Novel duplication mutation in the patatin domain of adipose triglyceride lipase (PNPLA2) in neutral lipid storage disease with severe myopathy

被引:71
作者
Akiyama, Masashi
Sakai, Kaori
Ogawa, Masaya
McMillan, James R.
Sawamura, Daisuke
Shimizu, Hiroshi
机构
[1] Hokkaido Univ, Grad Sch Med, Dept Dermatol, Sapporo, Hokkaido 060, Japan
[2] Aomori Prefect Cent Hosp, Dept Neurol, Hirosaki, Aomori, Japan
[3] Hirosaki Univ, Grad Sch Med, Dept Dermatol, Hirosaki, Aomori, Japan
关键词
ATGL; myopathy; patatin; PNPLA2;
D O I
10.1002/mus.20869
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Recently, mutations in PNPLA2 encoding adipose triglyceride lipase (ATGL) were reported to underlie a neutral lipid storage disease (NLSD) subgroup characterized by mild myopathy and the absence of ichthyosis. In the present study a novel homozygous PNPLA2 mutation c.475_478dupCTCC (p.Gln160ProfsX19) in the patatin domain, the ATGL active site, was detected in a woman with NLSD and severe myopathy. The present results suggest that a premature truncation mutation in the patatin domain causes NLSD with severe myopathy.
引用
收藏
页码:856 / 859
页数:4
相关论文
共 16 条
[1]   Truncation of CGI-58 protein causes malformation of lamellar granules resulting in ichthyosis in Dorfman-Chanarin syndrome [J].
Akiyama, M ;
Sawamura, D ;
Nomura, Y ;
Sugawara, M ;
Shimizu, H .
JOURNAL OF INVESTIGATIVE DERMATOLOGY, 2003, 121 (05) :1029-1034
[2]   NEUTRAL LIPID STORAGE DISEASE - NEW DISORDER OF LIPID-METABOLISM [J].
CHANARIN, I ;
PATEL, A ;
SLAVIN, G ;
WILLS, EJ ;
ANDREWS, TM ;
STEWART, G .
BRITISH MEDICAL JOURNAL, 1975, 1 (5957) :553-555
[3]   ICHTHYOSIFORM DERMATOSIS WITH SYSTEMIC LIPIDOSIS [J].
DORFMAN, ML ;
HERSHKO, C ;
EISENBERG, S ;
SAGHER, F .
ARCHIVES OF DERMATOLOGY, 1974, 110 (02) :261-266
[4]   The gene encoding adipose triglyceride lipase (PNPLA2) is mutated in neutral lipid storage disease with myopathy [J].
Fischer, Judith ;
Lefevre, Caroline ;
Morava, Eva ;
Mussini, Jean-Marie ;
Laforet, Pascal ;
Negre-Salvayre, Anne ;
Lathrop, Mark ;
Salvayre, Robert .
NATURE GENETICS, 2007, 39 (01) :28-30
[5]   Brummer lipase is an evolutionary conserved fat storage regulator in Drosophila [J].
Grönke, S ;
Mildner, A ;
Fellert, S ;
Tennagels, N ;
Petry, S ;
Müller, G ;
Jäckle, H ;
Kühnlein, RP .
CELL METABOLISM, 2005, 1 (05) :323-330
[6]   Defective lipolysis and altered energy metabolism in mice lacking adipose triglyceride lipase [J].
Haemmerle, G ;
Lass, A ;
Zimmermann, R ;
Gorkiewicz, G ;
Meyer, C ;
Rozman, J ;
Heldmaier, G ;
Maier, R ;
Theussl, C ;
Eder, S ;
Kratky, D ;
Wagner, EF ;
Klingenspor, M ;
Hoefler, G ;
Zechner, R .
SCIENCE, 2006, 312 (5774) :734-737
[7]   Identification, cloning, expression, and purification of three novel human calcium-independent phospholipase A2 family members possessing triacylglycerol lipase and acylglycerol transacylase activities [J].
Jenkins, CM ;
Mancuso, DJ ;
Yan, W ;
Sims, HF ;
Gibson, B ;
Gross, RW .
JOURNAL OF BIOLOGICAL CHEMISTRY, 2004, 279 (47) :48968-48975
[8]   Obese yeast: Triglyceride lipolysis is functionally conserved from mammals to yeast [J].
Kurat, CF ;
Natter, K ;
Petschnigg, J ;
Wolinski, H ;
Scheuringer, K ;
Scholz, H ;
Zimmermann, R ;
Leber, R ;
Zechner, R ;
Kohlwein, SD .
JOURNAL OF BIOLOGICAL CHEMISTRY, 2006, 281 (01) :491-500
[9]   Expression, regulation, and triglyceride hydrolase activity of Adiponutrin family members [J].
Lake, AC ;
Sun, Y ;
Li, JL ;
Kim, JE ;
Johnson, JW ;
Li, DM ;
Revett, T ;
Shih, HH ;
Liu, W ;
Paulsen, JE ;
Gimeno, RE .
JOURNAL OF LIPID RESEARCH, 2005, 46 (11) :2477-2487
[10]   Adipose triglyceridelipase-mediated lipolysis of cellular fat stores is activated by CGI-58 and defective in Chanarin-Dorfman Syndrome [J].
Lass, Achim ;
Zimmermann, Robert ;
Haemmerle, Guenter ;
Riederer, Monika ;
Schoiswohl, Gabriele ;
Schweiger, Martina ;
Kienesberger, Petra ;
Strauss, Juliane G. ;
Gorkiewicz, Gregor ;
Zechner, Rudolf .
CELL METABOLISM, 2006, 3 (05) :309-319