Literature DB >> 15915161

Loss of ZDHHC15 expression in a woman with a balanced translocation t(X;15)(q13.3;cen) and severe mental retardation.

Mahmoud Reza Mansouri1, Lena Marklund, Peter Gustavsson, Edward Davey, Birgit Carlsson, Catharina Larsson, Irene White, Karl-Henrik Gustavson, Niklas Dahl.   

Abstract

X-linked mental retardation (XLMR) affects one in 600 males and is highly heterogeneous. We describe here a 29-year-old woman with severe nonsyndromic mental retardation and a balanced reciprocal translocation between chromosomes X and 15 [46,XX,t(X;15)(q13.3;cen)]. Methylation studies showed a 100% skewed X-inactivation in patient-derived lymphocytes indicating that the normal chromosome X is retained inactive. Physical mapping of the breakpoints localised the Xq13.3 breakpoint to within 3.9 kb of the first exon of the ZDHHC15 gene encoding a zinc-finger and a DHHC domain containing product. Expression analysis revealed that different transcript variants of the gene are expressed in brain. ZDHHC15-specific RT-PCR analysis on lymphocytes from the patient revealed an absence of ZDHHC15 transcript variants, detected in control samples. We suggest that the absence of the ZDHHC15 transcripts in this patient contributes to her phenotype, and that the gene is a strong candidate for nonsyndromic XLMR.

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Year:  2005        PMID: 15915161     DOI: 10.1038/sj.ejhg.5201445

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  45 in total

1.  DHHC5 interacts with PDZ domain 3 of post-synaptic density-95 (PSD-95) protein and plays a role in learning and memory.

Authors:  Yi Li; Jie Hu; Klemens Höfer; Andrew M S Wong; Jonathan D Cooper; Shari G Birnbaum; Robert E Hammer; Sandra L Hofmann
Journal:  J Biol Chem       Date:  2010-02-22       Impact factor: 5.157

2.  Molecular genetic analysis of a de novo balanced translocation t(6;17)(p21.31;q11.2) associated with hypospadias and anorectal malformation.

Authors:  Mahmoud Reza Mansouri; Birgit Carlsson; Edward Davey; Agneta Nordenskjöld; Tomas Wester; Göran Annerén; Göran Läckgren; Niklas Dahl
Journal:  Hum Genet       Date:  2006-01-03       Impact factor: 4.132

Review 3.  Discovery of protein-palmitoylating enzymes.

Authors:  Ryouhei Tsutsumi; Yuko Fukata; Masaki Fukata
Journal:  Pflugers Arch       Date:  2008-01-30       Impact factor: 3.657

Review 4.  Balanced translocations in mental retardation.

Authors:  Geert Vandeweyer; R Frank Kooy
Journal:  Hum Genet       Date:  2009-04-05       Impact factor: 4.132

5.  XLID-causing mutations and associated genes challenged in light of data from large-scale human exome sequencing.

Authors:  Amélie Piton; Claire Redin; Jean-Louis Mandel
Journal:  Am J Hum Genet       Date:  2013-07-18       Impact factor: 11.025

6.  Breakpoint mapping and array CGH in translocations: comparison of a phenotypically normal and an abnormal cohort.

Authors:  Julia Baptista; Catherine Mercer; Elena Prigmore; Susan M Gribble; Nigel P Carter; Viv Maloney; N Simon Thomas; Patricia A Jacobs; John A Crolla
Journal:  Am J Hum Genet       Date:  2008-03-27       Impact factor: 11.025

7.  Tracking brain palmitoylation change: predominance of glial change in a mouse model of Huntington's disease.

Authors:  Junmei Wan; Jeffrey N Savas; Amy F Roth; Shaun S Sanders; Roshni R Singaraja; Michael R Hayden; John R Yates; Nicholas G Davis
Journal:  Chem Biol       Date:  2013-11-07

8.  Palmitoylation of cytoskeleton associated protein 4 by DHHC2 regulates antiproliferative factor-mediated signaling.

Authors:  Sonia L Planey; Susan K Keay; Chen-Ou Zhang; David A Zacharias
Journal:  Mol Biol Cell       Date:  2009-01-14       Impact factor: 4.138

9.  The palmitoyl acyltransferases ZDHHC5 and ZDHHC8 are uniquely present in DRG axons and control retrograde signaling via the Gp130/JAK/STAT3 pathway.

Authors:  Kaitlin M Collura; Jingwen Niu; Shaun S Sanders; Audrey Montersino; Sabrina M Holland; Gareth M Thomas
Journal:  J Biol Chem       Date:  2020-09-21       Impact factor: 5.157

10.  Palmitoylation regulates epidermal homeostasis and hair follicle differentiation.

Authors:  Pleasantine Mill; Angela W S Lee; Yuko Fukata; Ryouhei Tsutsumi; Masaki Fukata; Margaret Keighren; Rebecca M Porter; Lisa McKie; Ian Smyth; Ian J Jackson
Journal:  PLoS Genet       Date:  2009-11-26       Impact factor: 5.917

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