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Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature

INTRODUCTION: Vernal keratoconjunctivis (VKC) is a severe form of pediatric ocular allergy, characterized by acute and chronic corneoconjunctival inflammation that may lead to visual sequelae. Although topical immunosuppressive drugs such as cyclosporine are usually effective, some severe forms may...

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Autores principales: Doan, Serge, Amat, Flore, Gabison, Eric, Saf, Sarah, Cochereau, Isabelle, Just, Jocelyne
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Healthcare 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5449293/
https://www.ncbi.nlm.nih.gov/pubmed/27909980
http://dx.doi.org/10.1007/s40123-016-0074-2
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author Doan, Serge
Amat, Flore
Gabison, Eric
Saf, Sarah
Cochereau, Isabelle
Just, Jocelyne
author_facet Doan, Serge
Amat, Flore
Gabison, Eric
Saf, Sarah
Cochereau, Isabelle
Just, Jocelyne
author_sort Doan, Serge
collection PubMed
description INTRODUCTION: Vernal keratoconjunctivis (VKC) is a severe form of pediatric ocular allergy, characterized by acute and chronic corneoconjunctival inflammation that may lead to visual sequelae. Although topical immunosuppressive drugs such as cyclosporine are usually effective, some severe forms may be refractory and require prolonged steroid therapy. Very few papers report the use of omalizumab in VKC in the literature. In the present study, we describe our clinical experience with omalizumab in severe VKC children. METHODS: We retrospectively reviewed the files of four boys treated with omalizumab because of severe VKC, defined as persistent corneal inflammation despite continuous topical 2% cyclosporine and steroid eye drops. We also performed a literature review. RESULTS: Four boys, aged 7–13 years old, were treated. All children had asthma and one had severe lid eczema. Two patients had required intrapalpebral depot-steroid injections. Omalizumab was administered every 2 weeks by subcutaneous injections, at doses varying from 450 to 600 mg per injection. Three patients out of four responded to the treatment, with a decrease in global symptoms (median symptom rating decreasing from 89 to 29 on a 100-mm visual analog scale), frequency and in duration of the inflammatory flares, and also a decreased need for topical steroid. Their median clinical grade decreased from 4 to 3 (Bonini grading). However, the response was incomplete and they still had inflammatory corneoconjunctival flares despite continuous topical cyclosporine. On the other hand, asthma and lid eczema were completely controlled in these three patients. The fourth child did not respond to omalizumab and needed oral steroids for his VKC and his asthma. Noticeably, this latter patient did not have detectable sensitization to any allergen, contrary to the other cases. The treatment was stopped in this refractory case, but is still ongoing in all other cases, with a median duration of 33 months (range 16–42 months). In the literature (four cases), omalizumab may have a more complete efficacy in some cases, but the results are still variable. CONCLUSION: Omalizumab is an interesting treatment in severe refractory forms of VKC, but its efficacy is incomplete in these very severe cases.
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spelling pubmed-54492932017-06-15 Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature Doan, Serge Amat, Flore Gabison, Eric Saf, Sarah Cochereau, Isabelle Just, Jocelyne Ophthalmol Ther Case Series INTRODUCTION: Vernal keratoconjunctivis (VKC) is a severe form of pediatric ocular allergy, characterized by acute and chronic corneoconjunctival inflammation that may lead to visual sequelae. Although topical immunosuppressive drugs such as cyclosporine are usually effective, some severe forms may be refractory and require prolonged steroid therapy. Very few papers report the use of omalizumab in VKC in the literature. In the present study, we describe our clinical experience with omalizumab in severe VKC children. METHODS: We retrospectively reviewed the files of four boys treated with omalizumab because of severe VKC, defined as persistent corneal inflammation despite continuous topical 2% cyclosporine and steroid eye drops. We also performed a literature review. RESULTS: Four boys, aged 7–13 years old, were treated. All children had asthma and one had severe lid eczema. Two patients had required intrapalpebral depot-steroid injections. Omalizumab was administered every 2 weeks by subcutaneous injections, at doses varying from 450 to 600 mg per injection. Three patients out of four responded to the treatment, with a decrease in global symptoms (median symptom rating decreasing from 89 to 29 on a 100-mm visual analog scale), frequency and in duration of the inflammatory flares, and also a decreased need for topical steroid. Their median clinical grade decreased from 4 to 3 (Bonini grading). However, the response was incomplete and they still had inflammatory corneoconjunctival flares despite continuous topical cyclosporine. On the other hand, asthma and lid eczema were completely controlled in these three patients. The fourth child did not respond to omalizumab and needed oral steroids for his VKC and his asthma. Noticeably, this latter patient did not have detectable sensitization to any allergen, contrary to the other cases. The treatment was stopped in this refractory case, but is still ongoing in all other cases, with a median duration of 33 months (range 16–42 months). In the literature (four cases), omalizumab may have a more complete efficacy in some cases, but the results are still variable. CONCLUSION: Omalizumab is an interesting treatment in severe refractory forms of VKC, but its efficacy is incomplete in these very severe cases. Springer Healthcare 2016-12-01 2017-06 /pmc/articles/PMC5449293/ /pubmed/27909980 http://dx.doi.org/10.1007/s40123-016-0074-2 Text en © The Author(s) 2016 https://creativecommons.org/licenses/by-nc/4.0/This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License (http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) ), which permits any noncommercial use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.
spellingShingle Case Series
Doan, Serge
Amat, Flore
Gabison, Eric
Saf, Sarah
Cochereau, Isabelle
Just, Jocelyne
Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title_full Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title_fullStr Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title_full_unstemmed Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title_short Omalizumab in Severe Refractory Vernal Keratoconjunctivitis in Children: Case Series and Review of the Literature
title_sort omalizumab in severe refractory vernal keratoconjunctivitis in children: case series and review of the literature
topic Case Series
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5449293/
https://www.ncbi.nlm.nih.gov/pubmed/27909980
http://dx.doi.org/10.1007/s40123-016-0074-2
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