Bari Omar, Skillman Sarah, Lah Melissa D, Haggstrom Anita N
School of Medicine, University of California, San Diego, La Jolla, CA, USA.
Department of Dermatology, MetroDerm P.C., Atlanta, GE, USA.
Pediatr Dermatol. 2018 Jul;35(4):e218-e220. doi: 10.1111/pde.13498. Epub 2018 Apr 6.
Desmoplakin mutations are associated with a wide variety of phenotypes affecting the skin, nails, hair, and heart. A 21-month-old boy was born with multiple erosions resembling epidermolysis bullosa, complete alopecia, nail dystrophy, palmoplantar keratoderma, and areas of follicular hyperkeratosis. He was found to have two heterozygous mutations in the desmoplakin gene: c.478 C>T in exon 4 (p.Arg160X) and c.3630T>A in exon 23 (Tyr1210X). This case expands the clinical spectrum associated with desmoplakin mutations and highlights a mutation in exon 23 that has not been previously reported in the literature.
桥粒斑蛋白突变与影响皮肤、指甲、毛发和心脏的多种表型相关。一名21个月大的男孩出生时就有多处类似大疱性表皮松解症的糜烂、全秃、甲营养不良、掌跖角化病以及毛囊角化过度区域。他被发现桥粒斑蛋白基因中有两个杂合突变:外显子4中的c.478 C>T(p.Arg160X)和外显子23中的c.3630T>A(Tyr1210X)。该病例扩展了与桥粒斑蛋白突变相关的临床谱,并突出了外显子23中的一种此前文献未报道的突变。