Surgical management of hydatid cyst of the interventricular septum.

Cardiac hydatidosis constitutes about 0.5-2% of all hydatid diseases. The interventricular septum is involved in 4% of cardiac hydatidosis cases. We demonstrate the surgical management of a large (76 x 66 mm) septal hydatid cyst in a 20-year-old man. The hydatid cyst was approached through a right ventriculotomy under cardiopulmonary bypass. The germinative membrane was removed in toto. The cavity was then obliterated to prevent recurrence of the cyst. The patient had an uneventful postoperative recovery. Histopathological examination confirmed the diagnosis of hydatid cyst. The patient remains asymptomatic with echocardiographic and magnetic resonance imaging evidence of freedom from disease at the 3-month follow-up.

Risk of Chronic Cardiomyopathy Among Patients With the Acute Phase or Indeterminate Form of Chagas Disease: A Systematic Review and Meta-analysis.

Importance: Chagas cardiomyopathy is associated with substantial morbidity and mortality. Precise estimates of the risk of developing cardiomyopathy among patients with the acute or indeterminate chronic forms of Chagas disease are lacking. Objective: To estimate the risk of developing chronic cardiomyopathy in patients with acute and indeterminate chronic forms of Chagas disease. Data Sources: A systematic search in the Cochrane Library, Embase, Latin American and Caribbean Health Sciences Literature (LILACS), Medline, and Web of Science Core Collection databases was conducted from October 8 to October 24, 2018. Studies published between January 1, 1946, and October 24, 2018, that were written in the English, Spanish, and Portuguese languages were included. Search terms included Chagas disease; development of cardiomyopathy; latency duration; and determinants of the Chagas latency period. Study Selection: Longitudinal observational studies of participants diagnosed with the acute phase of Chagas infection or the indeterminate chronic form of Chagas disease who were followed up until the development of cardiomyopathy were included. Studies were excluded if they did not provide sufficient outcome data. Of 10 761 records initially screened, 32 studies met the criteria for analysis. Data Extraction and Synthesis: Critical appraisals of studies were performed using checklists from the Joanna Briggs Institute Reviewer's Manual, and data were collected from published studies. A random-effects meta-analysis was used to obtain pooled estimated annual rates. Data were analyzed from September 11 to December 4, 2019. This study followed the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) reporting guideline for the registration of the protocol, data collection and integrity, assessment of bias, and sensitivity analyses. Main Outcomes and Measures: Main outcomes were defined as the composite of the development of any new arrhythmias or changes in electrocardiogram results, dilated cardiomyopathy and segmental wall motion abnormalities in echocardiogram results, and mortality associated with Chagas disease. Results: A total of 5005 records were screened for eligibility. Of those, 298 full-text articles were reviewed, and 178 of those articles were considered for inclusion in the quantitative synthesis. After exclusions, 32 studies that included longitudinal observational outcomes were selected for the analysis; 23 of those studies comprised patients with the indeterminate chronic form of Chagas disease, and 9 of those studies comprised patients in the acute phase of Chagas infection. The analysis indicated that the pooled estimated annual rate of cardiomyopathy development was 1.9% (95% CI, 1.3%-3.0%; I2 = 98.0%; τ2 [ln scale] = 0.9992) in patients with indeterminate chronic Chagas disease and 4.6% (95% CI, 2.7%-7.9%; I2 = 86.6%; τ2 [ln scale] = 0.4946) in patients with acute Chagas infection. Conclusions and Relevance: Patients with the indeterminate chronic form of Chagas disease had a significant annual risk of developing cardiomyopathy. The annual risk was more than double among patients in the acute phase of Chagas infection.

A systematic review of the Trypanosoma cruzi genetic heterogeneity, host immune response and genetic factors as plausible drivers of chronic chagasic cardiomyopathy.

Chagas disease is a complex tropical pathology caused by the kinetoplastid Trypanosoma cruzi. This parasite displays massive genetic diversity and has been classified by international consensus in at least six Discrete Typing Units (DTUs) that are broadly distributed in the American continent. The main clinical manifestation of the disease is the chronic chagasic cardiomyopathy (CCC) that is lethal in the infected individuals. However, one intriguing feature is that only 30-40% of the infected individuals will develop CCC. Some authors have suggested that the immune response, host genetic factors, virulence factors and even the massive genetic heterogeneity of T. cruzi are responsible of this clinical pattern. To date, no conclusive data support the reason why a few percentages of the infected individuals will develop CCC. Therefore, we decided to conduct a systematic review analysing the host genetic factors, immune response, cytokine production, virulence factors and the plausible association of the parasite DTUs and CCC. The epidemiological and clinical implications are herein discussed.

[Cardiac Hydatid Cyst in a Child: A Case Report]. / Quiste hidatídico intracardíaco en una niña: Reporte de caso.

Echinococcosis is a parasitic infection caused by Echinococcus granulosus, which, in its cystic state, forms the socalled hydatid cyst. It presents important morbidity, with possible sequelae related to the location, and high costs due to surgical and prolonged pharmacological treatment. The liver and the lung are the most common anatomical locations, and much rarer are the kidney, spleen, brain, and heart, where the latter represents 0.5 to 2% of total cases. Peru is an endemic country of this anthropozoonosis and mainly records cases in the central highlands (95%). This paper presents the case of a 10-year-old girl, diagnosed with this disease, CE1 ultrasound classification, clinical group 1 (confirmed by pathological anatomy) with specific surgical and pharmacological treatment (albendazole) afterward. The patient recovered satisfactorily from the surgery and was discharged at 16 days, without complications.

La equinococosis es una infección parasitaria provocada por Echinococcus granulosus, que, en su estado quístico, forma al denominado quiste hidatídico. Presenta morbilidad importante, con posibles secuelas relacionadas con la ubicación, y altos costos debido al tratamiento quirúrgico y farmacológico prolongado. El hígado y el pulmón son las ubicaciones anatómicas más usuales, mucho más raras son el riñón, bazo, cerebro y corazón, este último representa el 0,5 % a 2 % del total de casos. El Perú es un país endémico de esta antropozoonosis y principalmente registra casos procedentes de la sierra central (95 %). Se presenta el caso de una niña de diez años, con diagnóstico de esta entidad, clasificación ecográfica CE 1, grupo clínico 1 (confirmado por anatomía patológica) con posterior tratamiento quirúrgico y farmacológico específico (albendazol). La paciente se recuperó satisfactoriamente de la cirugía practicada, y fue dada de alta a los 16 días, sin complicaciones.

Quiste hidatídico intracardíaco en una niña: Reporte de caso / Cardiac Hydatid Cyst in a Child: A Case Report

RESUMEN La equinococosis es una infección parasitaria provocada por Echinococcus granulosus, que, en su estado quístico, forma al denominado quiste hidatídico. Presenta morbilidad importante, con posibles secuelas relacionadas con la ubicación, y altos costos debido al tratamiento quirúrgico y farmacológico prolongado. El hígado y el pulmón son las ubicaciones anatómicas más usuales, mucho más raras son el riñón, bazo, cerebro y corazón, este último representa el 0,5 % a 2 % del total de casos. El Perú es un país endémico de esta antropozoonosis y principalmente registra casos procedentes de la sierra central (95 %). Se presenta el caso de una niña de diez años, con diagnóstico de esta entidad, clasificación ecográfica CE 1, grupo clínico 1 (confirmado por anatomía patológica) con posterior tratamiento quirúrgico y farmacológico específico (albendazol). La paciente se recuperó satisfactoriamente de la cirugía practicada, y fue dada de alta a los 16 días, sin complicaciones.

ABSTRACT Echinococcosis is a parasitic infection caused by Echinococcus granulosus, which, in its cystic state, forms the socalled hydatid cyst. It presents important morbidity, with possible sequelae related to the location, and high costs due to surgical and prolonged pharmacological treatment. The liver and the lung are the most common anatomical locations, and much rarer are the kidney, spleen, brain, and heart, where the latter represents 0.5 to 2% of total cases. Peru is an endemic country of this anthropozoonosis and mainly records cases in the central highlands (95%). This paper presents the case of a 10-year-old girl, diagnosed with this disease, CE1 ultrasound classification, clinical group 1 (confirmed by pathological anatomy) with specific surgical and pharmacological treatment (albendazole) afterward. The patient recovered satisfactorily from the surgery and was discharged at 16 days, without complications.

Inhibition of TGF-ß pathway reverts extracellular matrix remodeling in T. cruzi-infected cardiac spheroids.

Chagasic cardiomyopathy (CC) is the main manifestation of Chagas Disease (CD). CC is a progressive dysfunctional illness, in which transforming growth factor beta (TGF-ß) plays a central role in fibrogenesis and hypertrophy. In the present study, we tested in a three-dimensional (3D) model of cardiac cells culture (named cardiac spheroids), capable of mimicking the aspects of fibrosis and hypertrophy observed in CC, the role of TGF-ß pathway inhibition in restoring extracellular matrix (ECM) balance disrupted by T. cruzi infection. Treatment of T. cruzi-infected cardiac spheroids with SB 431542, a selective inhibitor of TGF-ß type I receptor, resulted in a reduction in the size of spheroids, which was accompanied by a decrease in parasite load and in fibronectin expression. The inhibition of TGF-ß pathway also promoted an increase in the activity of matrix metalloproteinase (MMP)-2 and a decrease in tissue inhibitor of matrix metalloproteinase (TIMP)-1 expression, which may be one of the mechanisms regulating extracellular matrix remodeling. Therefore, our study provides new insights into the molecular mechanisms by which inhibition of TGF-ß signaling reverts fibrosis and hypertrophy generated by T. cruzi during CC and also highlights the use of cardiac spheroids as a valuable tool for the study of fibrogenesis and anti-fibrotic compounds.

A case of intracardiac echinococcosis.

We describe a case of multisystemic cystic echinococcosis exhibiting intracardiac involvement. A lesion inside the lateral wall of the left ventricle induced negative T waves on leads V5 and V6.

Primary cardiac hydatid cyst presenting as noncompaction cardiomyopathy.

We report a case of cyst was initially labeled as left ventricular noncompaction cardiomyopathy. An accurate diagnosis is essential to establish the most effective treatment strategy. In particular, echocardiographic examination assists in identifying the correct diagnosis. In this case, two-dimensional and three-dimensional echocardiography and computed tomography were used for definitive diagnosis of cardiac hydatid cyst.

Evaluation of VDR gene polymorphisms in Trypanosoma cruzi infection and chronic Chagasic cardiomyopathy.

Vitamin D is an important modulator of the immune response. It acts over several immune cell types where the Vitamin D receptor (VDR) is expressed. Due to the high relevance of this signaling pathway, several studies have investigated the possible influence of genes involved in the metabolism of Vitamin D and its receptor in different human diseases. Here, we analyzed whether four single-nucleotide polymorphisms of the VDR gene (rs731236, rs7975232, rs1544410 and rs2228570) are involved in the susceptibility to infection by Trypanosoma cruzi and/or to chronic Chagas cardiomyopathy (CCC) in a Colombian endemic population for this parasite. Our results showed that the rs2228570*A allele is associated with CCC development (P = 4.46E-03, OR = 1.51). In summary, the data presented in this report suggest that variation within the VDR gene may affect the immune response against T. cruzi, increasing the probability of cardiac complications in infected individuals.

Immunoregulatory mechanisms in Chagas disease: modulation of apoptosis in T-cell mediated immune responses.

BACKGROUND: Chronic Chagas disease presents different clinical manifestations ranging from asymptomatic (namely indeterminate) to severe cardiac and/or digestive. Previous results have shown that the immune response plays an important role, although no all mechanisms are understood. Immunoregulatory mechanisms such as apoptosis are important for the control of Chagas disease, possibly affecting the morbidity in chronic clinical forms. Apoptosis has been suggested to be an important mechanism of cellular response during T. cruzi infection. We aimed to further understand the putative role of apoptosis in Chagas disease and its relation to the clinical forms of the disease. METHODS: Apoptosis of lymphocytes, under antigenic stimuli (soluble T. cruzi antigens - TcAg) where compared to that of non-stimulated cells. Apoptosis was evaluated using the expression of annexin and caspase 3(+) by T cells and the percentage of cells positive evaluated by flow cytometry. In addition activation and T cell markers were used for the identification of TCD4(+) and TCD8(+) subpopulations. The presence of intracellular and plasma cytokines were also evaluated. Analysis of the activation status of the peripheral blood cells showed that patients with Chagas disease presented higher levels of activation determined by the expression of activation markers, after TcAg stimulation. PCR array were used to evaluate the contribution of this mechanism in specific cell populations from patients with different clinical forms of human Chagas disease. RESULTS: Our results showed a reduced proliferative response associated a high expression of T CD4(+)CD62L(-) cells in CARD patients when compared with IND group and NI individuals. We also observed that both groups of patients presented a significant increase of CD4(+) and CD8(+) T cell subsets in undergoing apoptosis after in vitro stimulation with T. cruzi antigens. In CARD patients, both CD4(+) and CD8(+) T cells expressing TNF-α were highly susceptible to undergo apoptosis after in vitro stimulation. Interestingly, the in vitro TcAg stimulation increased considerably the expression of cell death TNF/TNFR superfamily and Caspase family receptors genes in CARD patients. CONCLUSIONS: Taken together, our results suggest that apoptosis may be an important mechanism for the control of morbidity in T. cruzi infection by modulating the expression of apoptosis genes, the cytokine environment and/or killing of effector cells.

Host and parasite genetics shape a link between Trypanosoma cruzi infection dynamics and chronic cardiomyopathy.

Host and parasite diversity are suspected to be key factors in Chagas disease pathogenesis. Experimental investigation of underlying mechanisms is hampered by a lack of tools to detect scarce, pleiotropic infection foci. We developed sensitive imaging models to track Trypanosoma cruzi infection dynamics and quantify tissue-specific parasite loads, with minimal sampling bias. We used this technology to investigate cardiomyopathy caused by highly divergent parasite strains in BALB/c, C3H/HeN and C57BL/6 mice. The gastrointestinal tract was unexpectedly found to be the primary site of chronic infection in all models. Immunosuppression induced expansion of parasite loads in the gut and was followed by widespread dissemination. These data indicate that differential immune control of T. cruzi occurs between tissues and shows that the large intestine and stomach provide permissive niches for active infection. The end-point frequency of heart-specific infections ranged from 0% in TcVI-CLBR-infected C57BL/6 to 88% in TcI-JR-infected C3H/HeN mice. Nevertheless, infection led to fibrotic cardiac pathology in all models. Heart disease severity was associated with the model-dependent frequency of dissemination outside the gut and inferred cumulative heart-specific parasite loads. We propose a model of cardiac pathogenesis driven by periodic trafficking of parasites into the heart, occurring at a frequency determined by host and parasite genetics.

Cardiac condition in young chagasic women.

RATIONALE: Chagas disease is a complex disorder caused by Trypanosoma cruzi. Most patients remain asymptomatic for several years and 30% of them progress quietly to developing cardiomyopathy. The factors that lead to chronic myocardial lesions are not fully understood. OBJECTIVE: To investigate the association between clinical symptoms and single nucleotide polymorphisms in chagasic and non-chagasic women younger and older than 55 years of age. METHODS AND RESULTS: we analyzed Ala-9Val and Ile58Thr polymorphisms of the SOD-Mn gene, 138ex1ins/del A of the endothelin-1 gene (ET-1) and H323H (T/C) of the endothelin receptor A gene (ETA), by PCR-RFLP using genomic DNA from leukocyte of 85 women. We also evaluated serum lipid profile, renal and liver function, chest X-rays, electrocardiograms (ECGs) and echocardiography (EchoCG). Biochemical profiling did not show differences between chagasic and non-chagasic patients. The polymorphisms analyses showed a significant association in the distribution of frequencies of the Mn-SOD Ile58Thr gene between both groups. Young chagasic patients had a significantly higher prevalence of abnormalities in X-rays, in ECGs and they showed grade II and III of NYHA functional classes. The chance of having an abnormal EchoCG was 5.87 higher in young chagasic patients (OR=5.87, 95% CI 1.47-23.4). DISCUSSION: We concluded that the parasite affects young females by accelerating the deterioration of cardiac function, independent of other cardiovascular risk factors and the cardioprotective action of estrogens present in the premenopausal stage.

Mannose-binding lectin regulates host resistance and pathology during experimental infection with Trypanosoma cruzi.

Mannose-binding lectin (MBL) is a humoral pattern-recognition molecule important for host defense. Although recent genetic studies suggest an involvement of MBL/MASP2-associated pathways in Chagas' disease, it is currently unknown whether MBL plays a role in host resistance to the intracellular protozoan Trypanosoma cruzi, the causative agent of Chagas' disease. In this study we employed MBL(-/-) mice to assess the role of MBL in resistance to experimental infection with T. cruzi. T. cruzi infection enhanced tissue expression of MBL both at the mRNA and protein level. Similarly, symptomatic acute Chagas' disease patients displayed increased serum concentrations of MBL compared to patients with indeterminate, asymptomatic forms of the disease. Furthermore, increased parasite loads in the blood and/or tissue were observed in MBL(-/-) mice compared to WT controls. This was associated with reduced systemic levels of IL-12/23p40 in MBL(-/-) mice. Importantly, MBL(-/-) mice infected with a cardiotropic strain of T. cruzi displayed increased myocarditis and cardiac fibrosis compared to WT controls. The latter was accompanied by elevated hydroxyproline content and mRNA levels of collagen-1 and -6 in the heart. These observations point to a previously unappreciated role for MBL in regulating host resistance and cardiac inflammation during infection with a major human pathogen.

Hidatidosis cardiaca: caso clínico / Cardiac hydatidosis: Report of one case

Background: Hydatidosis is an endemic zoonosis in Chile. We report a 48-year-old former slaughterman, with a previous history of pulmonary hydatidosis, who presented a stroke without associated cardiovascular symptoms. An echocardiogram revealed a tumor mass with cystic features in the left ventricle. The patient was operated and the cyst was successfully excised. During the follow up, the patient remains asymptomatic.

Decreased intensity of inflammation in benznidazole-treated mice inoculated with Trypanosoma cruzi I stocks from Mexico and persistence of circulating parasites.

We analyzed the intensity of inflammation and parasitism in BALB/c mice infected with Trypanosoma cruzi I stocks from Mexico with and without benznidazole treatment in the acute phase of disease. Heart and skeletal muscles were evaluated for parasites and inflammation and blood was evaluated for persistence of circulating parasites. Parasitemia was influenced by T. cruzi stocks used and benznidazole treatment. This treatment cleared circulating parasites three days after starting treatment when monitored by direct microscopy. There was a significant reduction of inflammation in skeletal muscles after benznidazole treatment in animals infected with Mexican T. cruzi I stocks (P < 0.05), but this reduction was not significant in the heart (P > 0.05). Trypanosoma cruzi I parasites from Mexico were demonstrated by polymerase chain reaction in tissues and blood of animals after benznidazole treatment.

[Anaesthetic treatment in the removal of a cardiac hydatid cyst]. / Tratamiento anestésico en la exéresis de un quiste hidatídico cardíaco.

Echinococcosis is an endemic infection in hot countries. Cardiac involvement is rare, but serious. The risk of anaphylactic shock during surgery requires a rapid intra-operative diagnosis and immediate treatment. We present the case of a 35 year-old male in whom a cardiac hydatid cyst was detected that required surgery. He was given preliminary treatment with oral albendazole for one month and prescribed anti-H1, anti-H2 and corticosteroids prior to the removal of the cyst using bypass surgery. During the operation he was also given a bolus of hydrocortisone and dexchlorpheniramine and the surgical area was protected by gauzes soaked in hypertonic saline. The patient required an infusion of noradrenaline to maintain haemodynamic stability. He progressed with no more complications, and was discharged at 14 days.