Yuan Shi-Min, Shinfeld Amihay, Mishaly David
Department of Cardiac and Thoracic Surgery, The Safra Children's Hospital, The Chaim Sheba Medical Center, Israel.
Chang Gung Med J. 2008 May-Jun;31(3):309-13.
Pentalogy of Cantrell is a rare entity of congenital defects involving the abdominal wall, sternum, diaphragm, pericardium and heart. The complete pentalogy and its variants have been described in the literature. We report a 47-day-old girl with an incomplete pentalogy of Cantrell presenting with congenital absence of the sternum, congenital heart defects, and an epigastric hernia. Two even rarer expressions of this disorder, tricuspid atresia type IIc and asymmetric kidneys, were also found. The patient was successfully managed with palliative pulmonary artery banding as preparation for a modified Fontan operation when she is older. We report this rare case, and discuss the pathologic findings and surgical strategy.
坎特雷尔五联症是一种罕见的先天性缺陷,涉及腹壁、胸骨、膈肌、心包和心脏。文献中已描述了完整的五联症及其变体。我们报告了一名47天大的女孩,患有不完全性坎特雷尔五联症,表现为先天性胸骨缺失、先天性心脏缺陷和脐上疝。还发现了该疾病的另外两种更罕见的表现,即IIc型三尖瓣闭锁和不对称肾脏。该患者通过姑息性肺动脉环扎术成功治疗,为其长大后进行改良Fontan手术做准备。我们报告了这个罕见病例,并讨论了病理结果和手术策略。