Omalizumab for the treatment of hyperimmunoglobulin E syndrome: A 12-year-old case

Akbaş, Pırıl Etikan; Kocatürk, Emek; Tüzün, Binnur; Köklü, Nagihan; Yazıcı, Ayça Cordan; Topal, İlteriş Oğuz

doi:10.1016/j.dsi.2016.07.002

Cited by 2 publications

(1 citation statement)

References 6 publications

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“…Moreover, SLE-like autoimmunity with predominant renal involvement have been identified in a cohort of patients with AD-HIES ( Goel et al, 2021 ). There are several reports on the use of omalizumab in HIES patients, mainly with recalcitrant skin lesions, with improvement of symptoms in all cases ( Bard et al, 2008 ; Marcotte, 2008 ; Chularojanamontri et al, 2009 ; Akbaş et al, 2017 ; Alonso-Bello et al, 2019 ; Gomes et al, 2020 ; Lan et al, 2022 ), but to provide ample evidence further studies and long-term follow-up are necessary. Increased serum IgE levels can also be seen in other primary immunodeficiencies, the main of which are two immune disregulation syndromes, autoimmune lymphoproliferative syndrome (ALPS) and the immunodysregulation, polyendrocrinopathy and enteropathy X‐linked (IPEX) syndrome.…”

Section: Diseases Where Self-reactive Ige Is Known To Have a Pathogen...mentioning

confidence: 99%

Self-reactive IgE and anti-IgE therapy in autoimmune diseases

Olewicz‐Gawlik

Kowala‐Piaskowska

2023

Front. Pharmacol.

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Growing evidence indicates the pathogenic role of autoreactive IgE in autoimmune diseases. Incidence of autoimmune and allergic diseases in the industrialized countries is consistently icreasing, thus leading to concerted efforts to comprehend the regulation of IgE-mediated mechanisms. The first reports of a presence of IgE autoantibodies in patients with autoimmune diseases have been published a long time ago, and it is now recognized that self-reactive IgE can mediate inflammatory response in bullous pemhigoid, systemic lupus erythematosus, chronic urticaria, and atopic dermatitis. The advances in understanding the pathomechanisms of these disorders brought to a successful use of anti-IgE strategies in their management. The present review discusses the current state of knowledge on the IgE-mediated autoimmunity and anti-IgE treatment, and pave the way for further exploration of the subject.

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Section: Diseases Where Self-reactive Ige Is Known To Have a Pathogen...mentioning

confidence: 99%

Self-reactive IgE and anti-IgE therapy in autoimmune diseases

Olewicz‐Gawlik

Kowala‐Piaskowska

2023

Front. Pharmacol.

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Omalizumab for STAT3 Hyper-IgE Syndromes in Adulthood: A Case Report and Literature Review

Zhang

Song

et al. 2022

Front. Med.

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BackgroundHyper-immunoglobulin E (IgE) syndromes (HIES) are a group of primary immune deficiencies disorders (PID) characterized by elevated serum IgE, eczema, recurrent skin, or respiratory system infections and may also be accompanied by some connective tissues and skeletal abnormalities. Currently, there is no complete cure or targeted treatment for HIES. Omalizumab is a humanized recombinant monoclonal antibody against IgE, reducing the level of free IgE, inhibiting the binding of IgE to receptors on the surface of effector cells, and reducing the activation of inflammatory cells and the release of multiple inflammatory mediators. However, the effect of omalizumab in treating HIES remains unknown. Herein, we described a case of an AD-HIES patient with chronic airway disease who benefited from omalizumab treatment.Case PresentationA 28-year-old Chinese woman was admitted for recurrent cough for 7 years, markedly elevated serum IgE level, and recurrent pneumonia caused by multiple pathogens, such as Pneumocystis jirovecii, Cytomegalovirus, Staphylococcus aureus, Aspergillus, and Mycobacterium tuberculosis. She had eczema-dermatitis, skin abscess, slightly traumatic fracture since childhood, and developed asthma and allergic bronchopulmonary aspergillosis (ABPA) lately. Using whole-exome sequencing, the STAT3 (c.1294G>T, p.Val432Leu) missense mutation for the autosomal dominant hyper-IgE syndrome was identified, and omalizumab was prescribed at 300 mg every 2 weeks. The patient responded well with the improvement of respiratory symptoms and lung function tests. The level of serum IgE remained stable on follow-up.ConclusionOmalizumab treatment proved beneficial in the case of HIES, especially with chronic airway disease, for which therapeutic options are limited. However, larger-scale prospective studies and long-term follow-up are required to establish the efficacy and safety of this therapeutic intervention.

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Omalizumab for the treatment of hyperimmunoglobulin E syndrome: A 12-year-old case

Cited by 2 publications

References 6 publications

Self-reactive IgE and anti-IgE therapy in autoimmune diseases

Self-reactive IgE and anti-IgE therapy in autoimmune diseases

Omalizumab for STAT3 Hyper-IgE Syndromes in Adulthood: A Case Report and Literature Review

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