Literature DB >> 8689688

LIM-kinase1 hemizygosity implicated in impaired visuospatial constructive cognition.

J M Frangiskakis1, A K Ewart, C A Morris, C B Mervis, J Bertrand, B F Robinson, B P Klein, G J Ensing, L A Everett, E D Green, C Pröschel, N J Gutowski, M Noble, D L Atkinson, S J Odelberg, M T Keating.   

Abstract

To identify genes important for human cognitive development, we studied Williams syndrome (WS), a developmental disorder that includes poor visuospatial constructive cognition. Here we describe two families with a partial WS phenotype; affected members have the specific WS cognitive profile and vascular disease, but lack other WS features. Submicroscopic chromosome 7q11.23 deletions cosegregate with this phenotype in both families. DNA sequence analyses of the region affected by the smallest deletion (83.6 kb) revealed two genes, elastin (ELN) and LIM-kinase1 (LIMK1). The latter encodes a novel protein kinase with LIM domains and is strongly expressed in the brain. Because ELN mutations cause vascular disease but not cognitive abnormalities, these data implicate LIMK1 hemizygosity in imparied visuospatial constructive cognition.

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Year:  1996        PMID: 8689688     DOI: 10.1016/s0092-8674(00)80077-x

Source DB:  PubMed          Journal:  Cell        ISSN: 0092-8674            Impact factor:   41.582


  98 in total

1.  The N-terminal LIM domain negatively regulates the kinase activity of LIM-kinase 1.

Authors:  K Nagata; K Ohashi; N Yang; K Mizuno
Journal:  Biochem J       Date:  1999-10-01       Impact factor: 3.857

Review 2.  Visuospatial construction.

Authors:  C B Mervis; B F Robinson; J R Pani
Journal:  Am J Hum Genet       Date:  1999-11       Impact factor: 11.025

3.  Williams syndrome: an update on clinical and molecular aspects.

Authors:  K Metcalfe
Journal:  Arch Dis Child       Date:  1999-09       Impact factor: 3.791

Review 4.  Behavioural phenotypes: what do they teach us?

Authors:  D H Skuse
Journal:  Arch Dis Child       Date:  2000-03       Impact factor: 3.791

5.  The Turner syndrome-associated neurocognitive phenotype maps to distal Xp.

Authors:  J L Ross; D Roeltgen; H Kushner; F Wei; A R Zinn
Journal:  Am J Hum Genet       Date:  2000-08-08       Impact factor: 11.025

6.  Generation and comparative analysis of approximately 3.3 Mb of mouse genomic sequence orthologous to the region of human chromosome 7q11.23 implicated in Williams syndrome.

Authors:  Udaya DeSilva; Laura Elnitski; Jacquelyn R Idol; Johannah L Doyle; Weiniu Gan; James W Thomas; Scott Schwartz; Nicole L Dietrich; Stephen M Beckstrom-Sternberg; Jennifer C McDowell; Robert W Blakesley; Gerard G Bouffard; Pamela J Thomas; Jeffrey W Touchman; Webb Miller; Eric D Green
Journal:  Genome Res       Date:  2002-01       Impact factor: 9.043

7.  A 1.5 million-base pair inversion polymorphism in families with Williams-Beuren syndrome.

Authors:  L R Osborne; M Li; B Pober; D Chitayat; J Bodurtha; A Mandel; T Costa; T Grebe; S Cox; L C Tsui; S W Scherer
Journal:  Nat Genet       Date:  2001-11       Impact factor: 38.330

8.  Control of growth cone motility and morphology by LIM kinase and Slingshot via phosphorylation and dephosphorylation of cofilin.

Authors:  Mitsuharu Endo; Kazumasa Ohashi; Yukio Sasaki; Yoshio Goshima; Ryusuke Niwa; Tadashi Uemura; Kensaku Mizuno
Journal:  J Neurosci       Date:  2003-04-01       Impact factor: 6.167

9.  Genomic interval engineering of mice identifies a novel modulator of triglyceride production.

Authors:  Y Zhu; M C Jong; K A Frazer; E Gong; R M Krauss; J F Cheng; D Boffelli; E M Rubin
Journal:  Proc Natl Acad Sci U S A       Date:  2000-02-01       Impact factor: 11.205

Review 10.  Methodological issues in group-matching designs: alpha levels for control variable comparisons and measurement characteristics of control and target variables.

Authors:  Carolyn B Mervis; Bonita P Klein-Tasman
Journal:  J Autism Dev Disord       Date:  2004-02
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