Literature DB >> 12070241

The p63 gene in EEC and other syndromes.

H G Brunner1, B C J Hamel, H Van Bokhoven.   

Abstract

Several autosomal dominantly inherited human syndromes have recently been shown to result from mutations in the p63 gene. These syndromes have various combinations of limb malformations fitting the split hand-split foot spectrum, orofacial clefting, and ectodermal dysplasia. The p63 syndrome family includes the EEC syndrome, AEC syndrome, ADULT syndrome, limb-mammary syndrome, and non-syndromic split hand/foot malformation. The pattern of heterozygous mutations is distinct for each of these syndromes. The functional effects on the p63 proteins also vary between syndromes. In all of these syndromes, the mutation appears to have both dominant negative and gain of function effects rather than causing a simple loss of function.

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Year:  2002        PMID: 12070241      PMCID: PMC1735150          DOI: 10.1136/jmg.39.6.377

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  30 in total

1.  p63, a p53 homolog at 3q27-29, encodes multiple products with transactivating, death-inducing, and dominant-negative activities.

Authors:  A Yang; M Kaghad; Y Wang; E Gillett; M D Fleming; V Dötsch; N C Andrews; D Caput; F McKeon
Journal:  Mol Cell       Date:  1998-09       Impact factor: 17.970

2.  Limb mammary syndrome: a new genetic disorder with mammary hypoplasia, ectrodactyly, and other Hand/Foot anomalies maps to human chromosome 3q27.

Authors:  H van Bokhoven; M Jung; A P Smits; S van Beersum; F Rüschendorf; M van Steensel; M Veenstra; J H Tuerlings; E C Mariman; H G Brunner; T F Wienker; A Reis; H H Ropers; B C Hamel
Journal:  Am J Hum Genet       Date:  1999-02       Impact factor: 11.025

Review 3.  The EEC syndrome: a literature study.

Authors:  N M Roelfsema; J M Cobben
Journal:  Clin Dysmorphol       Date:  1996-04       Impact factor: 0.816

4.  The syndrome of ankyloblepharon, ectodermal defects and cleft lip and palate: an autosomal dominant condition.

Authors:  R J Hay; R S Wells
Journal:  Br J Dermatol       Date:  1976-03       Impact factor: 9.302

5.  The lacrimo-auriculo-dento-digital syndrome.

Authors:  D W Hollister; S H Klein; H J De Jager; R S Lachman; D L Rimoin
Journal:  J Pediatr       Date:  1973-09       Impact factor: 4.406

6.  Anhidrotic ectodermal dysplasia: autosomal dominant inheritance with palate and lip anomalies.

Authors:  R S Rapp; W E Hodgkin
Journal:  J Med Genet       Date:  1968-12       Impact factor: 6.318

7.  Alveolar synechia-ankyloblepharon-ectodermal defects likely CHANDS.

Authors:  H V Toriello
Journal:  Am J Med Genet       Date:  1994-02-01

8.  Ectodermal dysplasia, mental retardation, cleft lip/palate and other anomalies in three sibs.

Authors:  P Bowen; H B Armstrong
Journal:  Clin Genet       Date:  1976-01       Impact factor: 4.438

9.  Gain-of-function mutation in ADULT syndrome reveals the presence of a second transactivation domain in p63.

Authors:  Pascal H G Duijf; Kaate R J Vanmolkot; Peter Propping; Waltraut Friedl; Elmar Krieger; Frank McKeon; Volker Dötsch; Han G Brunner; Hans van Bokhoven
Journal:  Hum Mol Genet       Date:  2002-04-01       Impact factor: 6.150

10.  Ectodermal dysplasia, Rapp-Hodgkin type in a mother and severe ectrodactyly-ectodermal dysplasia-clefting syndrome (EEC) in her child.

Authors:  P Moerman; J P Fryns
Journal:  Am J Med Genet       Date:  1996-06-14
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  49 in total

1.  Why study human limb malformations?

Authors:  Andrew O M Wilkie
Journal:  J Anat       Date:  2003-01       Impact factor: 2.610

Review 2.  The role of p53 gene family in reproduction.

Authors:  Wenwei Hu
Journal:  Cold Spring Harb Perspect Biol       Date:  2009-10-28       Impact factor: 10.005

3.  An unusual combination of EEC syndrome and hypomelanosis Ito due to a p63 mutation.

Authors:  Katarina Lehmann; Stefan Mundlos; Peter Meinecke
Journal:  Eur J Pediatr       Date:  2005-05-12       Impact factor: 3.183

4.  Severe persistent nasolacrimal duct obstruction: a typical finding in ADULT syndrome.

Authors:  N Eter; K Zerres; P Propping; P Roggenkämper; M Spitznas
Journal:  Br J Ophthalmol       Date:  2006-09       Impact factor: 4.638

5.  p63 Regulates adult neural precursor and newly born neuron survival to control hippocampal-dependent Behavior.

Authors:  Gonzalo I Cancino; Adelaide P Yiu; Michael P Fatt; Chandrasagar B Dugani; Elsa R Flores; Paul W Frankland; Sheena A Josselyn; Freda D Miller; David R Kaplan
Journal:  J Neurosci       Date:  2013-07-31       Impact factor: 6.167

6.  Two interesting cases of EEC syndrome.

Authors:  Mariyam Iqbal Ali; K Aravinda; Nitin Kumar Nigam; Iqbal Ali
Journal:  J Oral Biol Craniofac Res       Date:  2013-02-13

7.  Control of p53-dependent transcription and enhancer activity by the p53 family member p63.

Authors:  Gizem Karsli Uzunbas; Faraz Ahmed; Morgan A Sammons
Journal:  J Biol Chem       Date:  2019-05-21       Impact factor: 5.157

Review 8.  p63 in skin development and ectodermal dysplasias.

Authors:  Maranke I Koster
Journal:  J Invest Dermatol       Date:  2010-05-06       Impact factor: 8.551

9.  Impaired repair of cyclobutane pyrimidine dimers in human keratinocytes deficient in p53 and p63.

Authors:  Bridget E Ferguson-Yates; Hongyan Li; Tiffany K Dong; Jennifer L Hsiao; Dennis H Oh
Journal:  Carcinogenesis       Date:  2007-11-04       Impact factor: 4.944

Review 10.  Integrating animal models and in vitro tissue models to elucidate the role of desmosomal proteins in diseases.

Authors:  Maranke I Koster; Jason Dinella; Jiangli Chen; Charlene O'Shea; Peter J Koch
Journal:  Cell Commun Adhes       Date:  2014-02
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