Rashidighader Fariba, Mirzaaghayan Mohammad Reza
Department of Pediatric Cardiology, Rajaie Cardiovascular Medical and Research Institute, Iran University of Medical Sciences, Tehran, Iran.
Department of Cardiac Surgery, School of Medicine, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran.
J Tehran Heart Cent. 2024 Jan;19(1):60-65. doi: 10.18502/jthc.v19i1.15614.
Absent pulmonary valve syndrome (APVS) is a rare congenital anomaly characterized by rudimentary PV tissue with variable degrees of PV stenosis and regurgitant pulmonary blood flow. In most cases, it is associated with tetralogy of Fallot. In a minority of APVS cases, with an unknown frequency, intact ventricular septum (IVS), patent ductus arteriosus, and possible tricuspid atresia are present. This condition is known as non-Fallot type APVS. We describe a case of an antenatal diagnosis of APVS with IVS, a large patent ductus arteriosus, and ascending aorta dilatation. The mother was referred to our center at 32 weeks of gestation due to cardiomegaly on sonography. Fetal echocardiography revealed cardiomegaly, right atrial and ventricular enlargement, aneurysmal dilatation of the main pulmonary artery, and mild dilatation of the pulmonary artery branches. Also observed were IVS, rudimentary PV tissue with severe to-and-fro turbulence across the PV, a widely open ductus arteriosus, ascending aorta dilatation, and levorotation of the heart. After childbirth, our diagnosis was confirmed by echocardiography and surgery. The baby experienced severe respiratory distress. At 15 days of life, surgical intervention in the form of pulmonary artery arterioplasty was performed, resulting in good outcomes. The patient underwent follow-up for 6 months and showed reasonable health.
肺动脉瓣缺如综合征(APVS)是一种罕见的先天性异常,其特征为肺动脉瓣组织发育不全,伴有不同程度的肺动脉瓣狭窄和肺血流反流。在大多数情况下,它与法洛四联症相关。在少数APVS病例中,频率不详,存在室间隔完整(IVS)、动脉导管未闭以及可能的三尖瓣闭锁。这种情况被称为非法洛型APVS。我们描述了一例产前诊断为伴有IVS、大型动脉导管未闭和升主动脉扩张的APVS病例。母亲因超声检查发现心脏肿大,于妊娠32周时被转诊至我们中心。胎儿超声心动图显示心脏肿大、右心房和右心室扩大、主肺动脉瘤样扩张以及肺动脉分支轻度扩张。还观察到IVS、肺动脉瓣组织发育不全,肺动脉瓣处有严重的往返湍流、动脉导管广泛开放、升主动脉扩张以及心脏左旋。分娩后,超声心动图和手术证实了我们的诊断。婴儿出现严重呼吸窘迫。出生15天时,进行了肺动脉成形术的手术干预,结果良好。该患者接受了6个月的随访,健康状况良好。