Haffajee Jessica A, Finley John J, Brooks Erica L, Kuvin Jeffrey T, Patel Ayan R
Cardiovascular Imaging and Hemodynamic Laboratory, Tufts Medical Center, Boston 02111 MA, USA.
Eur J Echocardiogr. 2011 Mar;12(3):E17. doi: 10.1093/ejechocard/jeq170. Epub 2010 Dec 4.
Left ventricular (LV) apical hypoplasia is an unusual, recently identified cardiomyopathy, whose clinical course is uncertain. In this report, we describe a case of this cardiomyopathy occurring in an asymptomatic 50-year-old male with a remote history of a surgically corrected patent ductus arteriosus (PDA), primarily using transthoracic echocardiography (TTE) to illustrate the imaging characteristics. This patient had been referred to our institution for an abnormal electrocardiogram, and TTE subsequently (Figure 1) revealed a dilated left ventricle with moderately to severely reduced function; LV ejection fraction was 30% by two- and three-dimensional quantification. The left ventricle had a spherical appearance with a thin-walled, truncated, and akinetic distal LV. The right ventricle appeared elongated and was noted to wrap around the distal left ventricle, but right ventricular systolic function was normal. There were no significant valvular abnormalities, and no evidence of residual PDA flow. Subsequent cardiac magnetic resonance (CMR) imaging confirmed these findings (Figure 1). The TTE and CMR findings seen in this patient are consistent with LV apical hypoplasia. Until now, this cardiomyopathy has been described only as an isolated congenital anomaly primarily using CMR and cardiac computed tomography. To our knowledge, this is the first reported case of LV apical hypoplasia in conjunction with another congenital cardiac abnormality, and the findings demonstrate that the distinctive appearance of this cardiomyopathy can be easily identified with echocardiography. As more cases are recognized and patients are followed over time, the natural history and optimal treatment for this cardiomyopathy may be further elucidated.
左心室心尖发育不全是一种罕见的、最近才被确认的心肌病,其临床病程尚不确定。在本报告中,我们描述了一例发生在一名无症状50岁男性身上的这种心肌病,该患者有动脉导管未闭(PDA)手术矫正的既往史,主要使用经胸超声心动图(TTE)来说明其影像学特征。该患者因心电图异常转诊至我院,随后TTE(图1)显示左心室扩张,功能中度至重度降低;二维和三维定量分析显示左心室射血分数为30%。左心室呈球形,远端左心室壁薄、截断且运动减弱。右心室显得拉长,并环绕远端左心室,但右心室收缩功能正常。无明显瓣膜异常,也无动脉导管未闭残余血流的证据。随后的心脏磁共振(CMR)成像证实了这些发现(图1)。该患者的TTE和CMR表现与左心室心尖发育不全相符。到目前为止,这种心肌病仅被描述为一种主要通过CMR和心脏计算机断层扫描发现的孤立先天性异常。据我们所知,这是第一例报告的左心室心尖发育不全合并另一种先天性心脏异常的病例,这些发现表明这种心肌病的独特外观可以通过超声心动图轻松识别。随着更多病例被识别并对患者进行长期随访,这种心肌病的自然病程和最佳治疗方法可能会得到进一步阐明。