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伪装成进行性肌阵挛癫痫的青少年型亨廷顿舞蹈病。

Juvenile Huntington's disease masquerading as progressive myoclonus epilepsy.

作者信息

Thakor Bina, Jagtap Sujit A, Joshi Aniruddha

机构信息

Department of Paediatric Neurology, Bharati Vidyapeeth Medical College, Pune, India.

Bharati Vidyapeeth Medical College, Pune, India.

出版信息

Epilepsy Behav Rep. 2021 Jul 15;16:100470. doi: 10.1016/j.ebr.2021.100470. eCollection 2021.

Abstract

Juvenile Huntington's disease (JHD) has an onset before 20 years of age, and is characterized by behavioural issues, epilepsy, rigidity, bradykinesia and dystonia. It contributes to 0.5-5% of all Huntington disease (HD) cases. JHD demonstrates a more rapid progression and is characterised by dystonia, as opposed to the slow progression with predominant chorea seen in adult-onset HD. Seizures are described in 38% of JHD as compared to 2% in the adult onset HD. The different types of seizures reported in JHD are generalized seizures, myoclonus, absence seizures and less commonly tonic and focal seizures with impaired awareness. JHD patients have good seizure control initially and develop drug-resistant epilepsy in the later stages of the disease which is rarely reported. Here, we report the case of a 13 -year-old boy, who initially presented with generalized tonic-clonic seizures followed by myoclonic jerks, with subsequent cognitive decline, ataxia, involuntary movements and drug resistant epilepsy mimicking a progressive myoclonus sepilepsy. His EEG changed from normal background with generalized interictal epileptiform discharges to diffuse slowing with fast activity devoid of epileptiform activity to reflect electroclinical evolution of the disease process.

摘要

青少年型亨廷顿舞蹈病(JHD)在20岁之前发病,其特征为行为问题、癫痫、僵硬、运动迟缓及肌张力障碍。它占所有亨廷顿舞蹈病(HD)病例的0.5%-5%。与成人型HD中以舞蹈症为主的缓慢进展不同,JHD进展更快,以肌张力障碍为特征。JHD患者中38%有癫痫发作,而成人型HD患者中这一比例为2%。JHD中报告的不同类型癫痫发作包括全身性发作、肌阵挛、失神发作,较少见的有强直发作及意识障碍的局灶性发作。JHD患者最初癫痫控制良好,在疾病后期发展为药物难治性癫痫,这种情况鲜有报道。在此,我们报告一例13岁男孩的病例,他最初表现为全身性强直阵挛发作,随后出现肌阵挛抽搐,继而出现认知功能下降、共济失调、不自主运动及药物难治性癫痫,类似进行性肌阵挛癫痫。他的脑电图从背景正常伴全身性发作间期癫痫样放电,变为弥漫性慢波伴快速活动且无癫痫样活动,以反映疾病过程的电临床演变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6573/8327331/31d0063d5e19/gr1.jpg

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