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早发性心力衰竭、脱发及皮肤异常与桥粒斑蛋白的一种新型复合杂合突变相关。

Early-onset heart failure, alopecia, and cutaneous abnormalities associated with a novel compound heterozygous mutation in desmoplakin.

作者信息

Antonov Nina K, Kingsbery Mina Y, Rohena Luis O, Lee Teresa M, Christiano Angela, Garzon Maria C, Lauren Christine T

机构信息

College of Physicians and Surgeons, Columbia University, New York, New York.

出版信息

Pediatr Dermatol. 2015 Jan-Feb;32(1):102-8. doi: 10.1111/pde.12484. Epub 2014 Dec 16.

Abstract

Mutations in the desmosomal protein desmoplakin have been associated with various conditions affecting the skin and heart. The prototype is Carvajal syndrome, characterized by cardiomyopathy, woolly hair, palmoplantar keratoderma (PPK), and skin fragility. We report the case of a 3-year-old boy presenting with severe left-sided heart failure with a preceding history of cutaneous abnormalities including congenital alopecia, PPK, nail dystrophy, and follicular hyperkeratosis on the extensor surfaces. Genetic testing revealed a novel combination of two heterozygous mutations in the DSP gene encoding desmoplakin: R1400X and R2284X. Both are predicted to be deleterious to protein function. This case adds to our understanding of the spectrum of clinical presentations of syndromes associated with desmoplakin mutations and highlights the need for cardiac examination in patients with characteristic cutaneous findings.

摘要

桥粒蛋白桥粒斑蛋白的突变与影响皮肤和心脏的多种病症有关。典型病例是卡瓦哈尔综合征,其特征为心肌病、羊毛状头发、掌跖角化病(PPK)和皮肤脆弱。我们报告了一名3岁男孩的病例,该男孩患有严重的左侧心力衰竭,之前有皮肤异常病史,包括先天性脱发、PPK、指甲营养不良以及伸侧表面的毛囊角化过度。基因检测发现编码桥粒斑蛋白的DSP基因存在两个杂合突变的新组合:R1400X和R2284X。预计这两个突变均对蛋白质功能有害。该病例增进了我们对与桥粒斑蛋白突变相关综合征临床表现谱的理解,并强调了对具有特征性皮肤表现的患者进行心脏检查的必要性。

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