Literature DB >> 28801758

PRKAG2 mutations presenting in infancy.

Rachel D Torok1, Stephanie L Austin2, Chanika Phornphutkul3, Kathleen M Rotondo4, Deeksha Bali2, Gregory H Tatum1, Stephanie B Wechsler1,2, Anne F Buckley5, Priya S Kishnani6.   

Abstract

PRKAG2 encodes the γ2 subunit of AMP-activated protein kinase (AMPK), which is an important regulator of cardiac metabolism. Mutations in PRKAG2 cause a cardiac syndrome comprising ventricular hypertrophy, pre-excitation, and progressive conduction-system disease, which is typically not diagnosed until adolescence or young adulthood. However, significant variability exists in the presentation and outcomes of patients with PRKAG2 mutations, with presentation in infancy being underrecognized. The diagnosis of PRKAG2 can be challenging in infants, and we describe our experience with three patients who were initially suspected to have Pompe disease yet ultimately diagnosed with mutations in PRKAG2. A disease-causing PRKAG2 mutation was identified in each case, with a novel missense mutation described in one patient. We highlight the potential for patients with PRKAG2 mutations to mimic Pompe disease in infancy and the need for confirmatory testing when diagnosing Pompe disease.

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Year:  2017        PMID: 28801758     DOI: 10.1007/s10545-017-0072-0

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  28 in total

Review 1.  Hypertrophic cardiomyopathy in childhood.

Authors:  Steven D Colan
Journal:  Heart Fail Clin       Date:  2010-10       Impact factor: 3.179

2.  Alglucosidase alfa enzyme replacement therapy as a therapeutic approach for a patient presenting with a PRKAG2 mutation.

Authors:  Stephanie L Austin; Andrew Chiou; Baodong Sun; Laura E Case; Kenny Govendrageloo; Perrin Hansen; Priya S Kishnani
Journal:  Mol Genet Metab       Date:  2016-09-28       Impact factor: 4.797

3.  Methods of diagnosis of patients with Pompe disease: Data from the Pompe Registry.

Authors:  Priya S Kishnani; Hernán M Amartino; Christopher Lindberg; Timothy M Miller; Amanda Wilson; Joan Keutzer
Journal:  Mol Genet Metab       Date:  2014-07-16       Impact factor: 4.797

4.  Identification of a gene responsible for familial Wolff-Parkinson-White syndrome.

Authors:  M H Gollob; M S Green; A S Tang; T Gollob; A Karibe; A S Ali Hassan ; F Ahmad; R Lozado; G Shah; L Fananapazir; L L Bachinski; R Roberts; A S Hassan
Journal:  N Engl J Med       Date:  2001-06-14       Impact factor: 91.245

5.  Adenosine monophosphate-activated protein kinase disease mimicks hypertrophic cardiomyopathy and Wolff-Parkinson-White syndrome: natural history.

Authors:  Ross T Murphy; Jens Mogensen; Kate McGarry; Ajay Bahl; Alison Evans; Eyman Osman; Petros Syrris; Grainne Gorman; Michael Farrell; Janice L Holton; Michael G Hanna; Sian Hughes; Perry M Elliott; Calum A Macrae; William J McKenna
Journal:  J Am Coll Cardiol       Date:  2005-03-15       Impact factor: 24.094

6.  Identification of LAMP2 Mutations in Early-Onset Danon Disease With Hypertrophic Cardiomyopathy by Targeted Next-Generation Sequencing.

Authors:  Lijun Fu; Sushan Luo; Shuang Cai; Wenjing Hong; Ying Guo; Jinjin Wu; Tingliang Liu; Chongbo Zhao; Fen Li; Huimin Huang; Meirong Huang; Jian Wang
Journal:  Am J Cardiol       Date:  2016-06-27       Impact factor: 2.778

7.  A new mutation in PRKAG2 gene causing hypertrophic cardiomyopathy with conduction system disease and muscular glycogenosis.

Authors:  Pascal Laforêt; Pascale Richard; Mina Ait Said; Norma Beatriz Romero; Emmanuelle Lacene; Jean-Paul Leroy; Christiane Baussan; Jean-Yves Hogrel; Thomas Lavergne; Karim Wahbi; Bernard Hainque; Denis Duboc
Journal:  Neuromuscul Disord       Date:  2006-02-17       Impact factor: 4.296

Review 8.  Minireview: the AMP-activated protein kinase cascade: the key sensor of cellular energy status.

Authors:  D Grahame Hardie
Journal:  Endocrinology       Date:  2003-09-04       Impact factor: 4.736

9.  Hypertrophic cardiomyopathy during corticotropin therapy for infantile spasms. A clinical and echocardiographic study.

Authors:  G B Bobele; K E Ward; J B Bodensteiner
Journal:  Am J Dis Child       Date:  1993-02

10.  Familial Hypertrophic cardiomyopathy with Wolff-Parkinson-White syndrome maps to a locus on chromosome 7q3.

Authors:  C A MacRae; N Ghaisas; S Kass; S Donnelly; C T Basson; H C Watkins; R Anan; L H Thierfelder; K McGarry; E Rowland
Journal:  J Clin Invest       Date:  1995-09       Impact factor: 14.808

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  2 in total

1.  Infantile Onset Hypertrophic Cardiomyopathy Secondary to PRKAG2 Gene Mutation is Associated with Poor Prognosis.

Authors:  Sudheer R Gorla; Kishore R Raja; Ashish Garg; Deborah S Barbouth; Paolo G Rusconi
Journal:  J Pediatr Genet       Date:  2018-06-02

2.  Identification of candidate genes that specifically regulate subcutaneous and intramuscular fat deposition using transcriptomic and proteomic profiles in Dingyuan pigs.

Authors:  Pan Zhang; Qinggang Li; Yijing Wu; Yawen Zhang; Bo Zhang; Hao Zhang
Journal:  Sci Rep       Date:  2022-02-18       Impact factor: 4.379

  2 in total

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