Department of Surgery, Division of Gastroenterological, General and Transplant Surgery, Jichi Medical University, Shimotsuke, Toichigi, Japan.
Ann Transplant. 2021 Oct 1;26:e932994. doi: 10.12659/AOT.932994.
BACKGROUND There is no consensus about the long-term prognosis of pediatric patients with a variety of rare liver diseases but with inherited metabolic diseases (IMDs). We retrospectively reviewed the developmental outcomes of patients with IMDs undergoing living donor liver transplantation (LDLT). MATERIAL AND METHODS Between May 2001 and December 2020, of 314 pediatric patients who underwent LDLT, 44 (14%) had IMDs. The median age at LDLT was 3.0 years old (range 0-15.0 years). Associations between the post-transplant complications and graft survival rate in patients with IMDs and biliary atresia (BA) were calculated. We evaluated the safety of LDLT from heterozygous carrier donors, the prognosis of patients with IMDs who have metabolic defects expressed in other organs, and developmental outcomes of patients with IMDs. RESULTS The 10-year graft survival rates in patients with IMDs and BA were 87% and 94%, respectively (P=0.041), and the causes of graft failure included pneumocystis pneumonia, acute lung failure, hemophagocytic syndrome, hepatic vein thrombosis, portal vein thrombosis, and sepsis. The rate of post-transplant cytomegalovirus viremia in patients with IMDs was higher than that of patients with BA (P=0.039). Of 39 patients with IMDs, 15 patients (38%) had severe motor and intellectual disabilities in 4 patients, intellectual developmental disorders including epilepsy in 2, and attention-deficit hyperactivity disorder in 2. Of 28 patients with IMDs, 13 (46%) needed special education. CONCLUSIONS The long-term outcomes of LDLT in patients with IMDs are good. However, further long-term social and educational follow-up regarding intellectual developmental disorders is needed.
对于患有各种罕见肝脏疾病但伴有遗传性代谢疾病(IMD)的儿科患者,其长期预后尚无共识。我们回顾性分析了接受活体肝移植(LDLT)的 IMD 患者的发育结局。
2001 年 5 月至 2020 年 12 月,314 例接受 LDLT 的儿科患者中,44 例(14%)患有 IMD。LDLT 时的中位年龄为 3.0 岁(范围 0-15.0 岁)。计算了 IMD 患者与胆道闭锁(BA)患者移植后并发症与移植物存活率之间的相关性。我们评估了杂合子携带者供体 LDLT 的安全性、在其他器官表达代谢缺陷的 IMD 患者的预后以及 IMD 患者的发育结局。
IMD 患者和 BA 患者的 10 年移植物存活率分别为 87%和 94%(P=0.041),移植物衰竭的原因包括肺囊虫肺炎、急性呼吸衰竭、噬血细胞综合征、肝静脉血栓形成、门静脉血栓形成和败血症。IMD 患者移植后巨细胞病毒血症的发生率高于 BA 患者(P=0.039)。在 39 例 IMD 患者中,4 例有严重的运动和智力残疾,2 例有包括癫痫在内的智力发育障碍,2 例有注意力缺陷多动障碍。在 28 例 IMD 患者中,13 例(46%)需要特殊教育。
LDLT 治疗 IMD 患者的长期结局良好。然而,需要进一步对智力发育障碍进行长期的社会和教育随访。