Literature DB >> 20591709

Novel 47.5-kb deletion in RAB27A results in severe Griscelli Syndrome Type 2.

Lisa M Vincent1, Fred Gilbert, Jennifer I DiPace, Carla Ciccone, Thomas C Markello, Andrew Jeong, Heidi Dorward, Wendy Westbroek, William A Gahl, James B Bussel, Marjan Huizing.   

Abstract

Griscelli syndrome (GS), a rare autosomal recessive disorder characterized by partial albinism and immunological impairment and/or severe neurological impairment, results from mutations in the MYO5A (GS1), RAB27A (GS2), or MLPH (GS3) genes. We identified a Hispanic patient born of a consanguineous union who presented with immunodeficiency, partial albinism, hepatic dysfunction, hemophagocytosis, neurological impairment, nystagmus, and silvery hair indicative of Griscelli Syndrome Type 2 (GS2). We screened for point mutations, but only exons 2-6 of the patient's DNA could be PCR-amplified. Whole genome analysis using the Illumina 1M-Duo DNA Analysis BeadChip identified a homozygous deletion in the patient's DNA. The exact breakpoints of the 47.5-kb deletion were identified as chr15q15-q21.1: g.53332432_53379990del (NCBI Build 37.1); the patient lacks the promoter and 5'UTR regions of RAB27A, thus confirming the diagnosis of GS2.

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Year:  2010        PMID: 20591709      PMCID: PMC2922439          DOI: 10.1016/j.ymgme.2010.05.015

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  34 in total

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Authors:  Miguel C Seabra; Emilie H Mules; Alistair N Hume
Journal:  Trends Mol Med       Date:  2002-01       Impact factor: 11.951

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Authors:  F Schuster; D K Stachel; I Schmid; F A Baumeister; U B Graubner; M Weiss; R J Haas; B H Belohradsky
Journal:  Bone Marrow Transplant       Date:  2001-08       Impact factor: 5.483

3.  Clinical presentation of Griscelli syndrome type 2 and spectrum of RAB27A mutations.

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4.  Griscelli disease: genotype-phenotype correlation in an array of clinical heterogeneity.

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Journal:  J Clin Immunol       Date:  2002-07       Impact factor: 8.317

5.  Rab27b is up-regulated in human Griscelli syndrome type II melanocytes and linked to the actin cytoskeleton via exon F-Myosin Va transcripts.

Authors:  Wendy Westbroek; Jo Lambert; Sofie De Schepper; Robert Kleta; Karolien Van Den Bossche; Miguel C Seabra; Marjan Huizing; Mieke Mommaas; Jean Marie Naeyaert
Journal:  Pigment Cell Res       Date:  2004-10

6.  Griscelli syndrome restricted to hypopigmentation results from a melanophilin defect (GS3) or a MYO5A F-exon deletion (GS1).

Authors:  Gaël Ménasché; Chen Hsuan Ho; Ozden Sanal; Jérôme Feldmann; Ilhan Tezcan; Fügen Ersoy; Anne Houdusse; Alain Fischer; Geneviève de Saint Basile
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7.  Multiple factors contribute to inefficient prenylation of Rab27a in Rab prenylation diseases.

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Journal:  J Biol Chem       Date:  2003-08-25       Impact factor: 5.157

8.  Evidence that Griscelli syndrome with neurological involvement is caused by mutations in RAB27A, not MYO5A.

Authors:  Yair Anikster; Marjan Huizing; Paul D Anderson; Diana L Fitzpatrick; Aharon Klar; Eva Gross-Kieselstein; Yackov Berkun; Gila Shazberg; William A Gahl; Haggit Hurvitz
Journal:  Am J Hum Genet       Date:  2002-06-07       Impact factor: 11.025

9.  Griscelli syndrome: characterization of a new mutation and rescue of T-cytotoxic activity by retroviral transfer of RAB27A gene.

Authors:  João C S Bizario; Jérôme Feldmann; Fabíola A Castro; Gaël Ménasché; Cristina M A Jacob; L Cristofani; Erasmo B Casella; Júlio C Voltarelli; Geneviève de Saint-Basile; Enilza M Espreafico
Journal:  J Clin Immunol       Date:  2004-07       Impact factor: 8.317

10.  Rab27a is required for regulated secretion in cytotoxic T lymphocytes.

Authors:  J C Stinchcombe; D C Barral; E H Mules; S Booth; A N Hume; L M Machesky; M C Seabra; G M Griffiths
Journal:  J Cell Biol       Date:  2001-02-19       Impact factor: 10.539

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Review 2.  Rab GTPases implicated in inherited and acquired disorders.

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4.  High resolution copy number variation data in the NCI-60 cancer cell lines from whole genome microarrays accessible through CellMiner.

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5.  A RAB27A 5' untranslated region structural variant associated with late-onset hemophagocytic lymphohistiocytosis and normal pigmentation.

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