Literature DB >> 25516398

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

Nina K Antonov1, Mina Y Kingsbery, Luis O Rohena, Teresa M Lee, Angela Christiano, Maria C Garzon, Christine T Lauren.   

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.
© 2014 Wiley Periodicals, Inc.

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Year:  2014        PMID: 25516398     DOI: 10.1111/pde.12484

Source DB:  PubMed          Journal:  Pediatr Dermatol        ISSN: 0736-8046            Impact factor:   1.588


  6 in total

1.  Distinct Cellular Basis for Early Cardiac Arrhythmias, the Cardinal Manifestation of Arrhythmogenic Cardiomyopathy, and the Skin Phenotype of Cardiocutaneous Syndromes.

Authors:  Jennifer Karmouch; Qiong Q Zhou; Christina Y Miyake; Raffaella Lombardi; Kai Kretzschmar; Marie Bannier-Hélaouët; Hans Clevers; Xander H T Wehrens; James T Willerson; Ali J Marian
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Review 2.  Genetics of inherited cardiocutaneous syndromes: a review.

Authors:  Tara Bardawil; Samar Khalil; Christina Bergqvist; Ossama Abbas; Abdul Ghani Kibbi; Fadi Bitar; Georges Nemer; Mazen Kurban
Journal:  Open Heart       Date:  2016-11-22

3.  SLINGER: large-scale learning for predicting gene expression.

Authors:  Kévin Vervier; Jacob J Michaelson
Journal:  Sci Rep       Date:  2016-12-20       Impact factor: 4.379

4.  Autosomal dominant Carvajal plus syndrome due to the novel desmoplakin mutation c.1678A > T (p.Ile560Phe).

Authors:  Josef Finsterer; Claudia Stöllberger; Eva Wollmann; Susanne Dertinger; Franco Laccone
Journal:  Mol Genet Metab Rep       Date:  2016-06-02

5.  Dominant de novo DSP mutations cause erythrokeratodermia-cardiomyopathy syndrome.

Authors:  Lynn M Boyden; Chen Y Kam; Angela Hernández-Martín; Jing Zhou; Brittany G Craiglow; Robert Sidbury; Erin F Mathes; Sheilagh M Maguiness; Debra A Crumrine; Mary L Williams; Ronghua Hu; Richard P Lifton; Peter M Elias; Kathleen J Green; Keith A Choate
Journal:  Hum Mol Genet       Date:  2015-11-24       Impact factor: 6.150

6.  Improvement of Storage Medium for Cultured Human Retinal Pigment Epithelial Cells Using Factorial Design.

Authors:  L Pasovic; T P Utheim; S Reppe; A Z Khan; C J Jackson; B Thiede; J P Berg; E B Messelt; J R Eidet
Journal:  Sci Rep       Date:  2018-04-09       Impact factor: 4.379

  6 in total

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