HCRT的双等位基因变异导致常染色体隐性嗜睡症。

IF 1.6 4区 医学 Q3 CLINICAL NEUROLOGY Neurogenetics Pub Date : 2024-04-01 Epub Date: 2024-01-19 DOI:10.1007/s10048-024-00744-0
Wejdan Hakami, Farah Thabet, Amal Alhashem, Abdulaziz Alghamdi, Saad Alshahwan, Fowzan S Alkuraya, Brahim Tabarki
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引用次数: 0

摘要

伴有惊厥的嗜睡症是一种复杂的疾病,既有遗传因素,也有环境风险因素。为了进一步了解与同基因 HCRT 相关的嗜睡症,我们对来自两个近亲结婚家庭的五名患者进行了病例系列研究,每个家庭都有一个新的 HCRT c.17_18del 同基因变异体。所有患者在婴儿期都表现出严重的惊厥,并伴有嗜睡症症状。此外,随着时间的推移,大多数患者的惊厥症状都有所改善或消失。HCRT 的致病变异会导致常染色体隐性嗜睡症伴惊厥。应针对特定的嗜睡症亚组,尤其是那些早发、家族性病例和以惊厥表型为主的嗜睡症患者,进行 HCRT 基因的基因检测。
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Bi-allelic variants in HCRT cause autosomal recessive narcolepsy.

Narcolepsy with cataplexy is a complex disease with both genetic and environmental risk factors. To gain further insight into the homozygous HCRT-related narcolepsy, we present a case series of five patients from two consanguineous families, each harboring a novel homozygous variant of HCRT c.17_18del. All affected individuals exhibited severe cataplexy accompanied by narcolepsy symptoms during infancy. Additionally, cataplexy symptoms improved or disappeared in the majority of patients over time. Pathogenic variants in HCRT cause autosomal recessive narcolepsy with cataplexy. Genetic testing of the HCRT gene should be conducted in specific subgroups of narcolepsy, particularly those with early onset, familial cases, and a predominantly cataplexy phenotype.

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来源期刊
Neurogenetics
Neurogenetics 医学-临床神经学
CiteScore
3.90
自引率
0.00%
发文量
24
审稿时长
6 months
期刊介绍: Neurogenetics publishes findings that contribute to a better understanding of the genetic basis of normal and abnormal function of the nervous system. Neurogenetic disorders are the main focus of the journal. Neurogenetics therefore includes findings in humans and other organisms that help understand neurological disease mechanisms and publishes papers from many different fields such as biophysics, cell biology, human genetics, neuroanatomy, neurochemistry, neurology, neuropathology, neurosurgery and psychiatry. All papers submitted to Neurogenetics should be of sufficient immediate importance to justify urgent publication. They should present new scientific results. Data merely confirming previously published findings are not acceptable.
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