4 => "N.B. Ayed" 5 => "L. Abid" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.idcr.2021.e01056" "Revista" => array:5 [ "tituloSerie" => "IDCases" "fecha" => "2021" "volumen" => "23" "paginaInicial" => "e01056" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/33643842" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Rare case of chronic Q fever miocarditis in end stage heart failure patient: a case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. Goyal" 1 => "T. Dalia" 2 => "P. Bhyan" 3 => "H. Farhoud" 4 => "Z. Shah" 5 => "A. Vidic" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.4330/wjc.v14.i9.508" "Revista" => array:6 [ "tituloSerie" => "World J Cardiol" "fecha" => "2022" "volumen" => "14" "paginaInicial" => "508" "paginaFinal" => "513" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/36187426" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diagnóstico de las miocarditis: papel de las pruebas de imagen" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A. Mas-Stachurska" 1 => "M. Sitges" 2 => "S. Prat-Gonzalez" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Cardiocore" "fecha" => "2012" "volumen" => "47" "paginaInicial" => "143" "paginaFinal" => "146" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000016100000003/v1_202308021441/S2387020623002966/v1_202308021441/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000016100000003/v1_202308021441/S2387020623002966/v1_202308021441/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020623002966?idApp=UINPBA00004N"]

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Vol. 161. Issue 3.

Pages 135-136 (August 2023)

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