نتایج جستجو برای: bscl2 gene

تعداد نتایج: 1141398  

2017
Pooneh Nikuei Kianoosh Malekzadeh Minoo Rajaei Yousef Shafeghati

Congenital generalized lipodystrophies (CGLs) are very rare autosomal recessive disorders which have four types. Of the four CGL types, BSCL2 (Berardinelli–Seip Congenital lipodystrophy type 2) is the result of mutations in the BSCL2/seipin gene. BSCL2 that is the most severe lipodystrophic phenotype is characterized by generalized lipodystrophy, overgrowth, acanthosis nigricans, hepatomegaly, ...

Journal: :The Journal of clinical endocrinology and metabolism 2003
Anil K Agarwal Vinaya Simha Elif Arioglu Oral Stephanie A Moran Phillip Gorden Stephen O'Rahilly Zohra Zaidi Figen Gurakan Silva A Arslanian Aharon Klar Alyne Ricker Neil H White Lutz Bindl Karen Herbst Kurt Kennel Shailesh B Patel Lihadh Al-Gazali Abhimanyu Garg

Congenital generalized lipodystrophy (CGL) is a rare autosomal recessive disorder characterized by near complete absence of adipose tissue from birth. Recently, mutations in 1-acylglycerol-3-phosphate O-acyltransferase 2 (AGPAT2) and Berardinelli-Seip congenital lipodystrophy 2 (BSCL2) genes were reported in pedigrees linked to chromosomes 9q34 and 11q13, respectively. There are limited data re...

Journal: :Diabetes 2008
Victoria A. Payne Neil Grimsey Antoinette Tuthill Sam Virtue Sarah L. Gray Edoardo Dalla Nora Robert K. Semple Stephen O'Rahilly Justin J. Rochford

OBJECTIVE Berardinelli-Seip congenital lipodystrophy type 2 (BSCL2) is a recessive disorder featuring near complete absence of adipose tissue. Remarkably, although the causative gene, BSCL2, has been known for several years, its molecular function and its role in adipose tissue development have not been elucidated. Therefore, we examined whether BSCL2 is involved in the regulation of adipocyte ...

Journal: :genetics in the 3rd millennium 0
pooneh nikuei kianoosh malekzadeh minoo rajaei yousef shafeghati

congenital generalized lipodystrophies (cgls) are very rare autosomal recessive disorders which have four types.of the four cgl types, bscl2 (berardinelli–seip congenital lipodystrophy type 2) is the result of mutations in the bscl2/seipingene.bscl2 which is the most severe lipodystrophic phenotype is characterized by generalized lipodystrophy, overgrowth,acanthosisnigricans, hepatomegaly, insu...

2018
Kazushi Minami Shinichi Takahashi Yoshihiro Nihei Koichi Oki Shigeaki Suzuki Daisuke Ito Hiroshi Takashima Norihiro Suzuki

Seipinopathy is an autosomal dominant neurodegenerative disease caused by mutations of the Berardinelli-Seip Congenital Lipodystrophy 2 (BSCL2) gene. We report the first Japanese case of seipinopathy with a heterozygous mutation of p.N88S in the BSCL2 gene. The patient showed bilateral hyperreflexia of the biceps, triceps, brachioradialis, and knee, as well as the pes cavus and distal dominant ...

2013
Weiqin Chen Hongyi Zhou Siyang Liu Cassie J. Fhaner Bethany C. Gross Todd A. Lydic Gavin E. Reid

Mutations in BSCL2 underlie human congenital generalized lipodystrophy type 2 disease. We previously reported that Bscl2 (-/-) mice develop lipodystrophy of white adipose tissue (WAT) due to unbridled lipolysis. The residual epididymal WAT (EWAT) displays a browning phenotype with much smaller lipid droplets (LD) and higher expression of brown adipose tissue marker proteins. Here we used target...

2016
Cheng-Tsung Hsiao Pei-Chien Tsai Chou-Ching Lin Yo-Tsen Liu Yen-Hua Huang Yi-Chu Liao Han-Wei Huang Kon-Ping Lin Bing-Wen Soong Yi-Chung Lee David R Borchelt

BACKGROUND A small group of patients with inherited neuropathy that has been shown to be caused by mutations in the BSCL2 gene. However, little information is available about the role of BSCL2 mutations in inherited neuropathies in Taiwan. METHODOLOGY AND PRINCIPAL FINDINGS Utilizing targeted sequencing, 76 patients with molecularly unassigned Charcot-Marie-Tooth disease type 2 (CMT2) and 8 w...

Journal: :Journal of medical genetics 2002
L Van Maldergem J Magré T E Khallouf T Gedde-Dahl M Delépine O Trygstad E Seemanova T Stephenson C S Albott F Bonnici V R Panz J L Medina P Bogalho F Huet S Savasta A Verloes J J Robert H Loret M De Kerdanet N Tubiana-Rufi A Mégarbané J Maassen M Polak D Lacombe C R Kahn E L Silveira F H D'Abronzo F Grigorescu M Lathrop J Capeau S O'Rahilly

Generalised lipodystrophy of the Berardinelli-Seip type (BSCL) is a rare autosomal recessive human disorder with severe adverse metabolic consequences. A gene on chromosome 9 (BSCL1) has recently been identified, predominantly in African-American families. More recently, mutations in a previously undescribed gene of unknown function (BSCL2) on chromosome 11, termed seipin, have been found to be...

Journal: :Human molecular genetics 2011
Xin Cui Yuhui Wang Yin Tang Yixiao Liu Liping Zhao Jingna Deng Guoheng Xu Xingui Peng Shenghong Ju George Liu Hongyuan Yang

Berardinelli-Seip congenital lipodystrophy type 2 (BSCL2) is an autosomal recessive disorder characterized by an almost complete loss of adipose tissue, insulin resistance and fatty liver. Here, we create the first murine model of BSCL2 by targeted disruption of seipin, the causative gene for BSCL2. Compared with their wild-type littermates, the seipin(-/-) mice are viable and of normal weight ...

Journal: :American journal of physiology. Endocrinology and metabolism 2012
Xin Cui Yuhui Wang Lingjun Meng Weihua Fei Jingna Deng Guoheng Xu Xingui Peng Shenghong Ju Ling Zhang George Liu Liping Zhao Hongyuan Yang

Berardinelli-Seip congenital lipodystrophy type 2 (BSCL2) is a recessive disorder characterized by an almost complete loss of adipose tissue, insulin resistance, and fatty liver. BSCL2 is caused by loss-of-function mutations in the BSCL2/seipin gene, which encodes seipin. The essential role for seipin in adipogenesis has recently been established both in vitro and in vivo. However, seipin is hi...

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