Cargando…

Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report

Linear porokeratosis is a rare variant of porokeratosis that most often presents in newborns and children; development of this porokeratosis variant in adulthood is far less common. We report the case of a 25-year-old female who presented with a progressive eruption on the proximal upper extremity o...

Descripción completa

Detalles Bibliográficos
Autores principales: Nazarian, Roya S, Hosseinipour, Mojgan, Amin, Bijal, Cohen, Steven R
Formato: Online Artículo Texto
Lenguaje:English
Publicado: SAGE Publications 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7234341/
https://www.ncbi.nlm.nih.gov/pubmed/32477558
http://dx.doi.org/10.1177/2050313X20919613
_version_ 1783535739691073536
author Nazarian, Roya S
Hosseinipour, Mojgan
Amin, Bijal
Cohen, Steven R
author_facet Nazarian, Roya S
Hosseinipour, Mojgan
Amin, Bijal
Cohen, Steven R
author_sort Nazarian, Roya S
collection PubMed
description Linear porokeratosis is a rare variant of porokeratosis that most often presents in newborns and children; development of this porokeratosis variant in adulthood is far less common. We report the case of a 25-year-old female who presented with a progressive eruption on the proximal upper extremity of 6-year duration, which was ultimately diagnosed as adult-onset linear porokeratosis and safely treated with oral isotretinoin. We propose that a sporadic mutation resulting in mosaicism after birth may explain the development of linear porokeratosis in adulthood, although the exact trigger of such a somatic mutation is not known. This case also describes a unique clinical presentation, with linear porokeratosis lesions originating on the proximal extremity rather than on the more common distal extremity. This demonstrates a distinctive clinical presentation not seen in the pediatric forms of disease.
format Online
Article
Text
id pubmed-7234341
institution National Center for Biotechnology Information
language English
publishDate 2020
publisher SAGE Publications
record_format MEDLINE/PubMed
spelling pubmed-72343412020-05-29 Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report Nazarian, Roya S Hosseinipour, Mojgan Amin, Bijal Cohen, Steven R SAGE Open Med Case Rep JCMS Case Report Linear porokeratosis is a rare variant of porokeratosis that most often presents in newborns and children; development of this porokeratosis variant in adulthood is far less common. We report the case of a 25-year-old female who presented with a progressive eruption on the proximal upper extremity of 6-year duration, which was ultimately diagnosed as adult-onset linear porokeratosis and safely treated with oral isotretinoin. We propose that a sporadic mutation resulting in mosaicism after birth may explain the development of linear porokeratosis in adulthood, although the exact trigger of such a somatic mutation is not known. This case also describes a unique clinical presentation, with linear porokeratosis lesions originating on the proximal extremity rather than on the more common distal extremity. This demonstrates a distinctive clinical presentation not seen in the pediatric forms of disease. SAGE Publications 2020-05-06 /pmc/articles/PMC7234341/ /pubmed/32477558 http://dx.doi.org/10.1177/2050313X20919613 Text en © The Author(s) 2020 https://creativecommons.org/licenses/by-nc/4.0/ This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (https://creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage).
spellingShingle JCMS Case Report
Nazarian, Roya S
Hosseinipour, Mojgan
Amin, Bijal
Cohen, Steven R
Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title_full Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title_fullStr Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title_full_unstemmed Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title_short Linear porokeratosis presenting in adulthood: A diagnostic challenge: A case report
title_sort linear porokeratosis presenting in adulthood: a diagnostic challenge: a case report
topic JCMS Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7234341/
https://www.ncbi.nlm.nih.gov/pubmed/32477558
http://dx.doi.org/10.1177/2050313X20919613
work_keys_str_mv AT nazarianroyas linearporokeratosispresentinginadulthoodadiagnosticchallengeacasereport
AT hosseinipourmojgan linearporokeratosispresentinginadulthoodadiagnosticchallengeacasereport
AT aminbijal linearporokeratosispresentinginadulthoodadiagnosticchallengeacasereport
AT cohenstevenr linearporokeratosispresentinginadulthoodadiagnosticchallengeacasereport