Suppr超能文献

罕见病中的多系统表现:先天性角化不良症的经验。

Multisystemic Manifestations in Rare Diseases: The Experience of Dyskeratosis Congenita.

机构信息

Pediatric Dentistry and Special Dental Care Unit, Meyer Children's University Hospital, 50139 Florence, Italy.

Unit of Metabolism, Bambino Gesù Children's Research Hospital, Piazza Sant'Onofrio, 4, 00165 Rome, Italy.

出版信息

Genes (Basel). 2022 Mar 11;13(3):496. doi: 10.3390/genes13030496.

Abstract

Dyskeratosis congenital (DC) is the first genetic syndrome described among telomeropathies. Its classical phenotype is characterized by the mucocutaneous triad of reticulated pigmentation of skin lace, nail dystrophy and oral leukoplakia. The clinical presentation, however, is heterogeneous and serious clinical complications include bone marrow failure, hematological and solid tumors. It may also involve immunodeficiencies, dental, pulmonary and liver disorders, and other minor complication. Dyskeratosis congenita shows marked genetic heterogeneity, as at least 14 genes are responsible for the shortening of telomeres characteristic of this disease. This review discusses clinical characteristics, molecular genetics, disease evolution, available therapeutic options and differential diagnosis of dyskeratosis congenita to provide an interdisciplinary and personalized medical assessment that includes family genetic counseling.

摘要

先天性角化不良(DC)是首个在端粒病中描述的遗传综合征。其典型表型的特征是皮肤花边状网状色素沉着、指甲营养不良和口腔白斑的黏膜皮肤三联征。然而,临床表现具有异质性,严重的临床并发症包括骨髓衰竭、血液系统和实体瘤。它也可能涉及免疫缺陷、牙齿、肺部和肝脏疾病以及其他较小的并发症。先天性角化不良表现出明显的遗传异质性,因为至少有 14 个基因负责缩短这种疾病特征的端粒。这篇综述讨论了先天性角化不良的临床特征、分子遗传学、疾病进展、可用的治疗选择和鉴别诊断,以提供包括家族遗传咨询在内的跨学科和个性化的医学评估。

相似文献

3
Recent progress in dyskeratosis congenita.先天性角化不良症的最新进展。
Int J Hematol. 2010 Oct;92(3):419-24. doi: 10.1007/s12185-010-0695-5. Epub 2010 Oct 1.
4
[Research progress of dyskeratosis congenita].先天性角化不良的研究进展
Zhonghua Kou Qiang Yi Xue Za Zhi. 2019 Feb 9;54(2):130-134. doi: 10.3760/cma.j.issn.1002-0098.2019.02.010.
5
Dyskeratosis congenita associated with leukoplakia of the tongue.先天性角化不良伴舌部白斑
Int J Oral Maxillofac Surg. 2016 Jun;45(6):760-3. doi: 10.1016/j.ijom.2015.12.005. Epub 2016 Jan 5.
7
Malignant transformation of oral leukoplakia in a patient with dyskeratosis congenita.先天性角化不良患者口腔白斑的恶性转化。
Oral Surg Oral Med Oral Pathol Oral Radiol. 2017 Oct;124(4):e239-e242. doi: 10.1016/j.oooo.2017.08.001. Epub 2017 Aug 12.
8
Dyskeratosis congenita.先天性角化不良
Hematol Oncol Clin North Am. 2009 Apr;23(2):215-31. doi: 10.1016/j.hoc.2009.01.003.

引用本文的文献

5
Telomere biology: from disorders to hematological diseases.端粒生物学:从疾病到血液学疾病
Front Oncol. 2023 May 19;13:1167848. doi: 10.3389/fonc.2023.1167848. eCollection 2023.

本文引用的文献

5
Revesz syndrome revisited.重新审视雷维茨综合征。
Orphanet J Rare Dis. 2020 Oct 23;15(1):299. doi: 10.1186/s13023-020-01553-y.
9
Dyskeratosis congenita: a literature review.先天性角化不良:文献综述
J Dtsch Dermatol Ges. 2020 Sep;18(9):943-967. doi: 10.1111/ddg.14268. Epub 2020 Sep 15.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验