De Boeck K, Degreef H, Verwilghen R, Corbeel L, Casteels-Van Daele M
Pediatrics. 1981 Jun;67(6):898-903.
A case of dyskeratosis congenita is reported. This rare hereditary disease usually has the following progression: ectodermal dystrophy (reticular skin pigmentation, nail dystrophy, leukokeratosis of mucosal membranes), appearing in the first decade, followed in about 50% of these patients by a hematopoietic disorder resembling Fanconi's anemia, usually developing in the second or third decade. Carcinomas may occur in leukokeratotic areas in the third, fourth, or fifth decade. This patient's clinical course is interesting because the thrombocytopenia developed as an isolated symptom at the age of 5 years and preceded the skin anomalies by three years. The diagnosis of dyskeratosis congenita was made only after an evolution of five years. The diagnosis of dyskeratosis congenita--although it is a rare disease--should be considered in every child first seen with aplastic anemia or thrombocytopenia.
报告了1例先天性角化不良病例。这种罕见的遗传性疾病通常有以下病程:外胚层营养不良(网状皮肤色素沉着、指甲营养不良、黏膜白斑病)出现在第一个十年,约50%的这些患者随后会出现类似范可尼贫血的造血系统疾病,通常在第二个或第三个十年发展。在第三个、第四个或第五个十年,白斑病区域可能发生癌症。该患者的临床病程很有意思,因为血小板减少症在5岁时作为孤立症状出现,比皮肤异常早三年。先天性角化不良的诊断直到五年后才得以明确。先天性角化不良——尽管是一种罕见疾病——对于每一个首次因再生障碍性贫血或血小板减少症就诊的儿童都应予以考虑。