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Sept7b is essential for pronephric function and development of left-right asymmetry in zebrafish embryogenesis.
Dash, Surjya Narayan; Lehtonen, Eero; Wasik, Anita A; Schepis, Antonino; Paavola, Jere; Panula, Pertti; Nelson, W James; Lehtonen, Sanna.
Afiliación
  • Dash SN; University of Helsinki, Haartman Institute, Department of Pathology, Haartmaninkatu 3, 00290 Helsinki, Finland.
J Cell Sci ; 127(Pt 7): 1476-86, 2014 Apr 01.
Article en En | MEDLINE | ID: mdl-24496452
The conserved septin family of filamentous small GTPases plays important roles in mitosis, cell migration and cell morphogenesis by forming scaffolds and diffusion barriers. Recent studies in cultured cells in vitro indicate that a septin complex of septin 2, 7 and 9 is required for ciliogenesis and cilia function, but septin function in ciliogenesis in vertebrate organs in vivo is not understood. We show that sept7b is expressed in ciliated cells in different tissues during early zebrafish development. Knockdown of sept7b by using morpholino antisense oligonucleotides caused misorientation of basal bodies and cilia, reduction of apical actin and the shortening of motile cilia in Kupffer's vesicle and pronephric tubules. This resulted in pericardial and yolk sac edema, body axis curvature and hydrocephaly. Notably, in sept7b morphants we detected strong left-right asymmetry defects in the heart and lateral plate mesoderm (situs inversus), reduced fluid flow in the kidney, the formation of kidney cysts and loss of glomerular filtration barrier function. Thus, sept7b is essential during zebrafish development for pronephric function and ciliogenesis, and loss of expression of sept7b results in defects that resemble human ciliopathies.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pez Cebra / Proteínas de Pez Cebra / Septinas / Pronefro Límite: Animals Idioma: En Revista: J Cell Sci Año: 2014 Tipo del documento: Article País de afiliación: Finlandia Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Pez Cebra / Proteínas de Pez Cebra / Septinas / Pronefro Límite: Animals Idioma: En Revista: J Cell Sci Año: 2014 Tipo del documento: Article País de afiliación: Finlandia Pais de publicación: Reino Unido