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Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency

Chronic spontaneous urtcaria (CSU) can represent the leading sign of a wide spectrum of systemic diseases, including primary immunodeficiencies. We describe the case of a young adult female with coexisting CSU and common variable immunodeficiency (CVID) successfully treated with omalizumab. The pati...

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Autores principales: Comberiati, Pasquale, Costagliola, Giorgio, Carli, Niccolò, Legitimo, Annalisa, D'Elios, Sofia, Consolini, Rita, Peroni, Diego G.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6652742/
https://www.ncbi.nlm.nih.gov/pubmed/31379880
http://dx.doi.org/10.3389/fimmu.2019.01700
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author Comberiati, Pasquale
Costagliola, Giorgio
Carli, Niccolò
Legitimo, Annalisa
D'Elios, Sofia
Consolini, Rita
Peroni, Diego G.
author_facet Comberiati, Pasquale
Costagliola, Giorgio
Carli, Niccolò
Legitimo, Annalisa
D'Elios, Sofia
Consolini, Rita
Peroni, Diego G.
author_sort Comberiati, Pasquale
collection PubMed
description Chronic spontaneous urtcaria (CSU) can represent the leading sign of a wide spectrum of systemic diseases, including primary immunodeficiencies. We describe the case of a young adult female with coexisting CSU and common variable immunodeficiency (CVID) successfully treated with omalizumab. The patient, with a history of recurrent respiratory infections during childhood, was referred to clinical attention due to the development of refractory CSU. During the diagnostic workup for the research of secondary causes of urticaria, an immunological assessment was performed, showing markedly reduced levels of IgG and IgM, poor antibody response against vaccinating antigens in absence of a T cellular deficiency. Therefore, the diagnosis of CVID was posed. Despite the immunoglobulin replacement and a trial with intravenous immunoglobulin at immunomodulatory dosage, the patient continued to experience severe urticaria, with significant impairment in the quality of life. After 2 years from the diagnosis of CVID, a treatment with omalizumab was started, showing complete remission of cutaneous symptoms after the first injection. The drug was well-tolerated, and the patient did not experience adverse effects during a 12-months follow-up.
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spelling pubmed-66527422019-08-02 Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency Comberiati, Pasquale Costagliola, Giorgio Carli, Niccolò Legitimo, Annalisa D'Elios, Sofia Consolini, Rita Peroni, Diego G. Front Immunol Immunology Chronic spontaneous urtcaria (CSU) can represent the leading sign of a wide spectrum of systemic diseases, including primary immunodeficiencies. We describe the case of a young adult female with coexisting CSU and common variable immunodeficiency (CVID) successfully treated with omalizumab. The patient, with a history of recurrent respiratory infections during childhood, was referred to clinical attention due to the development of refractory CSU. During the diagnostic workup for the research of secondary causes of urticaria, an immunological assessment was performed, showing markedly reduced levels of IgG and IgM, poor antibody response against vaccinating antigens in absence of a T cellular deficiency. Therefore, the diagnosis of CVID was posed. Despite the immunoglobulin replacement and a trial with intravenous immunoglobulin at immunomodulatory dosage, the patient continued to experience severe urticaria, with significant impairment in the quality of life. After 2 years from the diagnosis of CVID, a treatment with omalizumab was started, showing complete remission of cutaneous symptoms after the first injection. The drug was well-tolerated, and the patient did not experience adverse effects during a 12-months follow-up. Frontiers Media S.A. 2019-07-17 /pmc/articles/PMC6652742/ /pubmed/31379880 http://dx.doi.org/10.3389/fimmu.2019.01700 Text en Copyright © 2019 Comberiati, Costagliola, Carli, Legitimo, D'Elios, Consolini and Peroni. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Immunology
Comberiati, Pasquale
Costagliola, Giorgio
Carli, Niccolò
Legitimo, Annalisa
D'Elios, Sofia
Consolini, Rita
Peroni, Diego G.
Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title_full Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title_fullStr Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title_full_unstemmed Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title_short Refractory Chronic Spontaneous Urticaria Treated With Omalizumab in an Adolescent With Common Variable Immunodeficiency
title_sort refractory chronic spontaneous urticaria treated with omalizumab in an adolescent with common variable immunodeficiency
topic Immunology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6652742/
https://www.ncbi.nlm.nih.gov/pubmed/31379880
http://dx.doi.org/10.3389/fimmu.2019.01700
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