gfpt1相关的先天性肌无力综合征误诊为肌病:临床和遗传学的见解。

IF 2 4区 医学 Q3 CLINICAL NEUROLOGY
Fatma Zehra Calikusu, Emel Oguz-Akarsu, Sebnem Ozemri Sag, Sehime G Temel, Hamdi Necdet Karli
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引用次数: 0

摘要

先天性肌无力综合征(CMS)是一种罕见的遗传性疾病,以神经肌肉连接功能障碍为特征,表现为波动性肌肉无力。本研究报告了一名25岁的土耳其裔男性患者,由于谷氨酰胺-果糖- 6-磷酸转氨酶1 (GFPT1)突变而疑似CMS,并伴有涉及其母亲和叔叔的家族性影响。临床、电生理和遗传评估进行了数年,显示进行性无力主要影响肢体近端肌肉。电生理研究显示肌源性受累,反复的神经刺激显示明显的损伤。全外显子组测序显示,患者及其母亲均存在GFPT1纯合子突变。使用乙酰胆碱酯酶抑制剂治疗有适度改善。该研究强调了在波动性虚弱病例中考虑CMS的重要性,强调了诊断挑战和基因检测在家族病例中的作用。进一步的研究需要阐明表型相关性并扩大对cms相关表现的理解。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
GFPT1-related congenital myasthenic syndrome misdiagnosed as myopathy: clinical and genetic insights.

Congenital myasthenic syndrome (CMS) is a rare genetic disorder characterized by neuromuscular junction dysfunction, presenting fluctuating muscle weakness. This study presents a 25-year-old male patient of Turkish origin with suspected CMS due to a glutamine-fructose- 6-phosphate transaminase 1 (GFPT1) mutation, along with familial implications involving his mother and uncle. Clinical, electrophysiologic, and genetic assessments were conducted over several years, revealing progressive weakness predominantly affecting proximal limb muscles. Electrophysiologic studies indicated myogenic involvement, with repetitive nerve stimulation demonstrating significant decrements. Whole-exome sequencing revealed a homozygous GFPT1 mutation in both the patient and his mother. Treatment with acetylcholinesterase inhibitors yielded modest improvement. The study underscores the importance of considering CMS in cases of fluctuating weakness, highlighting diagnostic challenges and the role of genetic testing in familial cases. Further research is warranted to elucidate phenotypic correlations and expand understanding of CMS-associated manifestations.

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来源期刊
Acta neurologica Belgica
Acta neurologica Belgica 医学-临床神经学
CiteScore
4.20
自引率
3.70%
发文量
300
审稿时长
6-12 weeks
期刊介绍: Peer-reviewed and published quarterly, Acta Neurologica Belgicapresents original articles in the clinical and basic neurosciences, and also reports the proceedings and the abstracts of the scientific meetings of the different partner societies. The contents include commentaries, editorials, review articles, case reports, neuro-images of interest, book reviews and letters to the editor. Acta Neurologica Belgica is the official journal of the following national societies: Belgian Neurological Society Belgian Society for Neuroscience Belgian Society of Clinical Neurophysiology Belgian Pediatric Neurology Society Belgian Study Group of Multiple Sclerosis Belgian Stroke Council Belgian Headache Society Belgian Study Group of Neuropathology
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