Cinicola Bianca Laura, Brindisi Giulia, Capponi Martina, Gori Alessandra, Loffredo Lorenzo, De Castro Giovanna, Anania Caterina, Spalice Alberto, Guido Cristiana Alessia, Milito Cinzia, Duse Marzia, Quinti Isabella, Pulvirenti Federica, Zicari Anna Maria
Department of Maternal Infantile and Urological Sciences, Sapienza University of Rome, 00161 Rome, Italy.
Department of Molecular Medicine, Sapienza University of Rome, 00161 Rome, Italy.
J Clin Med. 2022 Sep 27;11(19):5705. doi: 10.3390/jcm11195705.
Selective IgA deficiency (SIgAD) is the most common inborn error of immunity. The exact prevalence and pathogenesis of allergy in SIgAD have not yet been defined. We aimed to describe the prevalence and the characteristics of allergy in pediatric SIgAD subjects, evaluate the association between allergy and other comorbidities, and define the immune phenotype of allergic and non-allergic patients.
Clinical and immunological data from 67 SIgAD patients were collected over a 13-year period at a single center. Patients' characteristics were analyzed according to the presence of allergy.
Allergy was diagnosed in 34% of SIgAD patients, with a median age at allergy diagnosis of 8 years. Allergy was the second-most-common clinical manifestation, following recurrent respiratory infections. Among the allergic group, 74% had rhinitis, 30% asthma, 30% atopic dermatitis, and 22% food allergy; one out of three had more than one allergic manifestation. SIgAD patients showed more frequent transitory lymphopenia and a lower count of CD19+ at diagnosis than at last FU. However, compared to non-allergic subjects, allergic patients did not differ in their immune phenotype, number and severity of infections, or increased autoimmunity.
In our longitudinal study, compared to non-allergic SIgAD patients, those with allergies did not present a more severe immune defect or complex clinical phenotype. However, evaluation and early identification of allergy in the context of SIgAD assessment, both at diagnosis and during FU, and definition of a proper management are important to prevent complications and improve the patient's quality of life.
选择性IgA缺乏症(SIgAD)是最常见的先天性免疫缺陷病。SIgAD中过敏的确切患病率和发病机制尚未明确。我们旨在描述小儿SIgAD患者中过敏的患病率和特征,评估过敏与其他合并症之间的关联,并明确过敏和非过敏患者的免疫表型。
在13年期间,于单一中心收集了67例SIgAD患者的临床和免疫学数据。根据是否存在过敏对患者特征进行分析。
34%的SIgAD患者被诊断为过敏,过敏诊断的中位年龄为8岁。过敏是第二常见的临床表现,仅次于反复呼吸道感染。在过敏组中,74%患有鼻炎,30%患有哮喘,30%患有特应性皮炎,22%患有食物过敏;三分之一的患者有不止一种过敏表现。SIgAD患者在诊断时比末次随访时更频繁地出现短暂性淋巴细胞减少和CD19+计数降低。然而,与非过敏受试者相比,过敏患者在免疫表型、感染的数量和严重程度或自身免疫增加方面并无差异。
在我们的纵向研究中,与非过敏的SIgAD患者相比,过敏患者并未表现出更严重的免疫缺陷或复杂的临床表型。然而,在SIgAD评估过程中,无论是在诊断时还是随访期间,对过敏进行评估和早期识别以及确定适当的管理措施对于预防并发症和提高患者生活质量都很重要。