Khalil Ibrahim, Abrar Sakib, Hossain Md Imran
Department of Medicine, Dhaka Medical College and Hospital, Dhaka, Bangladesh.
Manikganj Medical College, Faculty of Medicine, Dhaka University, Dhaka, Bangladesh.
Radiol Case Rep. 2025 May 22;20(8):3930-3937. doi: 10.1016/j.radcr.2025.04.075. eCollection 2025 Aug.
Double Outlet Right Ventricle (DORV) is a rare congenital heart defect characterized by both the aorta and pulmonary artery originating from the right ventricle. It accounts for 1%-3% of all congenital heart defects and is often associated with additional structural anomalies such as ventricular septal defects (VSDs), which exacerbate hemodynamic complications. We present a case of a 16-year-old female with DORV and 2 VSDs, leading to bidirectional shunting and subsequent pulmonary hypertension. The patient presented with progressive exertional dyspnea, central cyanosis, and clubbing of the fingers, indicative of chronic hypoxia. Echocardiography and Doppler studies confirmed the diagnosis, revealing significant bidirectional shunting and moderate pulmonary hypertension. Two distinct VSDs were identified in the interventricular septum (IVS): the first, a larger perimembranous VSD involving the inlet part with extensions toward the subaortic region, measured 11 mm × 16 mm; the second, a smaller VSD in the mid-part of the trabecular septum, measured 4 mm × 4 mm. Both VSDs permitted bidirectional shunting, predominantly left-to-right, contributing to the patient's cyanosis and pulmonary hypertension, with the larger VSD being routable to the aorta. Treatment involved stabilization with medical management to address pulmonary hypertension and preparation for corrective surgery. Timely surgical correction, including the closure of VSDs and rerouting of the ventriculo-arterial connections, is crucial for improving outcomes. Ongoing follow-up is essential to monitor for complications such as arrhythmias, right heart failure, and progression of pulmonary hypertension. The case underscores the complexity of DORV and the need for a multidisciplinary approach in its management.
右心室双出口(DORV)是一种罕见的先天性心脏缺陷,其特征是主动脉和肺动脉均起源于右心室。它占所有先天性心脏缺陷的1%-3%,并且常与其他结构异常相关,如室间隔缺损(VSD),这会加剧血流动力学并发症。我们报告一例16岁女性患有DORV和2个VSD,导致双向分流及随后的肺动脉高压。患者表现为进行性劳力性呼吸困难、中心性发绀和手指杵状指,提示慢性缺氧。超声心动图和多普勒研究确诊了该疾病,显示存在显著的双向分流和中度肺动脉高压。在室间隔(IVS)中发现了两个不同的VSD:第一个是较大的膜周部VSD,累及入口部分并向主动脉下区域延伸,大小为11 mm×16 mm;第二个是小梁间隔中部较小的VSD,大小为4 mm×4 mm。两个VSD均允许双向分流,主要是左向右分流,导致患者发绀和肺动脉高压,较大的VSD可通向主动脉。治疗包括通过药物治疗稳定病情以控制肺动脉高压,并为矫正手术做准备。及时进行手术矫正,包括关闭VSD和重新构建心室-动脉连接,对于改善预后至关重要。持续随访对于监测心律失常、右心衰竭和肺动脉高压进展等并发症至关重要。该病例强调了DORV的复杂性以及在其管理中采用多学科方法的必要性。