Systemic thrombo-embolic events in a middle-aged male with Loeffler endocarditis without peripheral eosinophilia-a case report.

BACKGROUND: Loeffler-endocarditis (LE) is considered a chronic restrictive cardiomyopathy and manifestation of eosinophilic myocarditis characterized by eosinophilic infiltration. LE is a rare underdiagnosed disease and associated with high morbidity and mortality. CASE PRESENTATION: We report a case of a 46-year-old man suffering from LE associated with thromboembolic events without peripheral eosinophilia. The patient presented with typical clinical signs of acute onset of limb ischaemia, predominantly on the right limb, indicating immediate iliacal thrombectomy and due to a severe compartment syndrome additional fasciotomy. Total occlusion also of left popliteal artery suggesting an impaired chronic and aggravated impaired perfusion indicated also urgent left sided revascularization. Subsequent echocardiography revealed severe left ventricular dysfunction with a striking amount of spontaneous echo-contrast, noticeable in the left ventricular cavity. Furthermore the initial CT scan demonstrated asymptomatic left kidney- and brain infarctions. Diagnostic workup including endomyocardial biopsy (EMB) of the left ventricle, uncovered an underlying LE without peripheral eosinophilia. CONCLUSIONS: This case demonstrates and highlights the findings, treatment and outcome of a patient with LE and associated thrombo-embolic events without peripheral eosinophilia and emphazises the importance of awareness for LE in patients presenting with an acute cardiac decompensation and thrombo-embolic events. EMB should be performed early in unstable patients unsuitable for cardiovascular magnetic resonance imaging.

Lung fibroblasts produce IL-33 in response to stimulation with retinoblastoma-binding protein 9 via production of prostaglandin E2.

Intestinal nematode infection induces pulmonary eosinophilia via IL-33, although the mechanism of pulmonary IL-33 induction remains unclear. Because nematode migration damages lungs, we speculated that lung-derived damage-associated molecular patterns (DAMPs) possess an IL-33-inducing activity (IL33ia). Indeed, intra-nasal administration of a lung extract induced IL-33 production in lungs. Additionally, lung extracts increased Il33 mRNA expression in primary lung fibroblasts. Proteomic analysis identified retinoblastoma-binding protein 9 (RBBP9) as a major DAMP with IL33ia. RBBP9 was originally discovered as a protein that provides cells with resistance to the growth inhibitory effect of transforming growth factor (TGF)-ß1. Here, we found that stimulation by RBBP9 induced primary fibroblasts to produce prostaglandin E2 (PGE2) that, in turn, induced fibroblasts to produce IL-33. RBBP9-activated fibroblasts expressed mRNAs of cyclooxygenase-2 (COX-2) and PGE2 synthase-1 that convert arachidonic acid to PGE2. Furthermore, they expressed PGE2 receptors E-prostanoid (EP) 2 and EP4. Thus, treatment with a COX-2 inhibitor or EP2 and/or EP4 receptor antagonists inhibited RBBP9-induced IL-33 production. Nematode infection induced pulmonary Il33 mRNA expression, which was inhibited by the COX-2 inhibitor or EP2 and EP4 antagonists, suggesting that nematode infection induced pulmonary Il33 mRNA via PGE2. RBBP9 was expressed constitutively in the lung in the steady state, which did not increase after nematode infection. Finally, we found that Rbbp9-deficient mice had a significantly diminished capacity to increase pulmonary Il33 mRNA expression following nematode infection. Thus, the PGE2-EP2/EP4 pathway activated by RBBP9 released from damaged lungs is important for pulmonary IL-33 production in nematode-infected animals.

Loeffler endocarditis with intracardiac thrombus: case report and literature review.

BACKGROUND: Loeffler endocarditis is a relatively rare and potentially life-threatening heart disease. This study aimed to identify the characteristic features of Loeffler endocarditis with intracardiac thrombus on a background of hypereosinophilic syndrome (HES). CASE PRESENTATION: We described a 57-year-old woman with Loeffler endocarditis and intracardiac thrombus initially presenting with neurological symptoms, who had an embolic stroke in the setting of HES. After cardiac magnetic resonance (CMR), corticosteroids and warfarin were administered to control eosinophilia and thrombi, respectively. During a 10-month follow-up, the patient performed relatively well, with no adverse events. We also systematically searched PubMed and Embase for cases of Loeffler endocarditis with intracardiac thrombus published until July 2021. A total of 32 studies were eligible and included in our analysis. Further, 36.4% of recruited patients developed thromboembolic complications, and the mortality rate was relatively high (27.3%). CMR was a powerful noninvasive modality in providing diagnostic and follow-up information in these patients. Steroids were administered in 81.8% of patients, achieving a rapid decrease in the eosinophil count. Also, 69.7% of patients were treated with anticoagulant therapy, and the thrombus was completely resolved in 42.4% of patients. Heart failure and patients not treated with anticoagulation were associated with poor outcomes. CONCLUSIONS: Cardiac involvement in HES, especially Loeffler endocarditis with intracardiac thrombus, carries a pessimistic prognosis and significant mortality. Early steroids and anticoagulation therapy may be beneficial once a working diagnosis is established. Further studies are needed to provide evidence-based evidence for managing this uncommon manifestation of HES.

[Clinical and morphological observation of Löffler's endomyocarditis]. / Kliniko-morfologicheskoe nablyudenie endomiokardita Lefflera.

The article describes the case of a 42-year-old young woman with Löffler's endomyocarditis (fibroplastic endomyocarditis with eosinophilic syndrome). Pathomorphological (macroscopic and histological) criteria for the diagnosis of this rare form of restrictive cardiomyopathy are presented.

Morphologically proved ANCA positive Loeffler's pancarditis: medical and surgical treatment.

Loeffler's endocarditis remains is a very rare disease, develops due to eosinophilic inflammation predominantly of the endocardium with an outcome in fibrosis and massive thrombus formation and. He is generally characterized by an unfavorable prognosis. Clinical case of a 42-year-old patient with Loeffler endocarditis is presented. The development of the disease was preceded by a polyvalent allergy, mild dry eye syndrome and pansinusitis with a single eosinophilia of blood up to 16%. The reason for the hospitalization was the appearance of biventricular heart failure. During the previous year, the level of blood eosinophils remained normal, a threefold increase in the level of eosinophilic cationic protein was observed once. A 20-fold increase in the pANCA level, a 2.5-fold increase in the level of antibodies to DNA, an antibody to the nuclei of cardiomyocytes 1:160 were detected. The diagnosis was made on the basis of electrocardiography data (low QRS voltage, atrial hypertrophy), echocardiography, multispiral computed tomography and magnetic resonance imaging of the heart (thickening and delayed contrasting of the endocardium, massive thrombosis of the left ventricular apex with obliteration of its cavity, encapsulated fluid in the pericardium with compression of the right ventricle). Systolic dysfunction, severe signs of restriction and arrhythmias were absent. Trombectomy, tricuspid valve plasty, pericardial resection, suturing of an open oval window were performed. Signs of active inflammation with single eosinophils, vasculitis, perimuscular sclerosis, endocardial sclerosis were detected in morphological and immunohistochemical studies of endo-, myo-, pericardium. Viral genome was not found. The therapy with methylprednisolone 24 mg/day, azathioprine 75 mg/day was started. Six months after the operation, the symptoms of heart failure are completely absent, the thrombosis did not recur.

Multiparametric cardiac magnetic resonance imaging (CMR) for the diagnosis of Loeffler's endocarditis: a case report.

BACKGROUND: Endocarditis parietalis fibroplastica Löfflein (EPF) is a rare form of primary restrictive cardiomyopathy with poor prognosis. It is generally caused by hypereosinophilic syndrome with eosinophilic penetration of the heart. This leads to congestive heart failure in three different stages. As a frequent manifestation of neoplastic diseases, cardiac involvement means poor prognosis. CASE PRESENTATION: The present report deals with a case of EPF caused by non-specified T-cell lymphoma (T-NOS). Besides an elevated Troponin-T enzyme, the electrocardiogram and the transthoracic echocardiography did not show any characteristic results. Due to risk/benefit assessment and low thrombocyte amounts, endomyocardial biopsy and catheterization were discarded. Using cardiovascular magnetic resonance (CMR) with steady-state free precession sequences, T2-mappping, strain analysis and late gadolinium enhancement, we were able to clearly highlight cardiac involvement at different stages. These findings characterized T-NOS as a palliative situation. CONCLUSION: Multiparametric CMR can not only identify EPF but also characterize the patchy disease state. This provides an individual prognosis assessment. Aside from prognosis estimation, it can also be used for therapy monitoring.

Cardiac involvements in hypereosinophilia-associated syndrome: Case reports and a little review of the literature.

Hypereosinophilia-associated syndrome is a rare group of systemic diseases without certain underlying causes. Hypereosinophilic syndrome (HES) and eosinophilic granulomatosis with polyangiitis (EGPA), formerly known as Churg-Strauss syndrome (CSS), are initial considerations, when underlying causes remains unexplained despite of complete evaluation of hypereosinophilia. In this study, we report two rare cases, one case of HES with Loeffler endocarditis, and the other one of EGPA with restrictive cardiomyopathy mimicking myocardial infarction, to further address differential chief cardiac manifestations between HES and EGPA. Key roles of echocardiography played in detection of cardiac involvements, diagnosis, and prognosis prediction are also highlighted.

Usefulness of three-dimensional echocardiography in the assessment of valvular involvement in Loeffler endocarditis.

Loeffler endocarditis is a complication of hypereosinophilic syndrome resulting from eosinophilic infiltration of heart tissue. We report a case of Loeffler endocarditis in which three-dimensional transthoracic and transesophageal echocardiography provided additional information to what was found by two-dimensional transthoracic echocardiography alone. Our case illustrates the usefulness of combined two- and three-dimensional echocardiography in the assessment of Loeffler endocarditis. In addition, a summary of the features of hypereosinophilic syndrome and Loeffler endocarditis is provided in tabular form.

Paediatric presentation of cardiac involvement in hypereosinophilic syndrome.

Eosinophilic endomyocarditis is extremely rare in paediatrics. This case report aims to present the sequential changes from thrombotic to fibrotic stages of the cardiac involvement in a child with hypereosinophilic syndrome. Endomyocardial fibrosis progressively aggravated the severity of the mitral regurgitation. Bioprosthetic valve replacement was finally performed without recurrence of cardiac complications despite a late relapse of the underlying disease.

Early-Stage Loeffler's Endocarditis with Isolated Right Ventricular Involvement: Management, Long-Term Follow-Up, and Review of Literature.

Loeffler's endocarditis is a clinical condition characterized by combination of three key findings: unexplained prolonged and marked eosinophilia (>1500 eosinophils/mm(3) ), absence of a primary cause of hypereosinophilia, and evidence of eosinophil-mediated organ damage. We report a case of a 55-year-old African American male with symptoms of heart failure. Hematology showed white blood cell count of 17 670/mm(3) with 63% eosinophils and an absolute eosinophil count of 11 133/mm(3) . Echocardiogram and computed tomography showed near complete obliteration of right ventricular cavity. Endomyocardial biopsy showed diffuse myocyte necrosis with extensive eosinophilic infiltration without fibrosis consistent with early Loeffler's endocarditis. Molecular and cytogenetic analyses of bone marrow cells were negative for FIP1L1-PDGFRA fusion, PDGFRB mutation, abnormal myeloid maturation, or a lymphoproliferative disorder. Flow cytometry showed no clonality excluding chronic eosinophilic leukemia. There was a complete resolution of symptoms and eosinophilia after 1 month of steroid therapy.

Loeffler endocarditis in young woman - a case report.

Loeffler endocarditis is a rare acquired endocardial and myocardial disease characterized by a sharp decrease in the compliance of either or both ventricles with an acute diastolic dysfunction and massive mural thrombosis. This disease is presented in the classification of cardiomyopathies and is a variant of restrictive cardiomyopathy. Today Loeffler endocarditis is considered as a manifestation of hypereosinophilic syndrome with predominant heart involvement. The life-time diagnosis of myocardial injury due to eosinophilic infiltration is rare, or it is diagnosed at the stage of necrotizing endomyocarditis, when the treatment is no longer effective. A number of issues regarding the individual aspects of the pathogenesis of hypereosinophilic syndrome and Loeffler endocarditis are still not fully understood, as well as the long-term prospects for the use of drugs for the treatment of hypereosinophilic syndrome, especially in young and middle-aged persons. Loeffler endocarditis can be suspected in the presence of hypereosinophilia on the background of causeless (unexplainable) hypertrophy of the left ventricle or both ventricles. The article includes a case of the life-time diagnosis of this disease in a young woman with the retrospective analysis of the early stages of the disease, echocardiographic and radiologic imaging at the advanced stage of the disease and quite successful treatment option for this disease.

Transthoracic echocardiographic detection of pulmonary valve involvement in Löeffler's endocarditis.

We are describing pulmonary valve involvement with thickening and stenosis detected by two-dimensional transthoracic echocardiography in an adult presenting with Löeffler's endocarditis. To our knowledge, this has not been described previously. Complete regression of the lesions occurred with corticosteroid therapy. Tricuspid valve thickening and stenosis and thickening and thrombus formation in the right ventricle also present initially disappeared completely with therapy.

In-Depth Review of Loeffler Endocarditis: What Have We Learned?

Loeffler endocarditis, eosinophilic endocarditis or eosinophilic endomyocardial disease are conditions associated with hypereosinophilia and they affect the heart function. Loeffler endocarditis is a rare endomyocardial disorder thought to be caused by eosinophilic damage. The disorder is characterized by inflammatory infiltration, formation of thrombus within cardiovascular system, and ultimately fibrosis of the afflicted area. It can lead to multiple severe complications, including thromboembolic disease, thickening of fibrous tissue in the endocardium of ventricles, valve involvement, apical obliteration, and various heart disorders. Although early clinical intervention can lead to remission, the underlying mechanisms of the disorder remain unresolved. In the present article, we summarise the existing literature concerning Loeffler endocarditis based on PubMed, Web of Science, and other medical databases to conduct an in-depth review of the epidemiology, etiology, pathophysiological mechanisms, staging, diagnosis, treatment and prognosis of Loeffler endocarditis. Meanwhile, we provide novel patients data and clinical figures of Loeffler endocarditis to supplement the understanding of this cardiac disorder. The findings presented in this article provide a basis for further studies and can be used to improve management of the disorder.

Cutaneous Larva Migrans (CLM) may not be easy to diagnose: a case report and narrative review.

BACKGROUND: Cutaneous Larva Migrans (CLM) is one of the most common zoonotic dermatoses in subtropical and tropical regions and some European countries. It is caused by different types of hookworm, such as Ancylostoma braziliense, Ancylostoma caninum, and Uncinaria stenocephala. It is usually easy to diagnose, but the atypical presentation may occasionally mimic other dermatoses. CASE REPORT: A 32-year-old man presented with an extensive eczematous rash that developed during a recent vacation in Thailand. He didn't respond to antihistamines and systemic steroids. Finally, he was diagnosed with an atypical presentation of CLM and treated successfully with anthelminthic therapy. CONCLUSION: The report of an atypical presentation of CLM is crucial to increase awareness among healthcare workers, helping in early diagnosis and reducing potential psychological distress that patients may face.

A case report of right-sided heart failure secondary to JAK2-mutation Loeffler endocarditis.

Loeffler's endocarditis (LE) is the cardiac manifestation of hypereosinophilic syndrome. We present a case of LE in a 45-year-old female, resulting in diffuse endothelial fibrosis and severe right-sided heart failure. The patient was admitted with dyspnoea and oedema, with haematology revealing an absolute eosinophil count of 20.9 × 109. Imaging showed near-complete obliteration of the right ventricular apical and formation of thromboses. Endomyocardial biopsy indicated diffuse fibrous hyperplasia of the endocardium with fibrinous thrombi rich in eosinophils. Molecular and cytogenetic analyses of bone marrow cells showed no signs of FIP1L1-PDGFRA fusion, PDGFRB mutation, abnormal myeloid maturation, or a lymphoproliferative disorder. Flow cytometry indicated no clonality, ruling out chronic eosinophilic leukaemia. Gene mutation screening discovered a p.L583_A586delinesS mutation in the JAK2 gene. Following treatment with ruxolitinib, the patient's eosinophil levels normalized, but unfortunately, the damage to the heart was irreversible. The patient was hospitalized multiple times due to right heart failure and resistance to diuretics. After thorough discussions with the medical team, it was determined that a heart transplantation would be the most effective treatment. Following the surgery, the patient successfully navigated the postoperative critical period with the support of an intra-aortic balloon pump (IABP), continuous renal replacement therapy (CRRT), and ventilator-assisted ventilation but subsequently developed an acquired Intensive care unit-acquired weakness (ICU-AW) and a depressive state. Fortunately, the patient gradually recovered from these complications.

A Case of Thrombectomy for Loeffler's Endocarditis.

Loeffler's endocarditis, characterized by eosinophilic infiltration leading to thrombus formation and fibrosis in the ventricle, is associated with severe complications, such as embolism and heart failure. While anticoagulation and steroids are standard treatments, surgical thrombectomy is rarely reported. This is a case report of a 74-year-old man presented with an abnormal electrocardiogram. Echocardiography revealed a 38 × 29 mm mass in the left ventricular apex, and blood studies revealed hypereosinophilia, leading to a diagnosis of Loeffler's endocarditis. Despite warfarin treatment, the thrombus persisted. The left ventricular intracardiac thrombus exhibited significant mobility, and we decided to perform a thrombectomy via a trans-left ventricular approach. After the surgery, steroid therapy was initiated. The patient recovered without recurrence of the thrombus or deterioration in cardiac function. Left ventricular thrombectomy for Loeffler's endocarditis is considered a beneficial option to prevent thrombosis.

An update on the current and emerging pharmacotherapy for the treatment of human ascariasis.

INTRODUCTION: Globally, Ascaris lumbricoides is the commonest helminthic infection that affects people in underdeveloped countries and returning immigrants in industrialized nations. This article aims to provide latest updates on the epidemiology, clinical manifestations, and pharmacotherapy of ascariasis. AREAS COVERED: A PubMed search was conducted using Clinical Queries and the key terms 'human ascariasis' OR 'Ascaris lumbricoides.' Ascaris lumbricoides is highly endemic in tropical and subtropic regions and among returning immigrants in industrialized nations. Predisposing factors include poor sanitation and poverty. The prevalence is greatest in young children. Most infected patients are asymptomatic. Patients with A. lumbricoides infection should be treated with anti-helminthic drugs to prevent complications from migration of the worm. Mebendazole and albendazole are indicated for children and nonpregnant women. Pregnant individuals should be treated with pyrantel pamoate. EXPERT OPINION: Cure rates with anthelmintic treatment are high. No emerging pharmacotherapy can replace these existing drugs of good efficacy, safety profile and low cost for public health. It is opinioned that advances in the management of ascariasis include diagnostic accuracy at affordable costs, Emodepside is highly effective in single doses against ascarids in mammals and in human trials. The drug could be registered for human use in multiple neglected tropical diseases.

Echocardiographic monitoring of myocardial function in a female patient with right heart Loeffler endocarditis at thrombotic stage after Epstein-Barr-virus infection.

BACKGROUND: Transthoracic echocardiography is usually the first non-invasive imaging modality for the detection of Loeffler endocarditis at thrombotic stage. In the recent decade 3D echocardiography and deformation imaging already proved as a helpful tool for the monitoring of left and right ventricular heart disease. CASE PRESENTATION: The present case illustrates the diagnostic role of 3D echocardiography and deformation imaging in the acute stage of right sided Loeffler endocarditis in a 70-year-old Western European (German) woman. This case proves that myocardial involvement due to inflammation can be detected at subclinical stages by speckle tracking echocardiography. Acute deterioration of left and right ventricular function and the early response to prednisolone therapy can objectively be monitored. In addition, alterations of effective stroke volume can quantitatively be assessed by 3D right ventricular volumetry with exclusion of thrombus formation in the volume measurements. CONCLUSION: This case underlines the importance of 3D echocardiography and deformation imaging as a helpful diagnostic tool in disease management in the acute phase of Loeffler endocarditis at thrombotic stage.

Incessant Ventricular Tachycardia: An Atypical Presentation of Chronic Eosinophilic Leukemia.

Hypereosinophilic syndrome comprises a diverse and intricate array of rare disorders, exhibiting clinical manifestations that extend across various medical subspecialties. Within its myeloid form, chronic eosinophilic leukemia represents a rare myeloid malignancy characterized by severe hematological complications and distinctive organ dysfunction, notably affecting the cardiovascular system. This report presents a rare case of chronic eosinophilic leukemia and Loeffler syndrome with an initial presentation of ventricular tachycardia.

Hypereosinophilia and Left Ventricular Thrombus: A Case Report and Literature Review.

Left ventricular thrombus (LVT) has historically been reported as a complication of acute left ventricular (LV) myocardial infarction. It is most commonly observed in cases of LV systolic dysfunction attributed to ischemic or nonischemic etiologies. Conversely, the occurrence of LVT in normal LV systolic function is an exceptionally rare presentation and is predominantly associated with conditions such as hypereosinophilic syndrome (HES), cardiac amyloidosis, left ventricular noncompaction, hypertrophic cardiomyopathy (HCM), hypercoagulability states, immune-mediated disorders, and malignancies. Notably, hypereosinophilia (HE) has been linked with thrombotic events. Intracardiac thrombus is a well-known complication of eosinophilic myocarditis (EM) or Loeffler endomyocarditis, both of which are considered clinical manifestations of HES. We present a case of a 63-year-old male with normal LV systolic function, HE, and noncontributory hypercoagulability workup, who presented with thromboembolic complications arising from LVT. Interestingly, the diagnostic evaluation for EM and Loeffler endocarditis was nonconfirmatory. Additionally, we performed a literature review to delineate all similar cases. This article also outlines the pathophysiology, diagnosis, and treatment approaches for hypereosinophilic cardiac involvement with a specific focus on LVT.