儿童Hashimoto脑病临床特征分析与随访研究
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摘要
目的探讨儿童Hashimoto脑病(HE)的临床特点。方法回顾分析4例HE患儿的临床资料。结果 4例患儿均为学龄期,3例为女童,既往均体健。临床主要表现为痫性发作3例,精神症状2例,意识障碍2例,卒中样表现、记忆力下降及睡眠减少各1例。4例患儿脑电图背景活动均减慢,3例头颅MRI异常;4例患儿血清甲状腺抗体均明显升高,其中2例临床症状消失后甲状腺抗体恢复正常,另外2例明显降低,但未完全恢复正常;4例患儿中仅1例甲状腺功能异常。4例均对糖皮质激素治疗反应良好,其中1例自行停药后复发,继续原药治疗仍有效。结论儿童HE极少见,当出现其他疾病难以解释的痫性发作、精神异常、认知损害、运动障碍、意识障碍等时,应考虑到HE,血清甲状腺抗体增高为诊断必要条件。
Objective To explore the clinical characteristics of Hashimoto encephalopathy(HE) in children. Methods The clinical data of 4 children with HE were analyzed retrospectively. Results All the 4 cases were school-age children and 3 of them were girls. They were physically healthy before onset. The main clinical manifestations were epileptic seizures in 3 cases, mental symptoms in 2 cases, disturbance of consciousness in 2 cases, stroke like symptoms in 1 case, decreased memory and decreased sleep in 1 case. Electroencephalogram showed that the background activity was decreased in 4 cases, and MRI showed abnormal in 3 cases. Serum thyroid antibodies were significantly increased in 4 cases, and were returned to normal in 2 cases when clinical symptoms disappeared, while they were significantly reduced, but not completely back to normal in another 2 cases. Only one out of 4 cases had abnormal thyroid function. All the 4 cases responded well to corticosteroid therapy. One of them relapsed after discontinuation of the therapy, but it was still effective when the therapy was reassumed. Conclusions HE is rare in children. When there are manifestations of unknown cause, such as epileptic seizures, mental disorders, cognitive impairment, movement disorders and disturbance of consciousness, HE should be considered. In addition, the increase of serum thyroid antibody should be considered as a necessary condition for diagnosis.
Objective To explore the clinical characteristics of Hashimoto encephalopathy(HE) in children. Methods The clinical data of 4 children with HE were analyzed retrospectively. Results All the 4 cases were school-age children and 3 of them were girls. They were physically healthy before onset. The main clinical manifestations were epileptic seizures in 3 cases, mental symptoms in 2 cases, disturbance of consciousness in 2 cases, stroke like symptoms in 1 case, decreased memory and decreased sleep in 1 case. Electroencephalogram showed that the background activity was decreased in 4 cases, and MRI showed abnormal in 3 cases. Serum thyroid antibodies were significantly increased in 4 cases, and were returned to normal in 2 cases when clinical symptoms disappeared, while they were significantly reduced, but not completely back to normal in another 2 cases. Only one out of 4 cases had abnormal thyroid function. All the 4 cases responded well to corticosteroid therapy. One of them relapsed after discontinuation of the therapy, but it was still effective when the therapy was reassumed. Conclusions HE is rare in children. When there are manifestations of unknown cause, such as epileptic seizures, mental disorders, cognitive impairment, movement disorders and disturbance of consciousness, HE should be considered. In addition, the increase of serum thyroid antibody should be considered as a necessary condition for diagnosis.
引文
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[14]de Holanda NC,de Lima DD,Cavalcanti TB,et al.Hashimoto's encephalopathy:systematic review of the literature and an additional case[J].J Neuropsychiatry Clin Neurosci,2011,23(4):384-390.
[15]Ferracci F,Moretto G,Candeago RM,et al.Antithyroid antibodies in the CSF:their role in the pathogenes is of Hashimoto’s encephalopathy[J].Neurology,2003,60(4):712-714.
[16]F e r r a c c i F,B e r t i a t o G,M o r e t t o G.H a s h i m o t o’s encephalopathy:epidemiologic data and pathogenetic considerations[J].J Neurol Sci,2004,217(2):165-168.
[17]Koros C,Economou A,Mastorakos G,et al.A selective memory deficit caused by autoimmune encephalopathy associated with Hashimoto thyroiditis[J].Cogn Behav Neurol,2012,25(3):144-148.
[18]Montagna G,Imperiali M,Agazzi P,et al.Hashimoto's encephalopathy:a rare proteiform disorder[J].Autoimmun Rev,2016,15(5):466-476.
[19]Canelo-Aybar C,Loja-Oropeza D,Cuadra-Urteaga J,et al.Hashimoto's encephalopathy presenting with neurocognitive symptoms:a case report[J].J Med Case Rep,2010,4:337.
[20]Byrne OC,Zuberi SM,Madigan CA,et al.Hashimoto's thyroiditis--a rare but treatable cause of encephalopathy in children[J].Eur J Paediatr Neurol,2000,4(6):279-282.
[21]Berger I,Castiel Y,Dor T.Paediatric Hashimoto encephalopathy,refractory epilepsy and immunoglobulin treatment-unusual case report and review of the literature[J].Acta Paediatr,2010,99(12):1903-1905.
[22]Olmez I,Moses H,Sriram S,et al.Diagnostic and therapeutic aspects of Hashimoto's encephalopathy[J].J Neurol Sci,2013,331(1-2):67-71.
[2]Mocellin R,Walterfang M,Velakoulis D.Hashimoto’s encephalopathy:epidemiology,pathogenesis and management[J].CNS Drugs,2007,21(10):799-811.
[3]Yu HJ,Lee J,Seo DW,et al.Clinical manifestations and treatment response of steroid in pediatric Hashimoto encephalopathy[J].J Child Neurol,2014,29(7):938-942.
[4]Bektas?,Y?lmaz A,Kendirli T,et al.Hashimoto encephalopathy causing drug-resistant status epilepticus treated with plasmapheresis[J].Pediatr Neurol,2012,46(2):132-135.
[5]Miya K,Takahashi Y,Mori H.Anti-NMDAR autoimmune encephalitis[J].Brain Dev,2014,36(8):645-652.
[6]Mamoudjy N,Korff C,Maurey H,et al.Hashimoto’s encephalopathy:identification and long-term outcome in children[J].Eur J Paediatr Neurol,2013,17(3):280-287.
[7]Alink J,de Vries TW.Unexplained seizures,confusion or hallucinations:think Hashimoto encephalopathy[J].Acta Paediatr,2008,97(4):451-453.
[8]Watemberg N,Greenstein D,Levine A.Encephalopathy associated with Hashimoto thyroiditis:pediatric perspective[J].J Child Neurol,2006,21(1):1-5.
[9]Schiess N,Pardo CA.Hashimoto’s encephalopathy[J].Ann N Y Acad Sci,2008,1142:254-265.
[10]Vasconcellos E,Pi?a-Garza JE,Fakhoury T,et al.Pediatric manifestations of Hashimoto's encephalopathy[J].Pediatr Neurol,1999,20(5):394-398.
[11]Monti G,Pugnaghi M,Ariatti A,et al.Non-convulsive status epllepticus of frontal origin as the first manifestation of Hashimoto's encephalopathy[J]Epileptic Disord,2011,13(3):253-258.
[12]Ozbey N.Febrile Hashimoto encephalopathy mimicking thyroid storm[J].Endocrinologist,2002,12:497-499.
[13]Marshall GA,Doyle JJ.Long-term treatment of Hashimoto’s encephalopathy[J].J Neuropsychiatry Clin Neurosci,2006,18(1):14-20.
[14]de Holanda NC,de Lima DD,Cavalcanti TB,et al.Hashimoto's encephalopathy:systematic review of the literature and an additional case[J].J Neuropsychiatry Clin Neurosci,2011,23(4):384-390.
[15]Ferracci F,Moretto G,Candeago RM,et al.Antithyroid antibodies in the CSF:their role in the pathogenes is of Hashimoto’s encephalopathy[J].Neurology,2003,60(4):712-714.
[16]F e r r a c c i F,B e r t i a t o G,M o r e t t o G.H a s h i m o t o’s encephalopathy:epidemiologic data and pathogenetic considerations[J].J Neurol Sci,2004,217(2):165-168.
[17]Koros C,Economou A,Mastorakos G,et al.A selective memory deficit caused by autoimmune encephalopathy associated with Hashimoto thyroiditis[J].Cogn Behav Neurol,2012,25(3):144-148.
[18]Montagna G,Imperiali M,Agazzi P,et al.Hashimoto's encephalopathy:a rare proteiform disorder[J].Autoimmun Rev,2016,15(5):466-476.
[19]Canelo-Aybar C,Loja-Oropeza D,Cuadra-Urteaga J,et al.Hashimoto's encephalopathy presenting with neurocognitive symptoms:a case report[J].J Med Case Rep,2010,4:337.
[20]Byrne OC,Zuberi SM,Madigan CA,et al.Hashimoto's thyroiditis--a rare but treatable cause of encephalopathy in children[J].Eur J Paediatr Neurol,2000,4(6):279-282.
[21]Berger I,Castiel Y,Dor T.Paediatric Hashimoto encephalopathy,refractory epilepsy and immunoglobulin treatment-unusual case report and review of the literature[J].Acta Paediatr,2010,99(12):1903-1905.
[22]Olmez I,Moses H,Sriram S,et al.Diagnostic and therapeutic aspects of Hashimoto's encephalopathy[J].J Neurol Sci,2013,331(1-2):67-71.