Durner M, Zhou G, Fu D, Abreu P, Shinnar S, Resor S R, Moshe S L, Rosenbaum D, Cohen J, Harden C, Kang H, Wallace S, Luciano D, Ballaban-Gil K, Klotz I, Dicker E, Greenberg D A
Departments of Psychiatry,Mount Sinai Medical Center, 1 Gustave Levy Place, New York, NY l0029, USA.
Am J Hum Genet. 1999 May;64(5):1411-9. doi: 10.1086/302371.
Several loci and candidate genes for epilepsies or epileptic syndromes map or have been suggested to map to chromosome 8. We investigated families with adolescent-onset idiopathic generalized epilepsy (IGE), for linkage to markers spanning chromosome 8. The IGEs that we studied included juvenile myoclonic epilepsy (JME), epilepsy with only generalized tonic-clonic seizures occurring either randomly during the day (random grand mal) or on awakening (awakening grand mal), and juvenile absence epilepsy (JAE). We looked for a gene common to all these IGEs, but we also investigated linkage to specific subforms of IGE. We found evidence for linkage to chromosome 8 in adolescent-onset IGE families in which JME was not present. The maximum multipoint LOD score was 3.24 when family members with IGE or generalized spike-and-waves (SW) were considered affected. The LOD score remained very similar (3.18) when clinically normal family members with SW were not considered to be affected. Families with either pure grand mal epilepsy or absence epilepsy contributed equally to the positive LOD score. The area where the LOD score reaches the maximum encompasses the location of the gene for the beta3-subunit of the nicotinic acetylcholine receptor (CHRNB3), thus making this gene a possible candidate for these specific forms of adolescent-onset IGE. The data excluded linkage of JME to this region. These results indicate genetic heterogeneity within IGE and provide no evidence, on chromosome 8, for a gene common to all IGEs.
癫痫或癫痫综合征的几个基因座和候选基因已定位于或被认为定位于8号染色体。我们对青少年特发性全身性癫痫(IGE)家系进行了研究,以确定与跨越8号染色体的标记的连锁关系。我们研究的IGE包括青少年肌阵挛性癫痫(JME)、仅在白天随机发作(随机大发作)或觉醒时发作(觉醒大发作)的全身性强直阵挛发作癫痫,以及青少年失神癫痫(JAE)。我们寻找所有这些IGE共有的基因,但也研究了与IGE特定亚型的连锁关系。我们在不存在JME的青少年起病IGE家系中发现了与8号染色体连锁的证据。当将患有IGE或全身性棘波和慢波(SW)的家庭成员视为患病时,最大多点对数优势(LOD)分数为3.24。当不将具有SW的临床正常家庭成员视为患病时,LOD分数保持非常相似(3.18)。纯大发作癫痫或失神癫痫家系对阳性LOD分数的贡献相同。LOD分数达到最大值的区域包含烟碱型乙酰胆碱受体β3亚基(CHRNB3)基因的位置,因此使该基因成为这些特定形式的青少年起病IGE的可能候选基因。数据排除了JME与该区域的连锁关系。这些结果表明IGE内存在遗传异质性,并且在8号染色体上没有证据表明存在所有IGE共有的基因。