Eosinophilic annular erythema showing eosinophil cytolytic ETosis successfully treated with benralizumab

Abstract

A 56-year-old woman presented with repeated swelling of the lips and face. She had a history of childhood asthma; she had a recurrence of asthma when she was 54 years old and was taking inhaled corticosteroids, and other antiasthma drugs. The swelling of her lips and face improved temporarily with oral corticosteroids (OCS), but recurred soon after discontinuing OCS. Her peripheral blood eosinophil count was 632/μL (9.3%), and her serum was negative for myeloperoxidase-anti-neutrophil cytoplasmic antibody and serine proteinase3-anti-neutrophil cytoplasmic antibody. Hematoxylin and eosin staining of her back skin revealed abundant eosinophilic infiltrate around the vascular area of the shallow dermis layer, but no evidence of vasculitis and we diagnosed her as eosinophilic annular erythema (EAE). Punctate staining of galectin-10, chromatolytic eosinophils, and net-like DNA was also evident in close proximity to the free granules, indicating extracellular vesicles and eosinophil extracellular traps (ETosis). We started daily OCS to control her asthma and skin eruption/oedema. Three months after administering daily OCS, benralizumab was initiated for withdrawal from OCS dependence and treatment of severe asthma. After initiation of benralizumab, her skin and subcutaneous tissue symptoms promptly improved. OCS was discontinued, and no edematous erythema has been observed since then. Bronchial asthma has also been well-controlled. This is the first report on the evidence of eosinophil ETosis in the dermis of EAE patients and the efficacy of benralizumab in a patient with EAE. Benralizumab may be a useful drug for treating refractory EAE with severe eosinophilic asthma.

Case Report: Eosinophilic Bronchiolitis With Eosinophil ETosis in Mucus Plugs Successfully Treated With Benralizumab

“…In a previous case of ABPM, switching from mepolizumab to benralizumab resulted in rapid disappearance of mucus plugs (Tomomatsu et al, 2020). In addition, the therapeutic efficacy of benralizumab has been demonstrated in patients with EETosisoccurring eosinophilic annular erythema and chronic eosinophilic pneumonia (Masaki et al, 2021;Takano et al, 2021). Although a reported case of steroid-resistant eosinophilic bronchiolitis showed good therapeutic response to mepolizumab, benralizumab might be an additional option for the treatment of eosinophilic bronchiolitis.…”

Treatment of Eosinophilic Annular Erythema With Benralizumab

“…Another patient accomplished resolution with treatment with mepolizumab, an IL-5 inhibitor . Finally there is 1 case report of a patient with severe asthma and cutaneous EAE who, after initiating treatment with benralizumab for her asthma, experienced improvement of her cutaneous lesions . While further research is needed, antibodies targeting the IL-5 signaling pathway could be an effective therapy for patients with recalcitrant EAE.…”

Bronquiolitis eosinofílica con ETosis por eosinófilos en tapones mucosos tratada con éxito con benralizumab

“…En un caso anterior de ABPM, el cambio de mepolizumab por benralizumab dio lugar a una rápida desaparición de los tapones de moco [16]. Además, se ha demostrado la eficacia terapéutica del benralizumab en pacientes con eritema anular eosinofílico y neumonía eosinofílica crónica [17, 18]. Aunque se ha reportado un caso de bronquiolitis eosinofílica resistente a los esteroides con buena respuesta terapéutica al mepolizumab, el benralizumab podría ser una opción adicional para el tratamiento de la bronquiolitis eosinofílica.…”