Literature DB >> 22008442

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

Naoki Nakagawa1, Hiroki Maruyama, Takayuki Ishihara, Utako Seino, Jun-ichi Kawabe, Fumihiko Takahashi, Motoi Kobayashi, Atsushi Yamauchi, Yukie Sasaki, Naka Sakamoto, Hisanobu Ota, Yasuko Tanabe, Toshiharu Takeuchi, Toshihiro Takenaka, Kenjiro Kikuchi, Naoyuki Hasebe.   

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.

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Year:  2011        PMID: 22008442     DOI: 10.1536/ihj.52.308

Source DB:  PubMed          Journal:  Int Heart J        ISSN: 1349-2365            Impact factor:   1.862


  5 in total

1.  Non benign neonatal arrhythmias observed in a tertiary neonatal intensive care unit.

Authors:  Ahmet Afşin Kundak; Dilek Dilli; Belma Karagöl; Nilgün Karadağ; Ayşegül Zenciroğlu; Nurullah Okumuş; Vehbi Doğan; Nuran Uzunalıç
Journal:  Indian J Pediatr       Date:  2012-10-04       Impact factor: 1.967

2.  High-risk screening for Anderson-Fabry disease in patients with cardiac, renal, or neurological manifestations.

Authors:  Naoki Nakagawa; Jun Sawada; Naka Sakamoto; Toshiharu Takeuchi; Fumihiko Takahashi; Jun-Ich Maruyama; Ken Momosaki; Kimitoshi Nakamura; Fumio Endo; Naoyuki Hasebe
Journal:  J Hum Genet       Date:  2019-06-19       Impact factor: 3.172

3.  Unexpectedly high renal pathological scores of two female siblings with Fabry disease presenting with urinary mulberry cells without microalbuminuria.

Authors:  Natsuo Yamada; Hirofumi Sakuma; Mitsuru Yanai; Ayana Suzuki; Keisuke Maruyama; Motoki Matsuki; Naoki Nakagawa
Journal:  Mol Genet Metab Rep       Date:  2022-04-22

4.  Ventricular fibrillation associated with vasospastic angina pectoris in Fabry disease: a case report.

Authors:  Kenji Kodama; Tomoya Ozawa; Kenichi Dochi; Yoshiki Ueno
Journal:  Eur Heart J Case Rep       Date:  2019-10-21

5.  Characteristics of Neurological Symptoms in Adult Japanese Patients with Fabry Disease.

Authors:  Jun Sawada; Naoki Nakagawa; Kohei Kano; Tsukasa Saito; Takayuki Katayama; Takaaki Sawada; Ken Momosaki; Kimitoshi Nakamura; Naoyuki Hasebe
Journal:  Intern Med       Date:  2021-01-15       Impact factor: 1.271

  5 in total

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