Department of Pediatrics, Shifa Tameer-e-Millat University, Shifa College of Medicine, Islamabad, Pakistan.
Department of Pediatrics, Shifa Tameer-e-Millat University, Shifa College of Medicine, Shifa International Hospital, Islamabad, Pakistan.
Ann Transplant. 2021 Oct 5;26:e932606. doi: 10.12659/AOT.932606.
BACKGROUND Wilson disease (WD) is a rare genetic disorder with vast clinical presentations and a higher incidence in areas where consanguinity is common. Most patients can be treated with oral chelation, but some require advanced surgical intervention, like liver transplantation (LT). This study aims to review outcomes of WD patients presenting to a tertiary care center over a period of 10 years. MATERIAL AND METHODS This retrospective analysis was conducted at Shifa International Hospital, Islamabad, Pakistan. Patients <18 years who were diagnosed with WD per ESPAGHAN guidelines from 2010 to 2020 were included. Presentation, diagnosis, treatment, and LT and its complications were recorded. Follow-ups were recorded, and patients were contacted by phone in cases of interrupted follow-up. Frequencies and percentages of variables were calculated. RESULTS A total of 48 patients with WD were identified. Symptomatic disease was seen in 45 patients, with 3 diagnosed on screening. The hepatic form was common (62.2%). Mean age at diagnosis was 9.74 (range 5-17) years, 28 (58.3%) were male, while 17 (35.4%) were female. Urinary copper was increased in all patients (645.82±528.40). Oral treatment with penicillamine was given to 34 (75.5%) patients; 4 (8.9%) died while on oral treatment. Living donor LT was performed in 11 (22.9%) patients, who had a mean King's Wilson index of 11 (range, 6-14). Currently, all LT patients are alive, with maximum graft survival of 7 years. CONCLUSIONS LT offers a promising treatment with good outcomes in pediatric WD. However, timely diagnosis and management with oral chelation therapy can prolong survival without LT.
威尔逊病(WD)是一种罕见的遗传性疾病,临床表现广泛,在近亲结婚常见的地区发病率较高。大多数患者可以通过口服螯合剂治疗,但有些则需要肝移植(LT)等先进的手术干预。本研究旨在回顾在一个三级护理中心就诊的 WD 患者 10 年来的治疗结果。
这项回顾性分析在巴基斯坦伊斯兰堡的 Shifa 国际医院进行。纳入 2010 年至 2020 年期间根据 ESPAGHAN 指南诊断为 WD 的<18 岁患者。记录了患者的表现、诊断、治疗、LT 及其并发症。记录了随访情况,并在随访中断的情况下通过电话联系患者。计算了变量的频率和百分比。
共发现 48 例 WD 患者。45 例患者有症状性疾病,3 例为筛查发现。肝型较为常见(62.2%)。诊断时的平均年龄为 9.74 岁(范围 5-17 岁),28 例(58.3%)为男性,17 例(35.4%)为女性。所有患者的尿铜均增加(645.82±528.40)。34 例(75.5%)患者给予青霉胺口服治疗;4 例(8.9%)患者在口服治疗期间死亡。11 例(22.9%)患者进行了活体供肝 LT,其 King's Wilson 指数平均为 11(范围 6-14)。目前,所有 LT 患者均存活,最大移植物存活时间为 7 年。
LT 为儿科 WD 提供了一种有前途的治疗方法,且结果良好。然而,及时诊断和管理,结合口服螯合剂治疗,可以延长生存时间,无需 LT。