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Fahr综合征作为伴有可能癫痫发作活动和心脏骤停的线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)综合征的初始影像学特征:一例报告

Fahr's syndrome as the initial imaging characteristics of MELAS syndrome with a possible seizure activity and cardiac arrest: a case report.

作者信息

Zheng Yan, Wu Haohao, Zhang Meng, Huang Baogang, Yang Junsu, Liu Chuan, Wang Hanmin, Du Kang

机构信息

Department of Neurology, Qujing First People's Hospital, Qujing, Yunnan, China.

Teaching and Research Office of Internal Medicine, Qujing Medical College, Qujing, Yunnan, China.

出版信息

Front Genet. 2024 Aug 12;15:1393158. doi: 10.3389/fgene.2024.1393158. eCollection 2024.

DOI:10.3389/fgene.2024.1393158
PMID:39188287
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11345220/
Abstract

This study reported a case of MELAS syndrome presenting as the initial imaging characteristics of Fahr's syndrome with "near" sudden unexpected death in epilepsy (SUDEP) and lateralized periodic discharges (LPD). The patient, a young boy, experienced loss of consciousness 2 days prior, which was followed by two limb and facial convulsions. He was later found in cardiac arrest during hospitalization, but regained consciousness gradually after receiving cardiopulmonary resuscitation and tracheal intubation. The patient exhibited short stature, intellectual disability, poor sports abilities, and academic performance since childhood, but had no family history. Emergency head computed tomography (CT) revealed high density calcification in bilateral caudate nucleus, lentiform nucleus, thalamus, and dentate nucleus with evidence of an acute process. The patient was transferred to the neurology department where he continued to recover consciousness, though he experienced dysarthria, left limb hemiplegia, and hemiparesthesia. Changes in head magnetic resonance imaging (MRI) findings were noted at admission, 1 month later, and 6 months later. LPD were observed in his video electroencephalogram. The CT urography indicated a narrow left ureteropelvic junction with left hydronephrosis, which was suggestive of ureteropelvic junction obstruction. Ultimately, a diagnosis of near-SUDEP was suspected in this patient, indicating a rare case of MELAS syndrome with near-SUDEP and LPD. The gene tests results revealed the presence of the mitochondrial DNA A3243G mutation, leading to the final diagnosis of MELAS syndrome. This case expands the clinical disease spectrum of the MELAS syndrome.

摘要

本研究报告了一例线粒体脑肌病伴乳酸血症和卒中样发作(MELAS)综合征病例,该病例最初表现为 Fahr 综合征的影像学特征,伴有癫痫性猝死(SUDEP)“接近型”及侧化周期性放电(LPD)。患者为一名小男孩,2 天前出现意识丧失,随后出现两次肢体及面部抽搐。住院期间他后来被发现心脏骤停,但在接受心肺复苏和气管插管后逐渐恢复意识。该患者自童年起就身材矮小、智力残疾、运动能力差且学业成绩不佳,但无家族病史。急诊头颅计算机断层扫描(CT)显示双侧尾状核、豆状核、丘脑和齿状核有高密度钙化,并有急性病变的证据。患者被转至神经内科,在那里他继续恢复意识,不过出现了构音障碍、左侧肢体偏瘫和偏身感觉异常。在入院时、1 个月后及 6 个月后记录了头颅磁共振成像(MRI)结果的变化。在他的视频脑电图中观察到了 LPD。CT 尿路造影显示左输尿管肾盂连接处狭窄并伴有左肾积水,提示输尿管肾盂连接处梗阻。最终,怀疑该患者为接近 SUDEP,这表明是一例罕见的伴有接近 SUDEP 和 LPD 的 MELAS 综合征病例。基因检测结果显示存在线粒体 DNA A3243G 突变,从而最终诊断为 MELAS 综合征。该病例扩展了 MELAS 综合征的临床疾病谱。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7c28/11345220/9a5b52e71b39/fgene-15-1393158-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7c28/11345220/43d16c934d1d/fgene-15-1393158-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7c28/11345220/9a5b52e71b39/fgene-15-1393158-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7c28/11345220/43d16c934d1d/fgene-15-1393158-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7c28/11345220/9a5b52e71b39/fgene-15-1393158-g002.jpg

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本文引用的文献

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Acute myocardial infarction in a patient with MELAS syndrome: a possible link?线粒体脑肌病伴高乳酸血症和卒中样发作(MELAS)综合征患者发生急性心肌梗死:是否存在关联?
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