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Familial Dyskeratotic Comedones: A Case Report and Literature Review

Familial dyskeratotic comedones (FDC) is an autosomal dominant inherited skin disorder characterized by generalized multiple discrete comedone-like hyperkeratotic papules. The disease demonstrates a distinct histopathologic feature of dyskeratosis of the crater-like invaginated epidermis or follicle...

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Autores principales: Tejapira, Kasama, Suchonwanit, Poonkiat
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10328097/
https://www.ncbi.nlm.nih.gov/pubmed/37426083
http://dx.doi.org/10.2147/CCID.S420723
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author Tejapira, Kasama
Suchonwanit, Poonkiat
author_facet Tejapira, Kasama
Suchonwanit, Poonkiat
author_sort Tejapira, Kasama
collection PubMed
description Familial dyskeratotic comedones (FDC) is an autosomal dominant inherited skin disorder characterized by generalized multiple discrete comedone-like hyperkeratotic papules. The disease demonstrates a distinct histopathologic feature of dyskeratosis of the crater-like invaginated epidermis or follicle-like structures with or without acantholysis. Despite its asymptomatic and benign course, the condition is refractory to treatment. Herein, we report a case of a 54-year-old female presenting with progressively developed generalized multiple hyperkeratotic papules with central keratin plugs on the trunk and extremities for 20 years. A definite diagnosis was made by clinical manifestations and histopathological examination. The lesions were slightly improved after 3 months of topical retinoids and urea cream treatments. Besides, we first describe dermoscopic findings of FDC and reviewed 21 previously reported FDC cases from 11 families in the literature.
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spelling pubmed-103280972023-07-08 Familial Dyskeratotic Comedones: A Case Report and Literature Review Tejapira, Kasama Suchonwanit, Poonkiat Clin Cosmet Investig Dermatol Case Report Familial dyskeratotic comedones (FDC) is an autosomal dominant inherited skin disorder characterized by generalized multiple discrete comedone-like hyperkeratotic papules. The disease demonstrates a distinct histopathologic feature of dyskeratosis of the crater-like invaginated epidermis or follicle-like structures with or without acantholysis. Despite its asymptomatic and benign course, the condition is refractory to treatment. Herein, we report a case of a 54-year-old female presenting with progressively developed generalized multiple hyperkeratotic papules with central keratin plugs on the trunk and extremities for 20 years. A definite diagnosis was made by clinical manifestations and histopathological examination. The lesions were slightly improved after 3 months of topical retinoids and urea cream treatments. Besides, we first describe dermoscopic findings of FDC and reviewed 21 previously reported FDC cases from 11 families in the literature. Dove 2023-07-03 /pmc/articles/PMC10328097/ /pubmed/37426083 http://dx.doi.org/10.2147/CCID.S420723 Text en © 2023 Tejapira and Suchonwanit. 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
Tejapira, Kasama
Suchonwanit, Poonkiat
Familial Dyskeratotic Comedones: A Case Report and Literature Review
title Familial Dyskeratotic Comedones: A Case Report and Literature Review
title_full Familial Dyskeratotic Comedones: A Case Report and Literature Review
title_fullStr Familial Dyskeratotic Comedones: A Case Report and Literature Review
title_full_unstemmed Familial Dyskeratotic Comedones: A Case Report and Literature Review
title_short Familial Dyskeratotic Comedones: A Case Report and Literature Review
title_sort familial dyskeratotic comedones: a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10328097/
https://www.ncbi.nlm.nih.gov/pubmed/37426083
http://dx.doi.org/10.2147/CCID.S420723
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