Literature DB >> 30986449

A family with a mild form of congenital nystagmus and optic disc coloboma caused by a novel PAX6 mutation.

Byeonghyeon Lee1, Deok-Gyun Choi2, Bo Young Chun3, Eun Hye Oh4, Yun-Jeong Lee5, Un-Kyung Kim1, Jin-Sung Park6.   

Abstract

Congenital nystagmus (CN) is a heterogeneous disease that shows variable clinical features. There are a few mutations that are known to cause CN. Among them, a PAX6 mutation is known to cause CN with an extremely high frequency of aniridia. Here, we report on a family with an autosomal dominant PAX6 mutation, c.214G > A (p.Gly72Ser.), who presented with CN in the absence of aniridia. This study describes detailed clinical findings, including videonystagmography and fundus photography findings and emphasizes the importance of screening for the PAX6 gene in patients who present with CN in the absence of aniridia, as this will further elucidate the known phenotypes of PAX6-related diseases.
Copyright © 2019 Elsevier B.V. All rights reserved.

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Year:  2019        PMID: 30986449     DOI: 10.1016/j.gene.2019.04.035

Source DB:  PubMed          Journal:  Gene        ISSN: 0378-1119            Impact factor:   3.688


  2 in total

1.  An autosomal dominant ERLIN2 mutation leads to a pure HSP phenotype distinct from the autosomal recessive ERLIN2 mutations (SPG18).

Authors:  Jin-Mo Park; Byeonghyeon Lee; Jong-Heun Kim; Seong-Yong Park; Jinhoon Yu; Un-Kyung Kim; Jin-Sung Park
Journal:  Sci Rep       Date:  2020-02-24       Impact factor: 4.379

Review 2.  The Spectrum of PAX6 Mutations and Genotype-Phenotype Correlations in the Eye.

Authors:  Dulce Lima Cunha; Gavin Arno; Marta Corton; Mariya Moosajee
Journal:  Genes (Basel)       Date:  2019-12-17       Impact factor: 4.096

  2 in total

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