“Idiopathic Eosinophilic Vasculitis”: Another Side of Hypereosinophilic Syndrome? A Comprehensive Analysis of 117 Cases in Asthma-Free Patients

Lefèvre, Guillaume; Leurs, Amélie; Gibier, Jean‐Baptiste; Copin, Marie‐Christine; Staumont‐Sallé, D.; Dezoteux, F.; Chenivesse, Cécile; López, Benjamín; Terriou, Louis; Hachulla, É.; Launay, David; Lengliné, Étienne; Labalette, Myriam; Groote, Pascal de; Pontana, François; Quéméneur, T.; Hatron, Pierre‐Yves; Schleinitz, N.; Viallard, Jean‐François; Hamidou, M.; Martin, Thierry; Morati-Hafsaoui, Chafika; Groh, Matthieu; Lambert, Marc; Kahn, Jean‐Emmanuel

doi:10.1016/j.jaip.2019.12.011

Cited by 44 publications

(48 citation statements)

References 98 publications

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“…Interestingly, some F/P+ MN‐eo patients also met diagnostic and/or classification criteria for EGPA. Moreover, this diagnostic dilemma is further enriched by the recent report of both basic and clinical evidence of eosinophil‐related vascular toxicity, 39 including cases of HES‐related thromboangiitis obliterans 40 and eosinophilic vasculitis (whether single‐organ or systemic) 41 . Next, in line with similar reports in other HES subtypes, 42 we report on three F/P+ MN‐eo patients (otherwise free from all other thrombotic risk factors) with venous thromboembolic events occurring at disease onset, most presumably due to an increased pro‐coagulant state led by hypereosinophilia 43 .…”

Section: Discussionmentioning

confidence: 99%

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

et al. 2020

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FIP1L1-PDGFRA-positive myeloid neoplasm with eosinophilia (F/P+ MN-eo) is a rare disease: robust epidemiological data are lacking and reported issues are scarce, of low sample-size and limited follow-up. Imatinib mesylate (IM) is highly efficient but no predictive factor of relapse after discontinuation has yet been identified. One hundred and fifty-one patients with F/P+ MN-eo (143 males; mean age at diagnosis 49 years; mean annual incidence: 0.18 case per million population) were included in this retrospective nationwide study involving all French laboratories who perform the search of F/P fusion gene (study period: 2003-2019). The main organs involved included the spleen (44%), skin (32%), lungs (30%), heart (19%) and central nervous system (9%). Serum vitamin

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Section: Discussionmentioning

confidence: 99%

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

et al. 2020

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“…Eosinophils can accumulate in multiple organs, including the heart, skin, nervous system, and lungs, causing end-organ damage in patients with HES [46] . In the classical view of eosinophil-induced organ damage in HES, tissue infiltrates are often accompanied by tissue fibrosis, thrombosis, and/or the extracellular deposition of eosinophil-derived proteins [85] . The involvement of the heart can lead to an intraventricular thrombus because of the infiltration of eosinophils into the endomyocardium.…”

Section: Discussionmentioning

confidence: 99%

“…These findings indicate a potential role of eosinophils in vascular injury and inflammation [86] . Other clinical features of eosinophil-driven cardiovascular toxicity include endomyocardial fibrosis, eosinophil vasculitis and venous thromboembolism [85] , [87] , [88] . Eosinophil-mediated endomyocardial damage leads subendocardial eosinophilic infiltrates at the early, and generally asymptomatic stage.…”

Section: Discussionmentioning

confidence: 99%

Literature reviews of stroke with hypereosinophilic syndrome

Ono

Iwahana

Kato

et al. 2021

IJC Heart & Vasculature

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Hypereosinophilic syndrome (HES) is defined by persistently elevated blood eosinophil levels and is associated with evidence of organ damage. Cardiovascular involvement in HES is most commonly associated with Loffler endocarditis (cardiac HES). Cardiac HES is typically characterized by progressive subendocardial fibrosis with overlying mural thrombus formation, leading to restrictive dysfunction of the left ventricle. The thrombus from cardiac HES could result in cardiogenic stroke; however, most of the stroke cases with HES were not associated with huge thromboembolism rather multiple infarcts in the watershed area. The major clinical features of 97 previously reported cases of stroke with HES are as follows: the median age was 52 years, of which 61 (63%) were men; the initial presenting symptoms were neurological (73%), followed by headache (16%), respiratory symptoms (9%), and visual symptoms (9%). Almost half of the cases were diagnosed with cardiac HES. The characteristics of cardiac findings were mural thrombi, endomyocardial fibrosis, and a restrictive pattern of heart failure. Cerebral findings revealed 78 cases (80%) were described as multiple infarctions and 55 cases (57 %) were involved with watershed areas, whereas 11 cases (11％) were described as embolic stroke for one proximal large-vessel occlusion. Regarding treatment, 71 (73%), 28 (29%), and 16 (16%) patients were treated with steroids, anticoagulants, and antiplatelets, respectively. The overall mortality and recovery rates were 11% and 89%, respectively. Physicians should know most cases of stroke with HES are characterized by multiple infarctions in the watershed area, and cardiac HES is not always associated with stroke.

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“…1 From another perspective, 'idiopathic eosinophilic vasculitis' is also referred to in asthma-free patients with HES. 7 Digital ischaemia with an unproven trigger has previously been reported as a rare initial symptom of HES. Rohmer et al 8 presented 33 cases of distal ischaemia related to HES in the literature.…”

Section: Discussionmentioning

confidence: 99%

“…Eosinophil‐related organ damage involves fibrosis (lungs, heart, digestive tract and others), thrombosis or thromboembolism, neuropathy and others 1 . From another perspective, ‘idiopathic eosinophilic vasculitis’ is also referred to in asthma‐free patients with HES 7 . Digital ischaemia with an unproven trigger has previously been reported as a rare initial symptom of HES.…”

Section: Discussionmentioning

confidence: 99%

A case of hypereosinophilia with digital ischaemia successfully treated with intravenous prostaglandin E₁

Kaga

Takeuchi

Iwahara

et al. 2021

Skin Health and Disease

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In the therapeutic management of eosinophilic disorder, it is important to prevent hypereosinophilia (HE)-related organ damage even in the process of diagnosis. We describe here a unique clinical and histopathological findings of the patient with HE accompanied with digital ischaemia. Treatment with intravenous prostaglandin E 1 was essential for digital ischaemia in our case while benralizumab, humanized monoclonal antibody against interleukin-5 receptorα, did not affect. Our case suggests an earlier intervention for digital ischaemia in the therapeutic management of eosiniphilic disorder. | Diagnosis and treatmentEosinophil counts at an interval of 4 weeks in this case were 1.93 � 10 9 /L and 1.82 � 10 9 /L, respectively, fulfilling the diagnostic criteria for HE. Parasite infection was ruled out because he had no bowelThis is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

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“Idiopathic Eosinophilic Vasculitis”: Another Side of Hypereosinophilic Syndrome? A Comprehensive Analysis of 117 Cases in Asthma-Free Patients

Cited by 44 publications

References 98 publications

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

Literature reviews of stroke with hypereosinophilic syndrome

A case of hypereosinophilia with digital ischaemia successfully treated with intravenous prostaglandin E₁

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“Idiopathic Eosinophilic Vasculitis”: Another Side of Hypereosinophilic Syndrome? A Comprehensive Analysis of 117 Cases in Asthma-Free Patients

Cited by 44 publications

References 98 publications

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

Epidemiology, clinical picture and long‐term outcomes ofFIP1L1‐PDGFRA‐positive myeloid neoplasm with eosinophilia: Data from 151 patients

Literature reviews of stroke with hypereosinophilic syndrome

A case of hypereosinophilia with digital ischaemia successfully treated with intravenous prostaglandin E1

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A case of hypereosinophilia with digital ischaemia successfully treated with intravenous prostaglandin E₁