Divisions of Pediatric Cardiology, Duke University Medical Center, Durham, NC, USA.
Medical Genetics, Department of Pediatrics, Duke University Medical Center, DUMC 103856, 595 Lasalle Street, GSRB 1, 4th Floor, Room 4010, Durham, NC, 27710, USA.
J Inherit Metab Dis. 2017 Nov;40(6):823-830. doi: 10.1007/s10545-017-0072-0. Epub 2017 Aug 11.
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.
PRKAG2 编码 AMP 激活的蛋白激酶 (AMPK) 的 γ2 亚基,它是心脏代谢的重要调节剂。PRKAG2 突变导致一种心脏综合征,包括心室肥厚、预激和进行性传导系统疾病,通常直到青少年或成年早期才被诊断出来。然而,PRKAG2 突变患者的表现和结局存在显著的可变性,婴儿期的表现被低估。PRKAG2 的诊断在婴儿中具有挑战性,我们描述了我们对最初怀疑患有庞贝病但最终被诊断为 PRKAG2 突变的 3 名患者的经验。在每个病例中都发现了一种致病的 PRKAG2 突变,其中一名患者描述了一种新的错义突变。我们强调了 PRKAG2 突变患者在婴儿期可能模仿庞贝病的可能性,以及在诊断庞贝病时需要进行确认性检测。