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Dyskeratosis congenita: rare case report of Syria
Dyskeratosis congenita (DC) is an inherited disease characterized by the triad of abnormal skin pigmentation, nail dystrophy and mucosal leukoplakia. Non-cutaneous abnormalities (dental, gastrointestinal, genitourinary, neurological, ophthalmic, pulmonary and skeletal) have also been reported. Bone...
Autores principales: | , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8633647/ https://www.ncbi.nlm.nih.gov/pubmed/34858620 http://dx.doi.org/10.1093/omcr/omab041 |
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author | Hussein, Firas Omar, Zainab |
author_facet | Hussein, Firas Omar, Zainab |
author_sort | Hussein, Firas |
collection | PubMed |
description | Dyskeratosis congenita (DC) is an inherited disease characterized by the triad of abnormal skin pigmentation, nail dystrophy and mucosal leukoplakia. Non-cutaneous abnormalities (dental, gastrointestinal, genitourinary, neurological, ophthalmic, pulmonary and skeletal) have also been reported. Bone marrow failure (BMF) is the main cause of early mortality, with an additional predisposition to malignancy. DC results from an anomalous progressive shortening of telomeres resulting in DNA replication problems inducing replicative senescence. Men are more affected than women are and X-linked recessive, autosomal dominant and autosomal recessive forms of the disease are recognized. There are no targeted therapies for DC. Patients treated with androgens had a hematological response. We herein describe case of a 32-year-old man, presented with several characteristic systemic features of this condition, including the classic triad of lesions, dysplastic bone marrow, epiphora and liver cirrhosis with grade I esophageal varices. Therefore, a prophylactic propranolol was started in additional to danazol. Three-week later, the patient had subsequent increases in his platelet, red cell and white cell counts. |
format | Online Article Text |
id | pubmed-8633647 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-86336472021-12-01 Dyskeratosis congenita: rare case report of Syria Hussein, Firas Omar, Zainab Oxf Med Case Reports Case Report Dyskeratosis congenita (DC) is an inherited disease characterized by the triad of abnormal skin pigmentation, nail dystrophy and mucosal leukoplakia. Non-cutaneous abnormalities (dental, gastrointestinal, genitourinary, neurological, ophthalmic, pulmonary and skeletal) have also been reported. Bone marrow failure (BMF) is the main cause of early mortality, with an additional predisposition to malignancy. DC results from an anomalous progressive shortening of telomeres resulting in DNA replication problems inducing replicative senescence. Men are more affected than women are and X-linked recessive, autosomal dominant and autosomal recessive forms of the disease are recognized. There are no targeted therapies for DC. Patients treated with androgens had a hematological response. We herein describe case of a 32-year-old man, presented with several characteristic systemic features of this condition, including the classic triad of lesions, dysplastic bone marrow, epiphora and liver cirrhosis with grade I esophageal varices. Therefore, a prophylactic propranolol was started in additional to danazol. Three-week later, the patient had subsequent increases in his platelet, red cell and white cell counts. Oxford University Press 2021-11-25 /pmc/articles/PMC8633647/ /pubmed/34858620 http://dx.doi.org/10.1093/omcr/omab041 Text en © The Author(s) 2021. Published by Oxford University Press. https://creativecommons.org/licenses/by/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Hussein, Firas Omar, Zainab Dyskeratosis congenita: rare case report of Syria |
title | Dyskeratosis congenita: rare case report of Syria |
title_full | Dyskeratosis congenita: rare case report of Syria |
title_fullStr | Dyskeratosis congenita: rare case report of Syria |
title_full_unstemmed | Dyskeratosis congenita: rare case report of Syria |
title_short | Dyskeratosis congenita: rare case report of Syria |
title_sort | dyskeratosis congenita: rare case report of syria |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8633647/ https://www.ncbi.nlm.nih.gov/pubmed/34858620 http://dx.doi.org/10.1093/omcr/omab041 |
work_keys_str_mv | AT husseinfiras dyskeratosiscongenitararecasereportofsyria AT omarzainab dyskeratosiscongenitararecasereportofsyria |