Anushree Akansha, Kumar Sudesh, Bhattacharya Piyali, Tripathi Siddharth, Chattopadhyay Nandita
MGM Medical College and Hospital, Kishanganj, Bihar, India.
Glob Pediatr Health. 2023 Mar 25;10:2333794X231163418. doi: 10.1177/2333794X231163418. eCollection 2023.
Wilson disease is a rare autosomal recessive genetic disease, caused by the mutation of the ATP7B gene leading to decreased secretion of serum ceruloplasmin in blood and decrease biliary excretion of copper leading to toxic level accumulation in the liver, brain, kidney, and cornea, resulting in development of characteristic liver disease and neuropsychiatric symptoms. Our case presented with mainly clumsiness and gait abnormality without any psychiatric component and any history of liver disease. A 13-year old male, born out of non-consanguineous marriage, presented with clumsy walking and slurring of speech. The child also complained of poor handwriting and slipping of slipper from foot, without any history of abnormal behavior and poor scholastic performance. On examination gait was abnormal with sidewise swaying, increased muscle tone with rigidity and bilateral flexor plantar reflex. Slit lamp examination of eyes revealed bilateral Kayser-Fleischer rings. Serum ceruloplasmin was low (0.03 g/L) and 24-hour urinary copper was high (119.64 μg/day). MRI brain showed B/L putamen hyperintensity and panda sign suggestive of Wilson disease. After the diagnosis of Wilson disease was made, patient was treated with penicillamine and zinc. Child was also followed-up and re-examination showed slight improvement. Though not rare, Wilson disease is an uncommon entity with varied presentations and disabling consequences. Hence high index of suspicion and clinical correlation is required to diagnose it. Early initiation of treatment and good compliance ensure a better outcome.
威尔逊病是一种罕见的常染色体隐性遗传病,由ATP7B基因突变引起,导致血液中血清铜蓝蛋白分泌减少以及胆汁铜排泄减少,从而使肝脏、大脑、肾脏和角膜中铜蓄积至中毒水平,导致特征性肝病和神经精神症状的出现。我们的病例主要表现为笨拙和步态异常,无任何精神症状及肝病病史。一名13岁男性,非近亲结婚出生,表现为行走笨拙和言语不清。该患儿还主诉书写困难和拖鞋从脚上滑落,无任何异常行为和学业成绩不佳的病史。检查发现步态异常,有侧向摇摆,肌张力增加伴强直以及双侧跖屈反射。裂隙灯检查双眼可见双侧凯-弗环。血清铜蓝蛋白水平低(0.03g/L),24小时尿铜高(119.64μg/天)。脑部MRI显示双侧壳核高信号及“熊猫征”,提示威尔逊病。威尔逊病诊断明确后,患者接受青霉胺和锌治疗。患儿也进行了随访,复查显示有轻微改善。威尔逊病虽不罕见,但却是一种临床表现多样且后果严重的疾病。因此,需要高度的怀疑指数和临床相关性来进行诊断。早期开始治疗并良好依从可确保更好的预后。