Literature DB >> 30269248

Follow-up and outcome of symptomatic partial or absolute IgA deficiency in children.

Viviana Moschese1, Loredana Chini2, Simona Graziani2, Mayla Sgrulletti2, Vera Gallo3, Gigliola Di Matteo2, Simona Ferrari4, Silvia Di Cesare2, Emilia Cirillo3, Andrea Pession5, Claudio Pignata3, Fernando Specchia5.   

Abstract

Selective IgA deficiency is defined as absolute or partial when serum IgA level is < 7 mg/dl or 2 SD below normal for age, respectively. Few data are available on partial selective IgA deficiency, as probably most children with low serum IgA are seldom referred to a specialist clinic in common pediatric practice. The aim of our study was to better define the profile of both symptomatic forms and their clinical outcome in a pediatric immunology setting. Thus, clinical and immunological data from 103 symptomatic patients with selective IgA deficiency (53 absolute and 50 partial), 4-18 years of age, were collected at diagnosis and 80 patients (44 absolute and 36 partial) were monitored for a mean period of 5 years. Also, the prevalence of TNFRSF13B mutations has been assessed in 56 patients. The most common clinical features were infections (86/103; 83%), allergy (39/103; 38%), and autoimmunity (13/103; 13%). No significative differences were observed between absolute and partial selective IgA deficiency patients. However, a significative difference in the rate of IgA normalization between partial and absolute selective IgA deficiency patients (33 vs 9%, p = 0.01) was detected. Furthermore, a lower incidence of infections was associated to a normalization reversal compared to a final absolute or partial defect status (12 vs 53 and 64% respectively, p < 0.01).Conclusions: Regardless of a diagnosis of absolute or partial defect, monitoring of symptomatic patients with selective IgA deficiency is recommended overtime for prompt identification and treatment of associated diseases. Further, diagnostic workup protocols should be revisited in children with IgA deficiency. What is Known: ● Selective IgA Deficiency is the most common primary immunodeficiency and is usually asymptomatic. ● Symptomatic pediatric patients with selective IgA deficiency mostly suffer with respiratory and gastrointestinal infections. What is New: ● Symptomatic children with partial IgA defect may have similar clinical, immunological, and genetic features than symptomatic children with absolute IgA deficiency. ● Symptomatic children with partial IgA deficiency deserve accurate monitoring for associated diseases as per children with absolute IgA deficiency.

Entities:  

Keywords:  Antibody deficiency; Primary immunodeficiency; Recurrent respiratory infections; TNFRSF13B

Mesh:

Substances:

Year:  2018        PMID: 30269248     DOI: 10.1007/s00431-018-3248-1

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  6 in total

1.  Clinical and Laboratory Features of 184 Italian Pediatric Patients Affected with Selective IgA Deficiency (SIgAD): a Longitudinal Single-Center Study.

Authors:  Vassilios Lougaris; Annamaria Sorlini; Chiara Monfredini; Giulia Ingrasciotta; Andrea Caravaggio; Tiziana Lorenzini; Manuela Baronio; Marco Cattalini; Antonella Meini; Laura Ruggeri; Annamaria Salpietro; Alba Pilotta; Livia Grazzani; Elena Prandi; Barbara Felappi; Giulio Gualdi; Antonella Fabiano; Maurizio Fuoti; Alberto Ravelli; Vincenzo Villanacci; Annarosa Soresina; Raffaele Badolato; Alessandro Plebani
Journal:  J Clin Immunol       Date:  2019-05-25       Impact factor: 8.317

2.  Treatment with Distinct Antibiotic Classes Causes Different Pulmonary Outcomes on Allergic Airway Inflammation Associated with Modulation of Symbiotic Microbiota.

Authors:  Gregório Grama Cavalcante; Anna Gabriella Guimarães; Camila Pereira Queiroz-Glauss; Marcela Helena Gonçalves Pereira; Angélica Samer Lallo Dias; Laila Sampaio Horta; Jamil Silvano de Oliveira; Silvia Dantas Cangussú; Paula Prazeres Magalhães; Remo Castro Russo; H C Santiago
Journal:  J Immunol Res       Date:  2022-06-22       Impact factor: 4.493

3.  Plasma therapy leads to an increase in functional IgA and IgM concentration in the blood and saliva of a patient with X-linked agammaglobulinemia.

Authors:  Jeroen D Langereis; Joannes F M Jacobs; Marien I de Jonge; Marcel van Deuren
Journal:  J Transl Med       Date:  2019-05-23       Impact factor: 5.531

Review 4.  Update on Infections in Primary Antibody Deficiencies.

Authors:  Yesim Yilmaz Demirdag; Sudhir Gupta
Journal:  Front Immunol       Date:  2021-02-11       Impact factor: 7.561

5.  The Allergic Phenotype of Children and Adolescents with Selective IgA Deficiency: A Longitudinal Monocentric Study.

Authors:  Bianca Laura Cinicola; Giulia Brindisi; Martina Capponi; Alessandra Gori; Lorenzo Loffredo; Giovanna De Castro; Caterina Anania; Alberto Spalice; Cristiana Alessia Guido; Cinzia Milito; Marzia Duse; Isabella Quinti; Federica Pulvirenti; Anna Maria Zicari
Journal:  J Clin Med       Date:  2022-09-27       Impact factor: 4.964

Review 6.  Selective IgA Deficiency and Allergy: A Fresh Look to an Old Story.

Authors:  Bianca Laura Cinicola; Federica Pulvirenti; Martina Capponi; Marta Bonetti; Giulia Brindisi; Alessandra Gori; Giovanna De Castro; Caterina Anania; Marzia Duse; Anna Maria Zicari
Journal:  Medicina (Kaunas)       Date:  2022-01-15       Impact factor: 2.430

  6 in total

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