Literature DB >> 22106044

Long QT, syndactyly, joint contractures, stroke and novel CACNA1C mutation: expanding the spectrum of Timothy syndrome.

Jane Gillis1, Elena Burashnikov, Charles Antzelevitch, Susan Blaser, Gil Gross, Lesley Turner, Riyana Babul-Hirji, David Chitayat.   

Abstract

Timothy syndrome (TS) is an autosomal dominant condition with the constellation of features including prolonged QT interval, hand and foot abnormalities, and mental retardation or autism. Splawski et al. [2004] previously described two phenotypes associated with TS distinguished by two unique and different mutations within the CACNA1C gene. We report on a newborn who presented with prolonged QT interval and associated polymorphic ventricular tachycardia, dysmorphic facial features, syndactyly of the hands and feet, and joint contractures, suggestive of TS. He developed a stroke, subsequent intractable seizures, and was found to have cortical blindness and later profound developmental delay. Initial targeted mutation analysis did not identify either of the previously described TS associated mutations; however, full gene sequencing detected a novel CACNA1C gene mutation (p.Ala1473Gly). The clinical and genetic findings in our case expand both the clinical and molecular knowledge of TS.
Copyright © 2011 Wiley Periodicals, Inc.

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Year:  2011        PMID: 22106044      PMCID: PMC3319791          DOI: 10.1002/ajmg.a.34355

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

1.  Transcript scanning reveals novel and extensive splice variations in human l-type voltage-gated calcium channel, Cav1.2 alpha1 subunit.

Authors:  Zhen Zhi Tang; Mui Cheng Liang; Songqing Lu; Dejie Yu; Chye Yun Yu; David T Yue; Tuck Wah Soong
Journal:  J Biol Chem       Date:  2004-08-06       Impact factor: 5.157

2.  Syndactyly and long QT syndrome (CaV1.2 missense mutation G406R) is associated with hypertrophic cardiomyopathy.

Authors:  Shirley M Lo-A-Njoe; Arthur A Wilde; Lieselot van Erven; Nico A Blom
Journal:  Heart Rhythm       Date:  2005-12       Impact factor: 6.343

3.  Ca(v)1.2 splice variant with exon 9* is critical for regulation of cerebral artery diameter.

Authors:  Matthew A Nystoriak; Kentaro Murakami; Paul L Penar; George C Wellman
Journal:  Am J Physiol Heart Circ Physiol       Date:  2009-08-28       Impact factor: 4.733

Review 4.  The functions of two species of calcium channel in cardiac muscle excitation-contraction coupling.

Authors:  A J Williams
Journal:  Eur Heart J       Date:  1997-01       Impact factor: 29.983

5.  Severe arrhythmia disorder caused by cardiac L-type calcium channel mutations.

Authors:  Igor Splawski; Katherine W Timothy; Niels Decher; Pradeep Kumar; Frank B Sachse; Alan H Beggs; Michael C Sanguinetti; Mark T Keating
Journal:  Proc Natl Acad Sci U S A       Date:  2005-04-29       Impact factor: 11.205

6.  Ca(V)1.2 calcium channel dysfunction causes a multisystem disorder including arrhythmia and autism.

Authors:  Igor Splawski; Katherine W Timothy; Leah M Sharpe; Niels Decher; Pradeep Kumar; Raffaella Bloise; Carlo Napolitano; Peter J Schwartz; Robert M Joseph; Karen Condouris; Helen Tager-Flusberg; Silvia G Priori; Michael C Sanguinetti; Mark T Keating
Journal:  Cell       Date:  2004-10-01       Impact factor: 41.582

7.  Long QT syndrome associated with syndactyly identified in females.

Authors:  M L Marks; D L Trippel; M T Keating
Journal:  Am J Cardiol       Date:  1995-10-01       Impact factor: 2.778

Review 8.  A new form of long QT syndrome associated with syndactyly.

Authors:  M L Marks; S L Whisler; C Clericuzio; M Keating
Journal:  J Am Coll Cardiol       Date:  1995-01       Impact factor: 24.094

9.  [The heart-hand syndrome. A new variant of disorders of heart conduction and syndactylia including osseous changes in hands and feet].

Authors:  H Reichenbach; E M Meister; H Theile
Journal:  Kinderarztl Prax       Date:  1992-04
  9 in total
  41 in total

1.  Expanding clinical phenotype in CACNA1C related disorders: From neonatal onset severe epileptic encephalopathy to late-onset epilepsy.

Authors:  Xiuhua Bozarth; Jennifer N Dines; Qian Cong; Ghayda M Mirzaa; Kimberly Foss; J Lawrence Merritt; Jenny Thies; Heather C Mefford; Edward Novotny
Journal:  Am J Med Genet A       Date:  2018-12-04       Impact factor: 2.802

Review 2.  Inherited calcium channelopathies in the pathophysiology of arrhythmias.

Authors:  Luigi Venetucci; Marco Denegri; Carlo Napolitano; Silvia G Priori
Journal:  Nat Rev Cardiol       Date:  2012-06-26       Impact factor: 32.419

Review 3.  Calcium Revisited: New Insights Into the Molecular Basis of Long-QT Syndrome.

Authors:  John R Giudicessi; Michael J Ackerman
Journal:  Circ Arrhythm Electrophysiol       Date:  2016-07

4.  Novel Timothy syndrome mutation leading to increase in CACNA1C window current.

Authors:  Nicole J Boczek; Erin M Miller; Dan Ye; Vladislav V Nesterenko; David J Tester; Charles Antzelevitch; Richard J Czosek; Michael J Ackerman; Stephanie M Ware
Journal:  Heart Rhythm       Date:  2014-09-28       Impact factor: 6.343

5.  Functional characterization of CaVα2δ mutations associated with sudden cardiac death.

Authors:  Benoîte Bourdin; Behzad Shakeri; Marie-Philippe Tétreault; Rémy Sauvé; Sylvie Lesage; Lucie Parent
Journal:  J Biol Chem       Date:  2014-12-19       Impact factor: 5.157

6.  Dysfunctional Cav1.2 channel in Timothy syndrome, from cell to bedside.

Authors:  Dan Han; Xiaolin Xue; Yang Yan; Guoliang Li
Journal:  Exp Biol Med (Maywood)       Date:  2019-07-19

7.  Exome sequencing and systems biology converge to identify novel mutations in the L-type calcium channel, CACNA1C, linked to autosomal dominant long QT syndrome.

Authors:  Nicole J Boczek; Jabe M Best; David J Tester; John R Giudicessi; Sumit Middha; Jared M Evans; Timothy J Kamp; Michael J Ackerman
Journal:  Circ Cardiovasc Genet       Date:  2013-06

Review 8.  Practical Aspects in Genetic Testing for Cardiomyopathies and Channelopathies.

Authors:  Han-Chih Hencher Lee; Chor-Kwan Ching
Journal:  Clin Biochem Rev       Date:  2019-11

Review 9.  From Gene to Behavior: L-Type Calcium Channel Mechanisms Underlying Neuropsychiatric Symptoms.

Authors:  Zeeba D Kabir; Arlene Martínez-Rivera; Anjali M Rajadhyaksha
Journal:  Neurotherapeutics       Date:  2017-07       Impact factor: 7.620

10.  Identification and Functional Characterization of a Novel CACNA1C-Mediated Cardiac Disorder Characterized by Prolonged QT Intervals With Hypertrophic Cardiomyopathy, Congenital Heart Defects, and Sudden Cardiac Death.

Authors:  Nicole J Boczek; Dan Ye; Fang Jin; David J Tester; April Huseby; J Martijn Bos; Aaron J Johnson; Ronald Kanter; Michael J Ackerman
Journal:  Circ Arrhythm Electrophysiol       Date:  2015-08-07
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