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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...
Autores principales: | , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Dove
2023
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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. |
format | Online Article Text |
id | pubmed-10328097 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Dove |
record_format | MEDLINE/PubMed |
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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