Revell S P, Noble-Jamieson G, Johnston P, Rasmussen A, Jamieson N, Barnes N D
Department of Paediatrics, Addenbrookes NHS Trust, Cambridge.
Arch Dis Child. 1995 Nov;73(5):456-8. doi: 10.1136/adc.73.5.456.
Homozygous familial hypercholesterolaemia is a rare inherited condition with an incidence of approximately one in a million. It is associated with severe premature atherosclerosis and early death from cardiovascular complications. The results of liver transplantation reported to date have suggested only partially effective reduction of the hypercholesterolaemia. Three boys with familial hypercholesterolaemia, aged 10.0 to 15.1 years, received liver grafts at Addenbrooke's Hospital. Their untreated fasting lipid concentrations were grossly raised. All three had angiographic evidence of coronary atheroma and two had exertional angina. One child had such severe atheroma that coronary artery bypass surgery was considered necessary before liver transplantation. All three had straightforward operative and postoperative courses and their lipid concentrations returned rapidly to normal. One boy developed chronic rejection requiring retransplantation. Currently all three boys are well, on normal diets, and with normal liver function. It is concluded that (1) liver transplantation offers highly effective treatment for this lethal condition, (2) timing the operation is difficult but it should be undertaken before coronary artery disease has progressed too far (when combined liver and heart transplantation may be the only possibility), and (3) in well grown children with no previous abdominal surgery the immediate risks of liver transplantation are low but chronic rejection remains a danger.
纯合子家族性高胆固醇血症是一种罕见的遗传性疾病,发病率约为百万分之一。它与严重的早发性动脉粥样硬化和心血管并发症导致的早期死亡有关。迄今为止报道的肝移植结果表明,高胆固醇血症仅得到部分有效缓解。三名患有家族性高胆固醇血症的男孩,年龄在10.0至15.1岁之间,在阿登布鲁克医院接受了肝脏移植。他们未经治疗的空腹血脂浓度大幅升高。三人都有冠状动脉粥样硬化的血管造影证据,两人有劳力性心绞痛。一名儿童的动脉粥样硬化非常严重,以至于在肝移植前被认为有必要进行冠状动脉搭桥手术。三人的手术过程和术后恢复都很顺利,血脂浓度迅速恢复正常。一名男孩发生了慢性排斥反应,需要再次移植。目前,这三名男孩情况良好,饮食正常,肝功能正常。结论是:(1)肝移植为这种致命疾病提供了高效治疗;(2)手术时机难以把握,但应在冠状动脉疾病进展到太严重之前进行(此时联合肝心移植可能是唯一的选择);(3)在没有腹部手术史的发育良好的儿童中,肝移植的直接风险较低,但慢性排斥反应仍然是一个危险因素。