Literature DB >> 32700429

Knobloch syndrome in a patient from Chile.

Nicole Nakousi-Capurro1, Jonathan Huserman1, Silvia Castillo1,2, Luisa Herrera3, Pablo Romero4, Felipe Pizarro5, Cristian Quezada5, Francisco Cea6.   

Abstract

Knobloch Syndrome (KS) is a rare autosomal recessive hereditary disease. Despite its clinical heterogeneity, it is characterized by vitreoretinal degeneration and high myopia, with or without occipital skull defects. It is caused by mutations in the COL18A1 gene, which codifies for collagen XVIII, present in retina and vascular endothelium. Since the first description of the disease by doctors Knobloch and Layer in 1972, over 100 cases and 20 pathogenic or likely pathogenic mutations have been reported. We present the case of a child born from a consanguineous couple in Chile with congenital high myopia and dysmorphisms without an occipital skull defect. Whole exome sequencing analysis revealed an inherited homozygous variant in COL18A1, c.4224_4225delinsC, p.Pro1411Leufs*35.
© 2020 Wiley Periodicals LLC.

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Keywords:  COL18A1; Knobloch syndrome; high-grade myopia

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Year:  2020        PMID: 32700429     DOI: 10.1002/ajmg.a.61760

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  1 in total

1.  Knobloch Syndrome Associated with Novel COL18A1 Variants in Chinese Population.

Authors:  Songshan Li; You Wang; Limei Sun; Wenjia Yan; Li Huang; Zhaotian Zhang; Ting Zhang; Xiaoyan Ding
Journal:  Genes (Basel)       Date:  2021-09-26       Impact factor: 4.096

  1 in total

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