Ventricular arrhythmia as the first manifestation of congenital toxoplasmosis

Sanchez-Codez, María Isabel; Rodríguez-González, Moises; Castellano-Martínez, Ana; Zafra Rodríguez, Pamela

doi:10.1016/j.medcle.2019.07.024

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In Spain, the incidence of infection during pregnancy is 1.9/1000. Most of the affected fetuses do not present symptoms at birth, but up to 80% develop serious sequelae, especially ophthalmological and neurological disorders, with cardiac involvement being unusual.<a class="elsevierStyleCrossRef" href="#bib0005">1</a>We present the case of a male neonate, with arrhythmic heartbeat and tachycardia of up to 200 bpm from birth, without hemodynamic repercussions. The obstetric background included late pre-term, urgent cesarean section and birth weight of 2875 g. The pregnant woman came from Equatorial Guinea and reported an uncontrolled pregnancy, presenting a positive peripartum test for human immunodeficiency virus (HIV), without having received antiretroviral therapy before or during delivery. The maternal serology revealed positive IgM and IgG (low avidity) for Toxoplasma gondii (T. gondii). An electrocardiogram was performed, which detected ventricular extrasystoles, frequent in bigeminy. The 24 h Holter monitoring showed ventricular extrasystoles in 38% of the recording. The echocardiogram and chest X-ray were normal. Serial lab tests revealed leukopenia, thrombocytopenia, and hypertransaminasemia. HIV C-reactive protein (CRP) test at 48 h of life was negative. The TORCH serology (toxoplasmosis, rubella, cytomegalovirus, herpes simplex and HIV) showed positive IgM and IgG for T. gondii, with a positive PCR of T. gondii in blood and cerebrospinal fluid (CSF). The extension study in the CSF, eye fundus, transfontanelar ultrasound, and cranial magnetic resonance imaging (MRI) showed no disorders. The neonate was treated with pyrimethamine (P: 1 mg/kg every 12 h, the first 48 h; then 1 mg/kg/day), sulfadiazine (S: 100 mg/kg/day, every 12 h) and folinic acid (FA: 10 mg, every 72 h). He did not receive corticosteroids. During the first week, bouts of ventricular tachycardia, up to 2 min long, without hemodynamic repercussions were observed. Propranolol was started (up to 3 mg/kg/day) with progressive disappearance of ventricular arrhythmias. The child is currently 6 months old, and presents adequate psychomotor and statutory development. He is asymptomatic, without ventricular arrhythmias or adverse effects with the same treatment.Transplacental transmission is responsible for the CT. The development of the disease is favored by the immunosuppression of the host. Immunological immaturity and maternal HIV predisposed our patient. Cardiac involvement in toxoplasmosis is uncommon (33 cases since 1964; no cases in CT as the one we present).<a class="elsevierStyleCrossRefs" href="#bib0010">2–5</a> It usually affects young and immunosuppressed adults (chemotherapy, HIV, heart transplantation), with highly variable presentation, including sudden death.<a class="elsevierStyleCrossRef" href="#bib0010">2</a> The appearance of palpitations or syncope in these patients should alert to the possibility of severe arrhythmias.<a class="elsevierStyleCrossRef" href="#bib0015">3</a> Although the exact cardiac pathogenic mechanism is not known, it seems to be related to an inflammatory hypersensitivity or autoimmune reaction.<a class="elsevierStyleCrossRefs" href="#bib0005">1,3</a> In the congenital form, the tachyzoites invasion of the myocardium develops into fibrosis and necrosis. However, in adults, myocardial involvement occurs after the cysts rupture in a latent state, often during healing in the chronic phase. Our patient did not receive a cardiac MRI or endomyocardial biopsy (EMB), the gold standard diagnosis. The validity of cardiac MRI is limited in children younger than 4 months due to its poor temporal-spatial resolution. EMB is an invasive technique, reserved for severe myocarditis with poor evolution. There was only one case where the invasion of T. gondii was identified in EMB.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> The neonatal diagnosis in our patient was based on a history of gestational toxoplasmosis, compatible serology, and PCR in blood, and CSF positive for T. gondii. The involvement of the conduction tissue of the heart which manifested as ventricular arrhythmias, and which has previously been described in toxoplasmosis cases and is not justifiable by any other cause, would explain the cardiac symptoms. The response to antiparasitic agents is variable, and null in cystic forms. Some authors suggest steroid treatment due to its autoimmune basis.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> Our patient was treated as a congenital form with P, S and FA according to clinical guidelines, and associating beta blockers as antiarrhythmic agents. The cardiac prognosis for toxoplasmosis is not known exactly; Of the 33 cases reviewed, 10 had serious complications and 4 recurrent arrhythmias. Incomplete elimination of the cystic forms, their multiplication, new rupture, and inflammatory reaction explain the relapses, and this emphasis the need for long-term cardiological follow-up of these patients. So far our case has not presented recurrences. CT can present exclusively as cardiac involvement, a form not described to date. Early treatment and adequate follow-up is essential to prevent the risk of serious arrhythmias and sudden death.Thanks toDr. Fernando Baquero Artigao from La Paz University Hospital for sharing his experience in congenital toxoplasmosis." 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Hamilton" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1136/hrt.35.3.349" "Revista" => array:6 [ "tituloSerie" => "Br Heart J" "fecha" => "1973" "volumen" => "35" "paginaInicial" => "349" "paginaFinal" => "352" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/4692665" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Complete atrioventricular block associated with toxoplasma myocarditis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M. Mariani" 1 => "M. Pagani" 2 => "C. Inserra" 3 => "S. de Servi" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "EP Europace" "fecha" => "2006" "volumen" => "8" "paginaInicial" => "221" "paginaFinal" => "223" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Recurrent, life-threatening atrioventricular dissociation associated with toxoplasma myocarditis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J.S. Duffield" 1 => "A.J. Jacob" 2 => "H.C. 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Vol. 155. Issue 11.