Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab

<h3>Background</h3> <p>Autosomal recessive congenital ichthyosis refers to a group of rare inherited disorders of keratinization and defective epidermal barrier resulting in varying clinical presentations and severities ranging from harlequin ichthyosis to congenital ichthyosiform...

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Main Author: M. Steinhoff (14779126) (author)
Other Authors: F. Al‐Marri (14779129) (author), R. Al Chalabi (14779132) (author), U. Gieler (14779135) (author), J. Buddenkotte (14779138) (author)
Published: 2023
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author M. Steinhoff (14779126)
author2 F. Al‐Marri (14779129)
R. Al Chalabi (14779132)
U. Gieler (14779135)
J. Buddenkotte (14779138)
author2_role author
author
author
author
author_facet M. Steinhoff (14779126)
F. Al‐Marri (14779129)
R. Al Chalabi (14779132)
U. Gieler (14779135)
J. Buddenkotte (14779138)
author_role author
dc.creator.none.fl_str_mv M. Steinhoff (14779126)
F. Al‐Marri (14779129)
R. Al Chalabi (14779132)
U. Gieler (14779135)
J. Buddenkotte (14779138)
dc.date.none.fl_str_mv 2023-03-16T06:24:45Z
dc.identifier.none.fl_str_mv 10.1002/ski2.87
dc.relation.none.fl_str_mv https://figshare.com/articles/journal_contribution/Recalcitrant_erythrodermic_ichthyosis_with_atopic_dermatitis_successfully_treated_with_Dupilumab_in_combination_with_Guselkumab/22258396
dc.rights.none.fl_str_mv CC BY 4.0
info:eu-repo/semantics/openAccess
dc.subject.none.fl_str_mv Biomedical and clinical sciences
Clinical sciences
Skin Health and Disease
dc.title.none.fl_str_mv Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
dc.type.none.fl_str_mv Text
Journal contribution
info:eu-repo/semantics/publishedVersion
text
contribution to journal
description <h3>Background</h3> <p>Autosomal recessive congenital ichthyosis refers to a group of rare inherited disorders of keratinization and defective epidermal barrier resulting in varying clinical presentations and severities ranging from harlequin ichthyosis to congenital ichthyosiform erythroderma (CIE). Secondary atopic dermatitis (AD) can aggravate the disease state for CIE patients leading to recalcitrant CIE/AD with potentially unfavourable side effects and low tolerability.</p> <h3>Aims</h3> <p>Here, we report about a 38-year-old male patient with severe CIE as well as AD over the last 30 years.</p> <h3>Materials and Methods</h3> <p>The patients suffered from severe inflammation, pruritus and recurrent infections for decades without disease control and intolerable adverse events of previous therapies.</p> <h3>Results</h3> <p>Dupilumab (targeting IL-4Ra, 300 mg q2w) partially controlled pruritus, but only the combination of Dupilumab with Guselkumab (anti-IL23p19) controlled both CIE and AD with markedly reduced inflammation, itch and recurrent infections. Guselkumab alone was not sufficient to treat the severe CIE/AD.</p> <h3>Discussion</h3> <p>Further studies are required to assess the efficacy and safety of targeted therapies like Dupilumab/Guselkumab combination therapy in severe CIE/AD.</p> <h2>Other Information</h2> <p>Published in: Skin Health and Disease<br> License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br> See article on publisher's website: <a href="http://dx.doi.org/10.1002/ski2.87" target="_blank">http://dx.doi.org/10.1002/ski2.87</a></p>
eu_rights_str_mv openAccess
id Manara2_693468b2fb246385b1c5547d4afc7d30
identifier_str_mv 10.1002/ski2.87
network_acronym_str Manara2
network_name_str Manara2
oai_identifier_str oai:figshare.com:article/22258396
publishDate 2023
repository.mail.fl_str_mv
repository.name.fl_str_mv
repository_id_str
rights_invalid_str_mv CC BY 4.0
spelling Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with GuselkumabM. Steinhoff (14779126)F. Al‐Marri (14779129)R. Al Chalabi (14779132)U. Gieler (14779135)J. Buddenkotte (14779138)Biomedical and clinical sciencesClinical sciencesSkin Health and Disease<h3>Background</h3> <p>Autosomal recessive congenital ichthyosis refers to a group of rare inherited disorders of keratinization and defective epidermal barrier resulting in varying clinical presentations and severities ranging from harlequin ichthyosis to congenital ichthyosiform erythroderma (CIE). Secondary atopic dermatitis (AD) can aggravate the disease state for CIE patients leading to recalcitrant CIE/AD with potentially unfavourable side effects and low tolerability.</p> <h3>Aims</h3> <p>Here, we report about a 38-year-old male patient with severe CIE as well as AD over the last 30 years.</p> <h3>Materials and Methods</h3> <p>The patients suffered from severe inflammation, pruritus and recurrent infections for decades without disease control and intolerable adverse events of previous therapies.</p> <h3>Results</h3> <p>Dupilumab (targeting IL-4Ra, 300 mg q2w) partially controlled pruritus, but only the combination of Dupilumab with Guselkumab (anti-IL23p19) controlled both CIE and AD with markedly reduced inflammation, itch and recurrent infections. Guselkumab alone was not sufficient to treat the severe CIE/AD.</p> <h3>Discussion</h3> <p>Further studies are required to assess the efficacy and safety of targeted therapies like Dupilumab/Guselkumab combination therapy in severe CIE/AD.</p> <h2>Other Information</h2> <p>Published in: Skin Health and Disease<br> License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br> See article on publisher's website: <a href="http://dx.doi.org/10.1002/ski2.87" target="_blank">http://dx.doi.org/10.1002/ski2.87</a></p>2023-03-16T06:24:45ZTextJournal contributioninfo:eu-repo/semantics/publishedVersiontextcontribution to journal10.1002/ski2.87https://figshare.com/articles/journal_contribution/Recalcitrant_erythrodermic_ichthyosis_with_atopic_dermatitis_successfully_treated_with_Dupilumab_in_combination_with_Guselkumab/22258396CC BY 4.0info:eu-repo/semantics/openAccessoai:figshare.com:article/222583962023-03-16T06:24:45Z
spellingShingle Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
M. Steinhoff (14779126)
Biomedical and clinical sciences
Clinical sciences
Skin Health and Disease
status_str publishedVersion
title Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
title_full Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
title_fullStr Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
title_full_unstemmed Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
title_short Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
title_sort Recalcitrant erythrodermic ichthyosis with atopic dermatitis successfully treated with Dupilumab in combination with Guselkumab
topic Biomedical and clinical sciences
Clinical sciences
Skin Health and Disease