Single-Organ and Multisystem Hypereosinophilic Syndrome Patients with Gastrointestinal Manifestations Share Common Characteristics

Kuang, Fei Li; Curtin, Bryan F.; Alao, Hawwa; Piligian, Brent; Berry, Andrea; Holland-Thomas, Nicole; Powers, Astin S.; Quezado, Martha; Lumbard, Keith; Fay, Michael P.; Klion, Amy D.; Kumar, Sheila; Khoury, Paneez

doi:10.1016/j.jaip.2020.04.025

Cited by 22 publications

(20 citation statements)

References 24 publications

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“…EGIDs are defined by marked eosinophilia in the gastrointestinal (GI) tract resulting in a wide variety of GI symptoms. When accompanied by blood hypereosinophilia (HE; AEC ≥ 1500/mm 3 ), EGID can occur as an isolated gastrointestinal disorder (hypereosinophilic syndrome/EGID overlap) or as part of a multisystem hypereosinophilic syndrome 152 . In such syndromes, food allergy is frequent.…”

Section: Monoclonal Antibodies Showing Promise In Clinical Trials Formentioning

confidence: 99%

The use of biologics in food allergy

Fiocchi

Vickery

Wood

2021

Clin Experimental Allergy

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Background Food allergy continues to pose problems due to its increased frequency and its increasingly high severity. In this context, alongside the traditional avoidance strategies of allergenic foods and desensitization through the cautious progression of exposure to foods in the context of oral immunotherapy (OIT), alternative strategies have made their way in the last decades. We review the possibilities of intervention in food allergy with the use of biological drugs capable of interfering with the synthesis of IgE, with their mechanisms of action, or with complex biological mechanisms that lead to the establishment of a food allergy. Methods Repeated Entrez PubMed searches using the template algorithm “Food allergy” and “biologics” or “Omalizumab” or “Dupilumab” or “milk desensitization” or “oral tolerance induction” or “oral immunotherapy” or “Etokimab” or “Tezepelumab” or “Quilizumab” or “Ligelizumab” or “Tralokinumab” or “Nemolizumab” or “Mepolizumab” or “Reslizumab” or “Benralizumab”. The authors’ clinical experience in paediatric allergy units of University hospitals was also drawn upon. Results The landscape in this context has changed dramatically over the past 10 years. We have acquired knowledge mainly on the effect of different types of anti‐IgE treatments in poliallergic patients with food allergy, and in patients treated with OIT. However, other mediators are being targeted by specific biologic treatments. Among them, the alarmins Il‐33 and TSLP, IL‐4 and IL‐13, eosinophil‐related molecules as IL‐6, IL‐8, IL‐10, IL‐12, and mostly IL‐5, and integrins involved in the pathogenesis of eosinophilic gastrointestinal diseases (EGIDs), as SIGLEC‐8. Conclusions The ever‐better knowledge of the mechanisms of food allergy allowing these developments will improve not only the perspective of patients with the most serious immediate food allergies such as anaphylaxis, but also those of patients with related diseases such as atopic dermatitis, eosinophilic esophagitis, and EGIDs. Biologics are also intended to complement OIT strategies that have developed over the years.

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Section: Monoclonal Antibodies Showing Promise In Clinical Trials Formentioning

confidence: 99%

The use of biologics in food allergy

Fiocchi

Vickery

Wood

2021

Clin Experimental Allergy

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“…Gastrointestinal manifestations are some of the most frequent and debilitating aspects of HES [ 26 ]. The endoscopic features may include esophageal furrows, rings, plaques, mucosal erythema, and ulceration [ 27 ]. Gastroenterological symptoms in both HES with multiorgan system involvement and localized eosinophilic gastrointestinal disease may often be discordant with endoscopic or histopathologic appearance, as in the case of other inflammatory bowel diseases such as Crohn’s disease and ulcerative colitis [ 27 ].…”

Section: Episodic Angioedema With Hypereosinophilia Clinical and Lmentioning

confidence: 99%

“…The endoscopic features may include esophageal furrows, rings, plaques, mucosal erythema, and ulceration [ 27 ]. Gastroenterological symptoms in both HES with multiorgan system involvement and localized eosinophilic gastrointestinal disease may often be discordant with endoscopic or histopathologic appearance, as in the case of other inflammatory bowel diseases such as Crohn’s disease and ulcerative colitis [ 27 ]. In eosinophilic esophagitis, the endoscopic appearance of the esophagus may be normal in 10% to 25% of patients; for this reason, esophageal biopsies are recommended in patients in whom there is clinical suspicion of eosinophilic involvement of the esophagus, even if there are no endoscopic alterations of the esophageal mucosa [ 28 , 29 ].…”

Section: Episodic Angioedema With Hypereosinophilia Clinical and Lmentioning

confidence: 99%

Episodic Angioedema with Hypereosinophilia (Gleich’s Syndrome): A Case Report and Extensive Review of the Literature

Mormile

Petraroli

Loffredo

et al. 2021

JCM

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Episodic angioedema with eosinophilia (EAE) (Gleich’s syndrome) is a rare disease characterized by hypereosinophilia (up to 95 × 109 cells/L), recurrent episodes of angioedema, urticaria, weight gain, and fever, that occur at periodical intervals (usually every 3–4 weeks). The exact etiology of EAE is still unclear, but both eosinophils and abnormalities of cytokines homeostasis seem to play a pivotal role in the pathogenesis of the disease. In particular, the cyclic elevation of serum interleukin-5 before the increase in eosinophil count has been reported. Herein, we performed a broad literature review and report the case of a thirty-two-year-old woman with a two-year history of cyclic angioedema attacks, urticaria, periodic weight gain, and severe hypereosinophilia, diagnosed with EAE and treated with oral corticosteroids. Describing the most relevant clinical features of EAE reported so far in the literature, we aim to provide physicians with some useful tools to help them deal with this disease. In addition, we aim to raise awareness about this rare condition in which approved diagnostic classification criteria are currently missing.

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“…EGID can occur as an isolated disorder associated with hypereosinophilic syndrome (HES/EGID overlap) or can be part of a multisystem HES. The clinical presentation and management of both groups are remarkably similar [ 5 ].…”

Section: Introductionmentioning

confidence: 99%

Organ-Specific Hypereosinophilic Syndrome Presenting as Eosinophilic Gastroenteritis

Kumar¹,

Haider²,

Siddiqa³

2021

Cureus

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Gastrointestinal (GI) diseases have a substantial impact on the population health and healthcare resources of the United States. They constitute billions of dollars in expenditure and millions of office and hospital visits. With advancing diagnostic and treatment modalities, rare diseases are increasingly recognized and managed. However, after close to 80 years since the first description, eosinophilic GI disorders (EGID) are still uncommon, and only around 300 cases have been reported to date. Hypereosinophilic syndrome (HES) is well studied, but there are still no guidelines to direct management. We report the case of a 56-year-old female who presented with gastroenteritis and a persistent eosinophil count above 7 x 10 9 /L. Imaging was suggestive of bowel wall thickening, and endoscopy revealed normal-appearing mucosa. However, histologic examination revealed eosinophilic infiltration of the GI tract. She was diagnosed with HES and treated with oral prednisone with remarkable improvement of her symptoms.

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Single-Organ and Multisystem Hypereosinophilic Syndrome Patients with Gastrointestinal Manifestations Share Common Characteristics

Cited by 22 publications

References 24 publications

The use of biologics in food allergy

The use of biologics in food allergy

Episodic Angioedema with Hypereosinophilia (Gleich’s Syndrome): A Case Report and Extensive Review of the Literature

Organ-Specific Hypereosinophilic Syndrome Presenting as Eosinophilic Gastroenteritis

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