Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Hussein, Firas, Omar, Zainab
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2021
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
_version_ 1784607972212604928
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