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Novel Intragenic FRMD7 Deletion in a Pedigree with Congenital X-Linked Nystagmus
Journal article   Peer reviewed

Novel Intragenic FRMD7 Deletion in a Pedigree with Congenital X-Linked Nystagmus

John H Fingert, Ben Roos, Mari E Eyestone, Joshua D Pham, Mei L Mellot and Edwin Stone
Ophthalmic Genetics, Vol.31(2), pp.77-80
06/01/2010
DOI: 10.3109/13816810903584989
PMID: 20450309

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Abstract

Objective: To identify the disease-causing mutation in a large 3 generation pedigree of X-linked congenital nystagmus. Methods: Twenty-three members of a single pedigree, including 7 affected males, 2 affected females, 5 obligate carriers, and 9 unaffected family members were tested for mutations in the FRMD7 gene using PCR-based DNA sequencing assays and multiplex PCR assays for deletions. Results: A hemizygous deletion of exons 2, 3, and 4 of FRMD7 was detected in all affected males in the family and was absent from 40 control subjects. Conclusions: A range of missense, nonsense, frameshift, and splicing mutations in FRMD7 have been shown to cause X-linked congenital nystagmus. Here we show for the first time that large intragenic deletions of FRMD7 can also cause this form of nystagmus.
Genetics Nystagmus Mutation

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