Posterior Polymorphous Corneal Dystrophy in a Patient with a Novel ZEB1 Gene Mutation

被引:2
作者
Fernandez-Gutierrez, Eva [1 ]
Fernandez-Perez, Pedro [1 ]
Boto-De-los-Bueis, Ana [1 ]
Garcia-Fernandez, Laura [2 ,3 ]
Rodriguez-Solana, Patricia [4 ]
Solis, Mario [3 ,5 ]
Vallespin, Elena [3 ,4 ]
机构
[1] La Paz Univ Hosp, Dept Ophthalmol, Madrid 28046, Spain
[2] La Paz Univ Hosp, Med & Mol Genet Inst INGEMM IdiPaz, Mol Genet Sect, Madrid 28046, Spain
[3] Carlos III Hlth Inst ISCIII, Biomed Res Ctr Rare Dis Network CIBERER, Madrid 28029, Spain
[4] La Paz Univ Hosp, Med & Mol Genet Inst INGEMM IdiPaz, Mol Ophthalmol Sect, Madrid 28046, Spain
[5] La Paz Univ Hosp, Med & Mol Genet Inst INGEMM IdiPaz, Clin Bioinformat Sect, Madrid 28046, Spain
关键词
posterior polymorphous corneal dystrophy; keratoconus; iridocorneal endothelial syndrome; ZEB1; confocal microscopy;
D O I
10.3390/ijms24010209
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
Posterior polymorphous corneal dystrophy (PPCD), a rare, bilateral, autosomal-dominant, inherited corneal dystrophy, affects the Descemet membrane and corneal endothelium. We describe an unusual presentation of PPCD associated with a previously unknown genetic alteration in the ZEB1 gene. The proband is a 64-year-old woman diagnosed with keratoconus referred for a corneal endothelium study who presented endothelial lesions in both eyes suggestive of PPCD, corectopia and iridocorneal endothelial synechiae in the right eye and intrastromal segments in the left eye. The endothelial count was 825 in the right eye and 1361 in the left eye, with typical PPCD lesions visible under specular and confocal microscopy. In the next generation sequencing genetic analysis, a heterozygous c.1A > C (p.Met1Leu) mutation was found in the ZEB1 gene (TCF8). The PPCD3 subtype is associated with corneal ectasia, and both can appear due to a pathogenic mutation in the ZEB1 gene (OMIM #189909). However, our patient had a previously unreported mutation in the ZEB1 gene, which mediates the transition between cell lines and provides a pathogenic explanation for the epithelialisation of the corneal endothelium, a characteristic of PPCD.
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页数:11
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