Antineutrophil Cytoplasmic Antibodies and Organ-Specific Manifestations in Eosinophilic Granulomatosis with Polyangiitis: A Systematic Review and Meta-Analysis

Chang, Heng-Chia; Chou, Pai Chien; Lai, Chun Yu; Tsai, Hsiou Hsin

doi:10.1016/j.jaip.2020.07.038

Cited by 25 publications

(15 citation statements)

References 35 publications

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“…The positive rate of ANCA is only 30%‐35%. Glomerulonephritis is more likely to occur in the ANCA‐positive group, and cardiac dysfunction is more common in the negative group, 40,141 but there is no clear difference in respiratory symptoms and eosinophilia between them 136,140 …”

Section: Clinical Characteristics and Pathophysiology Of Adult‐onset mentioning

confidence: 99%

Adult‐onset eosinophilic airway diseases

Asano

Ueki

Tamari

et al. 2020

Allergy

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Allergic diseases in the upper and the lower airways such as allergic rhinitis and atopic asthma are usually accompanied by local eosinophilia and sometimes by peripheral blood eosinophilia. Chronic rhinosinusitis with nasal polyps (CRSwNP) and nonatopic asthma are also characterized by local and systemic eosinophilia; however, there are substantial differences in the age of onset and the presence of atopy. Atopic asthma and allergic rhinitis develop during childhood in most cases and mediated by type 1 hypersensitivity reactions to allergen(s), as are other childhood-onset allergic diseases such as food allergies and eczema. Patients often develop these diseases consecutively as an allergic march (Figure 1), suggesting the presence of common genetic backgrounds and pathophysiology. In contrast to childhood-onset disease, adult-onset asthma frequently presents as a nonatopic and eosinophilic condition, 1-6

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Section: Clinical Characteristics and Pathophysiology Of Adult‐onset mentioning

confidence: 99%

Adult‐onset eosinophilic airway diseases

Asano

Ueki

Tamari

et al. 2020

Allergy

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“…Eosinophilia in the peripheral blood is not included in the definition of relapse of vasculitis [ 16 ], and we confirmed that the number of eosinophils does not necessarily increase when EGPA relapses [ 17 ]. The association between the MPO-ANCA titer and disease severity has not been well evaluated [ 18 ].…”

Section: Discussionmentioning

confidence: 99%

Rituximab and dupilumab improve eosinophilic granulomatosis with polyangiitis with multiple pulmonary thrombi

Adachi

Oshikata

Kaneko

et al. 2022

Allergy Asthma Clin Immunol

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Background Eosinophilic granulomatosis with polyangiitis (EGPA) is characterized by a necrotizing vasculitis with tissue and peripheral blood eosinophilia affecting small and medium-sized arteries, capillaries, and veins. Venous thromboembolic events are uncommon in EGPA. Moreover, there are only a few reported cases of EGPA complicated by pulmonary embolism or infarction. Case presentation We report the case of a 43-year-old woman with eosinophilic granulomatosis with polyangiitis and acute respiratory and heart failure due to bilateral pulmonary artery thrombosis and left femoral vein thrombosis 12 years after disease onset. She also had cardiac involvement (myocarditis, pericardial effusion, and diastolic dysfunction), gastrointestinal symptoms, and peripheral neuropathy. The condition was refractory to treatment with systemic corticosteroids, intravenous cyclophosphamide, and mepolizumab, but the thrombosis and associated acute cardiac failure, as well as the cardiac and gastrointestinal symptoms and multiple polyneuropathy, improved after a switch to rituximab. However, the heart failure did not improve sufficiently and the patient continued to need inhaled oxygen at 1 L/min and asthma exacerbations occurred. We then swapped the patient’s mepolizumab treatment for dupilumab. Not only did she have no further asthma attacks after switching to dupilumab, but also her vasculitis symptoms improved. Oxygen therapy was discontinued as the heart failure improved 5 months after starting the dupilumab. Conclusions This may be the first case report of the successful treatment by rituximab of pulmonary thromboembolism associated with EGPA. In addition, in this patient, treatment with dupilumab was effective not only for the asthma symptoms but also for the symptoms of vasculitis and heart failure.

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“…Interestingly, ANCA is positive in only 30–40% of those with EGPA, and studies suggest that their ANCA status may portend differing disease manifestations. Compared with ANCA-positive cases, ANCA-negative EGPA is associated with a higher prevalence of cardiac or pulmonary complications [ 5 ] . Thus, it is crucial to recognize that a negative ANCA does not exclude EGPA.…”

Section: Discussionmentioning

confidence: 99%

Eosinophilic Myocarditis Presenting as Cardiac Tamponade: A Diagnostic Challenge

Nishimura

Yokoyama

et al. 2022

European Journal of Case Reports in Internal Medicine

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Eosinophilic myocarditis (EM) is a rare cause of acute heart failure. It can occur secondary to drug hypersensitivity, autoimmune diseases such as vasculitis, idiopathic hypereosinophilic syndrome (HES) or malignancy, but is often under-recognized and underdiagnosed, being confused with other causes of heart failure. While EM is associated with various clinical symptoms, it is rarely associated with cardiac tamponade that requires urgent pericardiocentesis. Here we describe a patient with EM who presented with cardiac tamponade and decompensated heart failure likely secondary to autoimmune disease.

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Antineutrophil Cytoplasmic Antibodies and Organ-Specific Manifestations in Eosinophilic Granulomatosis with Polyangiitis: A Systematic Review and Meta-Analysis

Cited by 25 publications

References 35 publications

Adult‐onset eosinophilic airway diseases

Adult‐onset eosinophilic airway diseases

Rituximab and dupilumab improve eosinophilic granulomatosis with polyangiitis with multiple pulmonary thrombi

Eosinophilic Myocarditis Presenting as Cardiac Tamponade: A Diagnostic Challenge

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