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Dyskeratosis congenita: rare case report of Syria
- Source :
- Oxford Medical Case Reports
- Publication Year :
- 2021
- Publisher :
- Oxford University Press (OUP), 2021.
-
Abstract
- 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.
- Subjects :
- Danazol
Pathology
medicine.medical_specialty
Cirrhosis
business.industry
Bone marrow failure
Case Report
High-digestive endoscopy
Esophageal varices
Malignancy
medicine.disease
Microbiology
omcrep/1000
omcrep/1500
Infectious Diseases
medicine.anatomical_structure
medicine
Parasitology
Bone marrow
AcademicSubjects/MED00010
business
Dyskeratosis congenita
Leukoplakia
medicine.drug
Subjects
Details
- ISSN :
- 20538855
- Volume :
- 2021
- Database :
- OpenAIRE
- Journal :
- Oxford Medical Case Reports
- Accession number :
- edsair.doi.dedup.....9ad42b24638db9a6623b1c4b1fc7e752
- Full Text :
- https://doi.org/10.1093/omcr/omab041