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儿童威尔逊病:孟加拉国视角

Childhood Wilson Disease: Bangladesh Perspective.

作者信息

Rukunuzzaman M, Karim A B, Nurullah M, Sultana F, Mazumder M W, Rahman M A, Billah S B, Begum F, Oliullah M

机构信息

Dr Md Rukunuzzaman, Associate Professor, Department of Pediatric Gastroenterology & Nutrition, Bangabandhu Sheikh Mujib Medical University (BSMMU), Dhaka, Bangladesh.

出版信息

Mymensingh Med J. 2017 Apr;26(2):406-413.

Abstract

Wilson's disease (WD) is an autosomal recessive disorder affecting copper metabolism causing copper induced damage to various organs. In children liver is commonly involved. Central nervous system, eyes, RBC, kidneys, brain and bones may also be affected. Aim of the study is to evaluate clinical & laboratory profile of Wilson's disease in children. This cross sectional descriptive study was conducted at the department of Paediatric Gastroenterology and Nutrition, Bangabandhu Sheikh Mujib Medical University (BSMMU), Dhaka, Bangladesh, from January 2011 to December, 2013. One hundred consecutive children of WD between 3 to 18 years of age were evaluated for clinical & biochemical profile. Mean age of studied children was 8.5±1.5 years. Male female ratio was 2:1. Ninety one percent patients were Muslim and nine percent Hindu. Consanguinity of marriage was found in 30% cases. Seven parents were first degree cousin. Family history of chronic liver disease was present in 15% of patients. Most (53%) cases of the hepatic WD presented between 5 to 10 years of age and most of the neurologic WD manifested in 10-15 years age group. Among 100 patients of WD, 69 children presented only with hepatic manifestations, 6 only with neurological manifestations, 14 with both hepatic & neurological manifestation, 10 children was asymptomatic and 1 patient presented with psychiatric features. WD presented as chronic liver disease (CLD) in 42%, CLD with portal hypertension in 34%, acute hepatitis in 20% and fulminant hepatic failure in 4% cases. Stigmata of chronic liver disease were found in 18% patients. Commonest stigmata was thenar and hypothenar wasting (n=8). Keiser- Fleischser ring (K-F ring) was found in 76% of the total patients. K-F ring was present in 84% ( 58 out of 69) of the hepatic only Wilsonian patients and in 90% (18 out of 20) of all neurologic Wilsonian patients. Asymptomatic and psychiatric patient had no K-F ring. About 26% of the WD patients had Coombs negative hemolytic anemia in PBF. Most of the WD patients had altered liver function. Elevated serum transaminase was found in 85% of all cases, prolonged prothrombin time in 59% cases & low serum albumin in 53% cases. Seventy three percent patients had low serum ceruloplasmin, basal urinary copper of >100μgm/day was found in 81% cases and urinary copper following penicillamine challenge of >1200μgm/day was found in 92% cases. In 28 cases with hepatic presentation esophageal varices were identified by upper gastrointestinal endoscopy. WD patient with hepatic presentations were given zinc sulphate along with penicillamine. All patients with neurological manifestation as well as asymptomatic cases were maintained on zinc therapy. WD is a treatable metabolic cause of liver disease. Majority of studied WD children presented with hepatic manifestation of which 76% presented with CLD. Any child presented with jaundice after the age of 3 years should be investigated for WD.

摘要

威尔逊病(WD)是一种常染色体隐性疾病,影响铜代谢,导致铜对多个器官造成损害。在儿童中,肝脏通常受累。中枢神经系统、眼睛、红细胞、肾脏、大脑和骨骼也可能受到影响。本研究的目的是评估儿童威尔逊病的临床和实验室特征。这项横断面描述性研究于2011年1月至2013年12月在孟加拉国达卡的班加班杜·谢赫·穆吉布医科大学(BSMMU)儿科胃肠病学和营养科进行。对100名年龄在3至18岁之间的连续威尔逊病患儿进行了临床和生化特征评估。研究患儿的平均年龄为8.5±1.5岁。男女比例为2:1。91%的患者为穆斯林,9%为印度教徒。30%的病例存在近亲结婚。7对父母是一级表亲。15%的患者有慢性肝病家族史。大多数(53%)肝型威尔逊病病例出现在5至10岁之间,大多数神经型威尔逊病出现在10至15岁年龄组。在100例威尔逊病患者中,69例儿童仅表现为肝脏症状,6例仅表现为神经症状,14例同时有肝脏和神经症状,10例儿童无症状,1例患者表现为精神症状。威尔逊病表现为慢性肝病(CLD)的占42%,伴有门静脉高压的CLD占34%,急性肝炎占20%,暴发性肝衰竭占4%。在18%的患者中发现了慢性肝病的体征。最常见的体征是大鱼际和小鱼际肌萎缩(n = 8)。在所有患者中,76%发现有凯泽-弗莱舍尔环(K-F环)。仅肝脏受累的威尔逊病患者中,84%(69例中的58例)有K-F环,所有神经型威尔逊病患者中90%(20例中的18例)有K-F环。无症状和精神症状患者没有K-F环。约26%的威尔逊病患者外周血涂片中有抗人球蛋白试验阴性的溶血性贫血。大多数威尔逊病患者肝功能异常。85%的病例血清转氨酶升高,59%的病例凝血酶原时间延长,53%的病例血清白蛋白降低。73%的患者血清铜蓝蛋白降低,81%的病例基础尿铜>100μg/天,92%的病例青霉胺激发试验后尿铜>1200μg/天。在28例有肝脏表现的病例中,通过上消化道内镜检查发现了食管静脉曲张。有肝脏表现的威尔逊病患者给予硫酸锌和青霉胺。所有有神经症状的患者以及无症状病例均采用锌治疗。威尔逊病是一种可治疗的肝病代谢病因。大多数研究的威尔逊病儿童表现为肝脏症状,其中76%表现为CLD。任何3岁后出现黄疸的儿童都应进行威尔逊病的检查。

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