Abstract:
:The processes of myelination remain incompletely understood but are of profound biomedical importance owing to the several dysmyelinating and demyelinating disorders known in humans. Here, we analyze the zebrafish puma mutant, isolated originally for pigment pattern defects limited to the adult stage. We show that puma mutants also have late-arising defects in Schwann cells of the peripheral nervous system, locomotor abnormalities, and sex-biased defects in adult craniofacial morphology. Using methods of positional cloning, we identify a critical genetic interval harboring two alpha tubulin loci, and we identify a chemically induced missense mutation in one of these, tubulin alpha 8-like 3a (tuba8l3a). We demonstrate tuba8l3a expression in the central nervous system (CNS), leading us to search for defects in the development of oligodendrocytes, the myelinating cells of the CNS. We find gross reductions in CNS myelin and oligodendrocyte numbers in adult puma mutants, and these deficits are apparent already during the larval-to-adult transformation. By contrast, analyses of embryos and early larvae reveal a normal complement of oligodendrocytes that nevertheless fail to localize normal amounts of myelin basic protein (mbp) mRNA in cellular processes, and fail to organize these processes as in the wild-type. This study identifies the puma mutant as a valuable model for studying microtubule-dependent events of myelination, as well as strategies for remyelination in the adult.
journal_name
Dev Bioljournal_title
Developmental biologyauthors
Larson TA,Gordon TN,Lau HE,Parichy DMdoi
10.1016/j.ydbio.2010.07.035subject
Has Abstractpub_date
2010-10-15 00:00:00pages
296-309issue
2eissn
0012-1606issn
1095-564Xpii
S0012-1606(10)00980-2journal_volume
346pub_type
杂志文章abstract::Cyclic protein-2 (CP-2) is secreted in vitro in substantial amounts by mature rat Sertoli cells in intact Stage VI and Stage VII seminiferous tubules. This stage-dependent secretion has led us to postulate that the biosynthesis of this molecule is stimulated by germ cells at a specific state of development. In order t...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/0012-1606(88)90412-5
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abstract::We have tested the role of proteoglycans in the development of feather pattern by culturing 7-day-old embryonic chick skins on medium containing para-nitrophenyl-beta-D-xyloside (2 mM). Xylosides compete with core proteins of proteoglycans by acting as exogenous acceptors for the synthesis of glycosaminoglycans leadin...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/0012-1606(88)90199-6
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journal_title:Developmental biology
pub_type: 杂志文章,评审
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abstract::Individual somatic muscles and heart progenitors are specified at defined positions within the mesodermal layer of Drosophila. The expression of the homeobox gene even-skipped (eve) identifies one specific subset of cells in the dorsal mesoderm, which give rise to particular pericardial cells and dorsal body wall musc...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1006/dbio.2001.0397
更新日期:2001-10-01 00:00:00
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journal_title:Developmental biology
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journal_title:Developmental biology
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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abstract::Oral-facial-digital type I (OFDI) syndrome is an X-linked male lethal developmental disorder. It is ascribed to ciliary dysfunction and characterized by malformation of the face, oral cavity, and digits. Conditional inactivation using different Cre lines allowed us to study the role of the Ofd1 transcript in limb deve...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2010.09.020
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2005.10.025
更新日期:2006-01-01 00:00:00
abstract::The Caenorhabditis elegans EGL-17/FGF protein is involved in the gonadal signaling that guides the migrations of sex myoblasts (SMs). egl-17::GFP reporter constructs are expressed dynamically in vulval cell lineages. Expression in the primary vulval cells is correlated with the precise positioning of SMs. We have inve...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/s0012-1606(03)00033-2
更新日期:2003-05-01 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
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更新日期:1997-08-15 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
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更新日期:2020-12-01 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/0012-1606(84)90081-2
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2005.10.045
更新日期:2006-03-01 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2010.08.017
更新日期:2010-12-01 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1006/dbio.1995.1149
更新日期:1995-05-01 00:00:00
abstract::In developing limb buds, Msx2 transcripts are expressed in the apical ectodermal ridge (AER) and in various regions of the limb mesenchyme. To identify DNA sequences responsible for Msx2 expression in the AER, we characterized the expression of LacZ reporter constructs driven by chicken Msx2 regulatory sequences in tr...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2004.03.005
更新日期:2004-06-15 00:00:00
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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abstract::To feed or breathe, the oral opening must connect with the gut. The foregut and oral tissues converge at the primary mouth, forming the buccopharyngeal membrane (BPM), a bilayer epithelium. Failure to form the opening between gut and mouth has significant ramifications, and many craniofacial disorders have been associ...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2014.09.029
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journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/0012-1606(84)90229-x
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journal_title:Developmental biology
pub_type: 杂志文章
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abstract::The Drosophila ovarian germline stem cells (GSCs) constantly experience self-renewal and differentiation, ensuring the female fertility throughout life. The balance between GSC self-renewal and differentiation is exquisitely regulated by the stem cell niche, the stem cells themselves and systemic factors. Increasing e...
journal_title:Developmental biology
pub_type: 杂志文章
doi:10.1016/j.ydbio.2013.04.015
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journal_title:Developmental biology
pub_type: 杂志文章
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journal_title:Developmental biology
pub_type: 杂志文章
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abstract::Hox genes of the Abdominal-B (Abd-B) class regulate gonadal development in diverse metazoans. Here we have investigated the role of the Abd-B homolog egl-5 in C. elegans gonadal development. Previous work showed that egl-5 is required male-specifically in the gonad and that mutant gonads are highly dysgenic and possib...
journal_title:Developmental biology
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