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publication name New missense variants in RELT causing hypomineralised amelogenesis imperfecta
Authors Georgios Nikolopoulos, Claire EL Smith, Steven J Brookes, Mohammed E El‐Asrag, Catriona J Brown, Anesha Patel, Gina Murillo, Mary J O'Connell, Chris F Inglehearn, Alan J Mighell
year 2020
keywords
journal Clinical genetics
volume 97
issue 5
pages 688-695
publisher Blackwell Publishing Ltd
Local/International International
Paper Link https://onlinelibrary.wiley.com/doi/full/10.1111/cge.13721
Full paper download
Supplementary materials Not Available
Abstract

Amelogenesis imperfecta (AI) is a heterogeneous group of genetic diseases characterised by dental enamel malformation. Pathogenic variants in at least 33 genes cause syndromic or non-syndromic AI. Recently variants in RELT, encoding an orphan receptor in the tumour necrosis factor (TNF) superfamily, were found to cause recessive AI, as part of a syndrome encompassing small stature and severe childhood infections. Here we describe four additional families with autosomal recessive hypomineralised AI due to previously unreported homozygous mutations in RELT. Three families carried a homozygous missense variant in the fourth exon (c.164C>T, p.(T55I)) and a fourth family carried a homozygous missense variant in the 11th exon (c.1264C>T, p.(R422W)). We found no evidence of additional syndromic symptoms in affected individuals. Analyses of tooth microstructure with computerised tomography and scanning electron microscopy suggest a role for RELT in ameloblasts' coordination and interaction with the enamel matrix. Microsatellite genotyping in families segregating the T55I variant reveals a shared founder haplotype. These findings extend the RELT pathogenic variant spectrum, reveal a founder mutation in the UK Pakistani population and provide detailed analysis of human teeth affected by this hypomineralised phenotype, but do not support a possible syndromic presentation in all those with RELT-variant associated AI.

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