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一个患有心脏法布里病的日本家庭的临床和遗传学研究。鉴定出一种新的α-半乳糖苷酶A错义突变(G195V)。

Clinical and genetic investigation of a Japanese family with cardiac fabry disease. Identification of a novel α-galactosidase A missense mutation (G195V).

作者信息

Nakagawa Naoki, Maruyama Hiroki, Ishihara Takayuki, Seino Utako, Kawabe Jun-ichi, Takahashi Fumihiko, Kobayashi Motoi, Yamauchi Atsushi, Sasaki Yukie, Sakamoto Naka, Ota Hisanobu, Tanabe Yasuko, Takeuchi Toshiharu, Takenaka Toshihiro, Kikuchi Kenjiro, Hasebe Naoyuki

机构信息

Division of Cardiology, Nephrology, Pulmonology and Neurology, Department of Internal Medicine, Asahikawa Medical University, Hokkaido, Japan.

出版信息

Int Heart J. 2011;52(5):308-11. doi: 10.1536/ihj.52.308.

DOI:10.1536/ihj.52.308
PMID:22008442
Abstract

Fabry disease is an X-linked lysosomal storage disorder caused by mutations of the α-galactosidase A gene (GLA), and the disease is a relatively prevalent cause of left ventricular hypertrophy mimicking idiopathic hypertrophic cardiomyopathy. We assessed clinically 5 patients of a three-generation family and also searched for GLA mutations in 10 family members. The proband had left ventricular hypertrophy with localized thinning in the basal posterior wall and late gadolinium enhancement (LGE) in the near-circumferential wall in cardiovascular magnetic resonance images and her sister had vasospastic angina pectoris without organic stenosis of the coronary arteries. LGE notably appeared in parallel with decreased α-galactosidase A activity and increased NT-pro BNP in our patients. We detected a new GLA missense mutation (G195V) in exon 4, resulting in a glycine-to-valine substitution. Of the 10 family members, 5 family members each were positive and negative for this mutation. These new data extend our clinical and molecular knowledge of GLA gene mutations and confirm that a novel missense mutation in the GLA gene is important not only for a precise diagnosis of heterozygous status, but also for confirming relatives who are negative for this mutation.

摘要

法布里病是一种由α-半乳糖苷酶A基因(GLA)突变引起的X连锁溶酶体贮积症,该病是导致左心室肥厚的相对常见原因,可酷似特发性肥厚型心肌病。我们对一个三代家庭的5名患者进行了临床评估,并对10名家庭成员进行了GLA突变检测。先证者在心血管磁共振图像中表现为左心室肥厚,基底后壁局部变薄,近圆周壁有延迟钆增强(LGE),她的妹妹患有血管痉挛性心绞痛,冠状动脉无器质性狭窄。在我们的患者中,LGE明显与α-半乳糖苷酶A活性降低和NT-pro BNP升高同时出现。我们在外显子4中检测到一个新的GLA错义突变(G195V),导致甘氨酸被缬氨酸取代。在10名家庭成员中,该突变分别有5名成员呈阳性和阴性。这些新数据扩展了我们对GLA基因突变的临床和分子认识,并证实GLA基因中的一种新错义突变不仅对于精确诊断杂合状态很重要,而且对于确认该突变阴性的亲属也很重要。

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1
Clinical and genetic investigation of a Japanese family with cardiac fabry disease. Identification of a novel α-galactosidase A missense mutation (G195V).一个患有心脏法布里病的日本家庭的临床和遗传学研究。鉴定出一种新的α-半乳糖苷酶A错义突变(G195V)。
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Mol Genet Metab Rep. 2022 Apr 22;31:100874. doi: 10.1016/j.ymgmr.2022.100874. eCollection 2022 Jun.
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Characteristics of Neurological Symptoms in Adult Japanese Patients with Fabry Disease.成年日本法布里病患者的神经症状特征。
Intern Med. 2021 Jun 15;60(12):1819-1826. doi: 10.2169/internalmedicine.6420-20. Epub 2021 Jan 15.
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Ventricular fibrillation associated with vasospastic angina pectoris in Fabry disease: a case report.
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Eur Heart J Case Rep. 2019 Oct 21;3(4):1-5. doi: 10.1093/ehjcr/ytz192. eCollection 2019 Dec.
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High-risk screening for Anderson-Fabry disease in patients with cardiac, renal, or neurological manifestations.对有心脏、肾脏或神经表现的患者进行安德森-法布里病的高危筛查。
J Hum Genet. 2019 Sep;64(9):891-898. doi: 10.1038/s10038-019-0633-1. Epub 2019 Jun 19.
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