儿童人群中GAMT缺乏:临床和分子特征及管理。

Child neurology open Pub Date : 2023-11-15 eCollection Date: 2023-01-01 DOI:10.1177/2329048X231215630
Majdah A Almaghrabi, Osama Y Muthaffar, Sereen A Alahmadi, Mashael A Abdulsbhan, Mashael Bamusa, Maram Ahmed Aljezani, Sarah Y Bahowarth, Anas S Alyazidi, Waheeb S Aggad
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引用次数: 0

摘要

目的:综合文献资料,分析实际应用中的治疗方法。方法:回顾性分析52例GAMT缺乏症患者,其中4例来自沙特阿拉伯,采用全外显子组测序诊断。所有数据均采用折线图和图解的形式进行图形表示。结果:患者平均年龄117个月(±29.03),年龄范围12 ~ 372个月。平均发病年龄28.32个月(±13.68)(8天- 252个月)。最常见的症状是发育迟缓,主要是语言和运动、癫痫发作和智力残疾。男女比例为3:1。采用多种治疗方法,54种药物干预,丙戊酸是最常见的。一水肌酐是流行的饮食干预,有25例患者报告改善。结论:该研究表明,通过适当的饮食干预进行有效的治疗可以改善患者的健康状况,并强调个性化的治疗方案对于治疗这种疾病至关重要。
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GAMT Deficiency Among Pediatric Population: Clinical and Molecular Characteristics and Management.

Objective: Analyze the treatment modalities used in real practice by synthesizing available literature. Methods: We reviewed and evaluated 52 cases of GAMT deficiency including 4 novel cases from Saudi Arabia diagnosed using whole-exome sequencing. All data utilized graphical presentation in the form of line charts and illustrated graphs. Results: The mean current age of was 117 months (±29.03) (range 12-372 months). The mean age of disease onset was 28.32 months (±13.68) (range 8 days - 252 months). The most prevalent symptom was developmental delays, mainly speech and motor, seizures, and intellectual disability. The male-to-female ratio was 3:1. Multiple treatments were used, with 54 pharmacological interventions, valproic acid being the most common. Creatinine monohydrate was the prevalent dietary intervention, with 25 patients reporting an improvement. Conclusion: The study suggests that efficient treatment with appropriate dietary intervention can improve patients' health, stressing that personalized treatment programs are essential in managing this disorder.

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