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PMID: 8845841 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Cloning and characterization of the human homologue of a dystrophin related phosphoprotein found at the Torpedo electric organ post-synaptic membrane.

Human molecular genetics ·Vol. 5 ·No. 4 ·1996-04-00 ·Pages 489-96

Sadoulet-Puccio HM, Khurana TS, Cohen JB, Kunkel LM

Abstract

Dystrophin is the protein product which is absent in Duchenne muscular dystrophy (DMD). In mammalian skeletal muscle, dystrophin is found in association with several integral and peripheral membrane proteins, forming a complex known as the dystrophin glycoprotein complex (DGC). In an expressed sequence tag (EST) database search to identify new dystrophin related genes, we isolated EST00891 which showed 57% homology to the cysteine-rich domain of dystrophin and localized to 18q12.1-12.2. This EST is also highly homologous (90%) to the Torpedo californica post-synaptic 87 kDa phosphoprotein. Screening human adult brain and skeletal muscle cDNA libraries with this EST resulted in cloning multiple cDNAs which encode several splice forms all homologous to the C-terminal domain of dystrophin. The largest open reading frame isolated shows 94% homology (86% identity) to the Torpedo 87 kDa protein and 50% homology to the cysteine-rich and carboxy-terminal domains of dystrophin. The other cDNAs isolated encode smaller splice forms of this gene which we have named dystrobrevin. The tissue distribution of dystrobrevin mRNA shows five distinct transcripts which are preferentially expressed between different tissues. In addition, antibodies against either the Torpedo 87 kDa protein or human dystrobrevin demonstrate that at least three of the splice forms are translated as proteins in human brain tissue extracts.

MeSH Terms
Alternative Splicing Amino Acid Sequence Animals Base Sequence Cloning, Molecular DNA, Complementary Dystrophin/genetics,metabolism Dystrophin-Associated Proteins Electric Organ/metabolism Humans Molecular Sequence Data Neuropeptides/genetics,metabolism Phosphoproteins/genetics,metabolism RNA Sequence Homology, Amino Acid Synaptic Membranes/metabolism Torpedo/metabolism
Chemicals
DNA, Complementary DTNA protein, human Dystrophin Dystrophin-Associated Proteins Neuropeptides Phosphoproteins dystrobrevin RNA
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Sadoulet-Puccio H M
Department of Genetics, Harvard Medical School, Boston, MA 02115, USA.
Khurana T S
Cohen J B
Kunkel L M
Article Info
Journal
Human molecular genetics
Abbr.
Hum Mol Genet
ISSN
0964-6906
Published
1996-04-00
Pages
489-96
Language
English
Region
England
NLM ID
9208958
Subset
IM
Grants
NINDS NIH HHS · 5 R01 NS 23740-10 · United States
NINDS NIH HHS · NS29343 · United States
Databases
GENBANK
U26742, U26743, U26744, U46744, U46745, U46746
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