Familial Dyskeratotic Comedones: A Case Report and Literature Review
Kasama Tejapira, Poonkiat Suchonwanit Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, ThailandCorrespondence: Poonkiat Suchonwanit, Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mah...
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| Format: | Article |
| Language: | English |
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Dove Medical Press
2023-07-01
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| Series: | Clinical, Cosmetic and Investigational Dermatology |
| Subjects: | |
| Online Access: | https://www.dovepress.com/familial-dyskeratotic-comedones-a-case-report-and-literature-review-peer-reviewed-fulltext-article-CCID |
| _version_ | 1797787459697770496 |
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| author | Tejapira K Suchonwanit P |
| author_facet | Tejapira K Suchonwanit P |
| author_sort | Tejapira K |
| collection | DOAJ |
| description | Kasama Tejapira, Poonkiat Suchonwanit Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, ThailandCorrespondence: Poonkiat Suchonwanit, Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, 270 Rama VI Road, Ratchathewi, Bangkok, 10400, Thailand, Tel +66-2-2011141, Fax +66-2-201-1211 ext 4, Email poonkiat@hotmail.comAbstract: 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.Keywords: acne, autosomal dominant, comedone-like lesions, dermoscopy, dyskeratosis, hyperkeratosis |
| first_indexed | 2024-03-13T01:22:09Z |
| format | Article |
| id | doaj.art-47534eba886b4cb59be70111d841c48f |
| institution | Directory Open Access Journal |
| issn | 1178-7015 |
| language | English |
| last_indexed | 2024-03-13T01:22:09Z |
| publishDate | 2023-07-01 |
| publisher | Dove Medical Press |
| record_format | Article |
| series | Clinical, Cosmetic and Investigational Dermatology |
| spelling | doaj.art-47534eba886b4cb59be70111d841c48f2023-07-04T19:06:10ZengDove Medical PressClinical, Cosmetic and Investigational Dermatology1178-70152023-07-01Volume 161729173584847Familial Dyskeratotic Comedones: A Case Report and Literature ReviewTejapira KSuchonwanit PKasama Tejapira, Poonkiat Suchonwanit Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, ThailandCorrespondence: Poonkiat Suchonwanit, Division of Dermatology, Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, 270 Rama VI Road, Ratchathewi, Bangkok, 10400, Thailand, Tel +66-2-2011141, Fax +66-2-201-1211 ext 4, Email poonkiat@hotmail.comAbstract: 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.Keywords: acne, autosomal dominant, comedone-like lesions, dermoscopy, dyskeratosis, hyperkeratosishttps://www.dovepress.com/familial-dyskeratotic-comedones-a-case-report-and-literature-review-peer-reviewed-fulltext-article-CCIDacneautosomal dominantcomedone-like lesionsdermoscopydyskeratosishyperkeratosis. |
| spellingShingle | Tejapira K Suchonwanit P Familial Dyskeratotic Comedones: A Case Report and Literature Review Clinical, Cosmetic and Investigational Dermatology acne autosomal dominant comedone-like lesions dermoscopy dyskeratosis hyperkeratosis. |
| 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 | acne autosomal dominant comedone-like lesions dermoscopy dyskeratosis hyperkeratosis. |
| url | https://www.dovepress.com/familial-dyskeratotic-comedones-a-case-report-and-literature-review-peer-reviewed-fulltext-article-CCID |
| work_keys_str_mv | AT tejapirak familialdyskeratoticcomedonesacasereportandliteraturereview AT suchonwanitp familialdyskeratoticcomedonesacasereportandliteraturereview |