Koh Carline, Lee Pui-Wah, Yung Tak-Cheung, Lun Kin-Shing, Cheung Yiu-Fai
Department of Paediatrics and Adolescent Medicine, Division of Paediatric Cardiology, Grantham Hospital, The University of Hong Kong, Hong Kong, China.
Congenit Heart Dis. 2009 Jul-Aug;4(4):288-94. doi: 10.1111/j.1747-0803.2009.00269.x.
Left ventricular noncompaction (LVNC) is an uncommon type of cardiomyopathy in children. We sought to determine the clinical presentations and outcomes of children diagnosed to have LVNC.
The case records of children diagnosed to have LVNC between 1999 and 2007 were reviewed. The diagnosis was based on echocardiographic finding of a thick noncompacted myocardium layer characterized by a trabecular meshwork with deep endomyocardial spaces.
Ten patients (seven males) were diagnosed to have LVNC at a median age of 2 years (range, 7 days to 12 years). Seven patients had isolated LVNC while three had associated structural congenital heart diseases. The right ventricle was also involved in two patients. Clinical presentations included congestive heart failure in eight patients, asymptomatic heart murmur in one, and syncope with exercise intolerance in one. At presentation, cardiomegaly was found in nine patients, electrocardiographic abnormalities in nine, and impaired LV contraction in six. Eight patients received anti-heart failure medications. Three patients died at a median of 1.5 years after diagnosis, two died suddenly of unknown causes, and one of heart failure. The seven surviving patients were followed up for a median of 2 years (range, 2 months to 3 years). Three patients developed cardiac arrhythmias. The LV function improved in three patients and worsened in one on follow-up. None of the patients developed thromboembolic complications.
Left ventricular noncompaction in children is a heterogeneous condition. Long-term follow-up for development of progressive LV dysfunction and cardiac arrhythmias is indicated.
左心室心肌致密化不全(LVNC)是儿童中一种不常见的心肌病类型。我们试图确定诊断为LVNC的儿童的临床表现和预后。
回顾了1999年至2007年间诊断为LVNC的儿童的病例记录。诊断基于超声心动图发现的增厚的非致密心肌层,其特征为带有深陷心内膜间隙的小梁网状结构。
10例患者(7例男性)被诊断为LVNC,中位年龄为2岁(范围为7天至12岁)。7例患者为孤立性LVNC,3例合并结构性先天性心脏病。2例患者右心室也受累。临床表现包括8例充血性心力衰竭、1例无症状心脏杂音、1例运动不耐受伴晕厥。就诊时,9例患者发现心脏扩大,9例心电图异常,6例左心室收缩功能受损。8例患者接受了抗心力衰竭药物治疗。3例患者在诊断后中位1.5年死亡,2例不明原因突然死亡,1例死于心力衰竭。7例存活患者中位随访2年(范围为2个月至3年)。3例患者发生心律失常。随访中3例患者左心室功能改善,1例恶化。所有患者均未发生血栓栓塞并发症。
儿童左心室心肌致密化不全是一种异质性疾病。需要对进行性左心室功能障碍和心律失常的发生进行长期随访。