Literature DB >> 29345414

Molecular characterization of a series of 990 index patients with albinism.

Eulalie Lasseaux1, Claudio Plaisant1, Vincent Michaud1, Perrine Pennamen1,2, Aurelien Trimouille1,2, Laetitia Gaston1, Solène Monfermé3, Didier Lacombe1,2, Caroline Rooryck1,2, Fanny Morice-Picard4, Benoît Arveiler1,2.   

Abstract

Albinism is a clinically and genetically heterogeneous disease characterized by variable degrees of hypopigmentation and by nystagmus, foveal hypoplasia, and chiasmatic misrouting of the optic nerves. The wide phenotypic heterogeneity impedes the establishment of phenotype-genotype correlations. To obtain a precise diagnosis, we screened the 19 known albinism genes in 990 index patients using targeted next-generation sequencing (NGS) and high-resolution comparative genomic hybridization. A molecular diagnosis was obtained in 72.32% of patients. A total of 243 new pathogenic variants were identified. Intragenic rearrangements represented 10.8% of all pathogenic alleles. NGS panel analysis allowed establishing a diagnosis for the rarest forms of the disease, which could not be diagnosed otherwise. Because of the clinical overlap between the different forms of the disease, diagnosis nowadays clearly relies on molecular grounds.
© 2018 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  albinism; deletions; mutations; next-generation sequencing

Mesh:

Year:  2018        PMID: 29345414     DOI: 10.1111/pcmr.12688

Source DB:  PubMed          Journal:  Pigment Cell Melanoma Res        ISSN: 1755-1471            Impact factor:   4.693


  31 in total

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Authors:  Shanna L Bowman; Jing Bi-Karchin; Linh Le; Michael S Marks
Journal:  Traffic       Date:  2019-06       Impact factor: 6.215

2.  The contribution of common regulatory and protein-coding TYR variants to the genetic architecture of albinism.

Authors:  Vincent Michaud; Eulalie Lasseaux; David J Green; Dave T Gerrard; Claudio Plaisant; Tomas Fitzgerald; Ewan Birney; Benoît Arveiler; Graeme C Black; Panagiotis I Sergouniotis
Journal:  Nat Commun       Date:  2022-07-08       Impact factor: 17.694

3.  A community-science approach identifies genetic variants associated with three color morphs in ball pythons (Python regius).

Authors:  Autumn R Brown; Kaylee Comai; Dominic Mannino; Haily McCullough; Yamini Donekal; Hunter C Meyers; Chiron W Graves; Hannah S Seidel
Journal:  PLoS One       Date:  2022-10-19       Impact factor: 3.752

4.  Molecular characterization of SLC24A5 variants and evaluation of Nitisinone treatment efficacy in a zebrafish model of OCA6.

Authors:  Sairah Yousaf; Saumil Sethna; Muhammad A Chaudhary; Rehan S Shaikh; Saima Riazuddin; Zubair M Ahmed
Journal:  Pigment Cell Melanoma Res       Date:  2020-04-27       Impact factor: 4.693

5.  A custom capture sequence approach for oculocutaneous albinism identifies structural variant alleles at the OCA2 locus.

Authors:  Stacie K Loftus; Linnea Lundh; Dawn E Watkins-Chow; Laura L Baxter; Erola Pairo-Castineira; Ian J Jackson; William S Oetting; William J Pavan; David R Adams
Journal:  Hum Mutat       Date:  2021-08-01       Impact factor: 4.700

6.  Clinical utility gene card for oculocutaneous (OCA) and ocular albinism (OA)-an update.

Authors:  Abdullah Aamir; Helen J Kuht; Karen Grønskov; Brian P Brooks; Mervyn G Thomas
Journal:  Eur J Hum Genet       Date:  2021-01-27       Impact factor: 5.351

7.  A pathogenic haplotype, common in Europeans, causes autosomal recessive albinism and uncovers missing heritability in OCA1.

Authors:  Karen Grønskov; Cathrine Jespersgaard; Gitte Hoffmann Bruun; Pernille Harris; Karen Brøndum-Nielsen; Brage S Andresen; Thomas Rosenberg
Journal:  Sci Rep       Date:  2019-01-24       Impact factor: 4.379

8.  Evident hypopigmentation without other ocular deficits in Dutch patients with oculocutaneous albinism type 4.

Authors:  C C Kruijt; N E Schalij-Delfos; G C de Wit; R J Florijn; M M van Genderen
Journal:  Sci Rep       Date:  2021-06-02       Impact factor: 4.379

9.  SLC45A2 protein stability and regulation of melanosome pH determine melanocyte pigmentation.

Authors:  Linh Le; Iliana E Escobar; Tina Ho; Ariel J Lefkovith; Emily Latteri; Kirk D Haltaufderhyde; Megan K Dennis; Lynn Plowright; Elena V Sviderskaya; Dorothy C Bennett; Elena Oancea; Michael S Marks
Journal:  Mol Biol Cell       Date:  2020-09-23       Impact factor: 4.138

10.  Hermansky-Pudlak syndrome: Mutation update.

Authors:  Marjan Huizing; May C V Malicdan; Jennifer A Wang; Hadass Pri-Chen; Richard A Hess; Roxanne Fischer; Kevin J O'Brien; Melissa A Merideth; William A Gahl; Bernadette R Gochuico
Journal:  Hum Mutat       Date:  2020-01-23       Impact factor: 4.700

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