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• W3038671865 abstract "We read with great interest the description by Lefèvre et al1Lefèvre G. Leurs A. Gibier J.B. Copin M.C. Staumont-Salle D. Dezoteux F. et al.Idiopathic eosinophilic vasculitis: another side of hypereosinophilic syndrome? A comprehensive analysis of 117 cases in asthma-free patients.J Allergy Clin Immunol Pract. 2020; 8: 1329-1340.e3Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar of a retrospective cohort and literature review on patients with idiopathic eosinophilic vasculitis (EoV) occurring in asthma-free patients without antineutrophil-cytoplasmic antibodies (ANCAs). The hallmark of this proposed new entity is the concomitant presence of blood and tissue eosinophilia together with histologic and clinical features of necrotizing vasculitis. Idiopathic EoV, as defined by the authors,1Lefèvre G. Leurs A. Gibier J.B. Copin M.C. Staumont-Salle D. Dezoteux F. et al.Idiopathic eosinophilic vasculitis: another side of hypereosinophilic syndrome? A comprehensive analysis of 117 cases in asthma-free patients.J Allergy Clin Immunol Pract. 2020; 8: 1329-1340.e3Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar differs from eosinophilic granulomatosis with polyangiitis (EGPA) by the absence of asthma, and by distinct histopathology with the absence of granuloma despite evidence of necrotizing vasculitis. EoV also differs from the recently described EGPA limited to the lung.2Nasser M. Thivolet-Bejui F. Seve P. Cottin V. Lung-limited or lung-dominant variant of eosinophilic granulomatosis with polyangiitis.J Allergy Clin Immunol Pract. 2020; 8: 2092-2095Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar Pulmonary involvement in EoV was infrequent and nonspecific, reported as “chronic pneumonia,” “lung infiltrates,” and “nodular cavitary lung lesion.”1Lefèvre G. Leurs A. Gibier J.B. Copin M.C. Staumont-Salle D. Dezoteux F. et al.Idiopathic eosinophilic vasculitis: another side of hypereosinophilic syndrome? A comprehensive analysis of 117 cases in asthma-free patients.J Allergy Clin Immunol Pract. 2020; 8: 1329-1340.e3Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar The literature review also included 3 cases of so-called “EGPA without asthma,” with skin involvement but no lung biopsy available. To further illustrate the spectrum of these conditions, we report herein on a 70-year-old male patient diagnosed with biopsy-proven EoV limited to the lung, which however was non-necrotizing in this case. The patient was admitted for recent progressive dyspnea and relapsing hemoptysis. He was an ex-smoker (20 pack-years) with mild chronic obstructive pulmonary disease and no history of asthma or atopy. Eosinophil blood count was 2.88 × 106/L. ANCAs were negative. C-reactive protein was 35 mg/L. Chest computed tomography demonstrated abundant left pleural effusion with multiple bilateral lung nodules with halo sign. Multiple nasal polyps and nasal crust were present at fiberoptic examination. Cardiac ultrasound ruled out cardiac involvement. Bronchoscopy only found ongoing bleeding necessitating bronchial artery embolization. Pleural effusion was hemorrhagic. Lung biopsy examination found transmural eosinophil-rich vasculitis with eosinophilic pneumonia and alveolar hemorrhage, without necrosis or granuloma (Figure 1). The pleura was characterized by polymorphic eosinophil-rich inflammation without granuloma. The patient was successfully treated with pulse glucocorticoid therapy for 3 days followed by pulses of intravenous cyclophosphamide, then rituximab. Whether EoV represents a genuine distinct disease is unknown. Lefèvre et al1Lefèvre G. Leurs A. Gibier J.B. Copin M.C. Staumont-Salle D. Dezoteux F. et al.Idiopathic eosinophilic vasculitis: another side of hypereosinophilic syndrome? A comprehensive analysis of 117 cases in asthma-free patients.J Allergy Clin Immunol Pract. 2020; 8: 1329-1340.e3Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar suggested that it may represent a form of hypereosinophilic syndrome with eosinophil-driven vascular injury, rather than EGPA occurring in nonasthmatics. Indeed, pulmonary manifestations that are frequent in EGPA were generally absent in their series of EoV. Similarly, our patient had lung nodules, alveolar hemorrhage, and abundant pleural effusion, which are uncommon in EGPA.3Cottin V. Bel E. Bottero P. Dalhoff K. Humbert M. Lazor R. et al.Respiratory manifestations of eosinophilic granulomatosis with polyangiitis (Churg-Strauss).Eur Respir J. 2016; 48: 1429-1441Crossref PubMed Scopus (72) Google Scholar Moreover, histopathologic features corresponded to non-necrotizing EoV1Lefèvre G. Leurs A. Gibier J.B. Copin M.C. Staumont-Salle D. Dezoteux F. et al.Idiopathic eosinophilic vasculitis: another side of hypereosinophilic syndrome? A comprehensive analysis of 117 cases in asthma-free patients.J Allergy Clin Immunol Pract. 2020; 8: 1329-1340.e3Abstract Full Text Full Text PDF PubMed Scopus (23) Google Scholar and differed from EGPA even lung-limited.2Nasser M. Thivolet-Bejui F. Seve P. Cottin V. Lung-limited or lung-dominant variant of eosinophilic granulomatosis with polyangiitis.J Allergy Clin Immunol Pract. 2020; 8: 2092-2095Abstract Full Text Full Text PDF PubMed Scopus (3) Google Scholar Differentiating EoV from EGPA may be challenging, however, because the 3 histologic cardinal features of EGPA may not be found in the same tissue sample,4Katzenstein A.L. Diagnostic features and differential diagnosis of Churg-Strauss syndrome in the lung. A review.Am J Clin Pathol. 2000; 114: 767-772Crossref PubMed Scopus (103) Google Scholar and ANCAs are only found in <40% of cases of EGPA.5Jennette J.C. Falk R.J. Bacon P.A. Basu N. Cid M.C. Ferrario F. et al.2012 revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides.Arthritis Rheum. 2013; 65: 1-11Crossref PubMed Scopus (3937) Google Scholar To conclude, we agree that EoV is an entity distinct from EGPA, encompassing features of both systemic vasculitis and hypereosinophilic syndrome. The present case further demonstrates that EoV can be limited to the lung and that the vasculitis in EoV is not always necrotizing, supporting the concept that EGPA, lung-limited EGPA, and eosinophilic vasculitis (necrotizing or not) may be part of a common spectrum. All authors fulfill the ICMJE criteria for authorship. V. Cottin was responsible for design, analysis, and interpretation of data. All authors participated to data collection. All authors prepared the manuscript. All authors were responsible for manuscript review and revision. All authors approved the final version of the manuscript. The corresponding author (V. Cottin) had full access to all data in the study and had final responsibility for the decision to submit for publication. “Idiopathic Eosinophilic Vasculitis”: Another Side of Hypereosinophilic Syndrome? A Comprehensive Analysis of 117 Cases in Asthma-Free PatientsThe Journal of Allergy and Clinical Immunology: In PracticeVol. 8Issue 4PreviewThe absence of asthma may rule out a diagnosis of eosinophilic granulomatosis with polyangiitis in patients with hypereosinophilic syndrome (HES) and features of vasculitis. Full-Text PDF Reply to “Idiopathic non-necrotizing eosinophilic vasculitis limited to the lung: Part of a complex spectrum”The Journal of Allergy and Clinical Immunology: In PracticeVol. 8Issue 7PreviewIt was with great interest that we read Nasser et al's1,2 report on a case of biopsy-proven eosinophilic vasculitis (EoV) limited to the lung and 3 cases of biopsy-proven eosinophilic granulomatosis with polyangiitis (EGPA) limited to the lung. Full-Text PDF" @default.
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