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Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab

Lamellar ichthyosis (LI) is a rare autosomal cornification disorder, with most cases due to a mutation in the transglutaminase-1 (TGM1) gene on chromosome 14. Patients with LI usually present with a collodion membrane and mild erythroderma at birth, with the collodion membranes shedding within the f...

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Autores principales: Binkhonain, Faisal K, Aldokhayel, Sara, BinJadeed, Hessah, Madani, Abdulaziz
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9236574/
https://www.ncbi.nlm.nih.gov/pubmed/35769328
http://dx.doi.org/10.2147/BTT.S362391
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author Binkhonain, Faisal K
Aldokhayel, Sara
BinJadeed, Hessah
Madani, Abdulaziz
author_facet Binkhonain, Faisal K
Aldokhayel, Sara
BinJadeed, Hessah
Madani, Abdulaziz
author_sort Binkhonain, Faisal K
collection PubMed
description Lamellar ichthyosis (LI) is a rare autosomal cornification disorder, with most cases due to a mutation in the transglutaminase-1 (TGM1) gene on chromosome 14. Patients with LI usually present with a collodion membrane and mild erythroderma at birth, with the collodion membranes shedding within the first weeks of life and being replaced by a generalized scale. Typically, LI is managed with oral retinoids, emollients, and keratolytic agents, eg, lactic acid. We report an LI case associated with atopic dermatitis and asthma that showed a marked improvement with dupilumab treatment. This finding is highly significant as it may represent a breakthrough in the treatment of LI, thus more research is needed to investigate the potential benefits of dupilumab for the treatment of ichthyosis, such as the effects observed in our patient.
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spelling pubmed-92365742022-06-28 Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab Binkhonain, Faisal K Aldokhayel, Sara BinJadeed, Hessah Madani, Abdulaziz Biologics Case Report Lamellar ichthyosis (LI) is a rare autosomal cornification disorder, with most cases due to a mutation in the transglutaminase-1 (TGM1) gene on chromosome 14. Patients with LI usually present with a collodion membrane and mild erythroderma at birth, with the collodion membranes shedding within the first weeks of life and being replaced by a generalized scale. Typically, LI is managed with oral retinoids, emollients, and keratolytic agents, eg, lactic acid. We report an LI case associated with atopic dermatitis and asthma that showed a marked improvement with dupilumab treatment. This finding is highly significant as it may represent a breakthrough in the treatment of LI, thus more research is needed to investigate the potential benefits of dupilumab for the treatment of ichthyosis, such as the effects observed in our patient. Dove 2022-06-23 /pmc/articles/PMC9236574/ /pubmed/35769328 http://dx.doi.org/10.2147/BTT.S362391 Text en © 2022 Binkhonain et al. https://creativecommons.org/licenses/by-nc/3.0/This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/ (https://creativecommons.org/licenses/by-nc/3.0/) ). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms (https://www.dovepress.com/terms.php).
spellingShingle Case Report
Binkhonain, Faisal K
Aldokhayel, Sara
BinJadeed, Hessah
Madani, Abdulaziz
Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title_full Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title_fullStr Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title_full_unstemmed Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title_short Successful Treatment of an Adult with Atopic Dermatitis and Lamellar Ichthyosis Using Dupilumab
title_sort successful treatment of an adult with atopic dermatitis and lamellar ichthyosis using dupilumab
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9236574/
https://www.ncbi.nlm.nih.gov/pubmed/35769328
http://dx.doi.org/10.2147/BTT.S362391
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