Human Molecular Genetics, Vol 7, 589-595, Copyright © 1998 by Oxford University Press
RG Roberts and M Bobrow
Members of the dystrophin family of proteins perform a critical but
incompletely characterized role in the maintenance of membrane- associated
complexes at points of intercellular contact in many vertebrate cell types.
They interact with, amongst others, the transmembrane laminin receptor
dystroglycan, cytoskeletal actin and, indirectly, the intracellular
membrane-associated signalling enzyme neuronal nitric oxide synthase
(nNOS). Here we describe sequences of a range of dystrophin-related
proteins from vertebrate and invertebrate animals (including the important
model organism Drosophila melanogaster ) and infer an evolutionary history
of this family and its relationship to the distantly related dystrobrevins.
It appears that most metazoa possess sequences encoding a single highly
conserved dystrophin-like protein in addition to a presumed distinct
dystrobrevin, derived from an early duplication of an ancestral gene. In
the vertebrates (but not the protochordate Amphioxus), the single
invertebrate dystrophin-like gene has undergone serial duplication to
generate at least three distinct genes encoding proteins which have adopted
specialized roles. It is hoped that this broadening of the biology of the
dystrophins will afford further opportunities for the advancement of our
understanding of the fundamental defect underlying the variety of human
genetic disorders which result from aberrant or absent
dystrophin-associated complexes.
ARTICLES
Dystrophins in vertebrates and invertebrates
Department of Medical Genetics, Addenbrookes Hospital, Cambridge, UK. rroberts@hgmp.mrc.ac.uk
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