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来自18个国家的帕金森病患者中LRRK2基因第41外显子突变的临床遗传学研究。

Clinicogenetic study of mutations in LRRK2 exon 41 in Parkinson's disease patients from 18 countries.

作者信息

Tomiyama Hiroyuki, Li Yuanzhe, Funayama Manabu, Hasegawa Kazuko, Yoshino Hiroyo, Kubo Shin-Ichiro, Sato Kenichi, Hattori Tatsuya, Lu Chin-Song, Inzelberg Rivka, Djaldetti Ruth, Melamed Eldad, Amouri Rim, Gouider-Khouja Neziha, Hentati Faycal, Hatano Yasuko, Wang Mei, Imamichi Yoko, Mizoguchi Koichi, Miyajima Hiroaki, Obata Fumiya, Toda Tatsushi, Farrer Matthew J, Mizuno Yoshikuni, Hattori Nobutaka

机构信息

Department of Neurology, Juntendo University School of Medicine, Tokyo, Japan.

出版信息

Mov Disord. 2006 Aug;21(8):1102-8. doi: 10.1002/mds.20886.

Abstract

We screened LRRK2 mutations in exon 41 in 904 parkin-negative Parkinson's disease (PD) patients (868 probands) from 18 countries across 5 continents. We found three heterozygous missense (novel I2012T, G2019S, and I2020T) mutations in LRRK2 exon 41. We identified 11 (1.3%) among 868 PD probands, including 2 sporadic cases and 8 (6.2%) of 130 autosomal dominant PD families. The LRRK2 mutations in exon 41 exhibited relatively common and worldwide distribution. Among the three mutations in exon 41, it has been reported that Caucasian patients with G2019S mutation have a single-founder effect. In the present study, Japanese patients with G2019S were unlikely to have a single founder from the Caucasian patients. In contrast, I2020T mutation has a single-founder effect in Japanese patients. Clinically, patients with LRRK2 mutations had typical idiopathic PD. Notably, several patients developed dementia and psychosis, and one with I2020T had low cardiac (123)I-metaiodobenzylguanidine (MIBG) heart/mediastinum ratio, although the ratio was not low in other patients with I2020T or G2019S. Clinical phenotypes including psychosis, dementia, and MIBG ratios are also heterogeneous, similar to neuropathology, in PD associated with LRRK2 mutations.

摘要

我们对来自五大洲18个国家的904例无帕金森病蛋白(parkin)突变的帕金森病(PD)患者(868名先证者)进行了LRRK2基因第41外显子突变筛查。我们在LRRK2基因第41外显子中发现了3个杂合错义突变(新的I2012T、G2019S和I2020T)。我们在868例PD先证者中鉴定出11例(1.3%),包括2例散发病例和130个常染色体显性PD家族中的8例(6.2%)。LRRK2基因第41外显子中的突变呈现出相对常见且在全球分布的特点。在第41外显子的3个突变中,据报道携带G2019S突变的白种人患者存在单一奠基者效应。在本研究中,携带G2019S突变的日本患者不太可能有来自白种人的单一奠基者。相反,I2020T突变在日本患者中有单一奠基者效应。临床上,携带LRRK2突变的患者具有典型的特发性PD。值得注意的是,有几名患者出现了痴呆和精神病症状,1例携带I2020T突变的患者心脏(¹²³)I-间碘苄胍(MIBG)心/纵隔比值较低,不过其他携带I2020T或G2019S突变的患者该比值并不低。与LRRK2突变相关的PD中,包括精神病、痴呆和MIBG比值在内的临床表型也具有异质性,类似于神经病理学表现。

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