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中国大陆先天性角化不良的临床特征:病例报告及文献综述

Clinical features of dyskeratosis congenita in mainland China: case reports and literature review.

作者信息

Li Fuxing, Li Wei, Qiao Xiaohong, Xie Xiaotian

机构信息

Department of Pediatrics, Tongji Hospital, Tongji University School of Medicine, Xincun Road 389, Shanghai, 200065, People's Republic of China.

出版信息

Int J Hematol. 2019 Mar;109(3):328-335. doi: 10.1007/s12185-018-02582-x. Epub 2019 Jan 3.

Abstract

Dyskeratosis congenita (DC) is a rare-inherited bone marrow failure syndrome associated with multi-system disorder. To summarize the clinical features, epidemiology, and treatment of DC in mainland China, we retrospectively reviewed the medical records of two patients diagnosed with DC at our hospital and published reports on other DC patients in mainland China. The clinical features of 82 DC patients were summarized. The median age of onset was 5 years, but the median age at diagnosis was 16 years. Bone marrow failure occurred at a high rate of 44% and early, with a median onset age of 6 years (range 1-40 years). Only DKC1, TINF2, and TERT mutations were reported, which is a relatively simple signature. Aplastic anemia was treated mainly with low-dose androgens, glucocorticoids, or allogeneic hematopoietic stem cell transplantation, with an efficacy of 39% (14/36). In China, DC is relatively common in infants, with early age of onset but delayed diagnosis. Bone marrow failure occurred at a high rate and early. Improvement in the knowledge and awareness of DC combined with gene mutation tests will facilitate diagnosis and therapy in its early stages.

摘要

先天性角化不良(DC)是一种罕见的遗传性骨髓衰竭综合征,与多系统疾病相关。为总结中国大陆DC的临床特征、流行病学及治疗情况,我们回顾性分析了我院确诊的2例DC患者的病历以及中国大陆其他DC患者的已发表报告。总结了82例DC患者的临床特征。发病中位年龄为5岁,但诊断中位年龄为16岁。骨髓衰竭发生率高,达44%,且发病早,发病中位年龄为6岁(范围1 - 40岁)。仅报告了DKC1、TINF2和TERT突变,这是一个相对简单的特征。再生障碍性贫血主要采用小剂量雄激素、糖皮质激素或异基因造血干细胞移植治疗,有效率为39%(14/36)。在中国,DC在婴幼儿中相对常见,发病早但诊断延迟。骨髓衰竭发生率高且发病早。提高对DC的认识和了解并结合基因突变检测将有助于早期诊断和治疗。

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