Abstract
The C-terminal Eps15 homology domain-containing (EHD) proteins play a key role in endocytic recycling, a fundamental cellular process that ensures the return of endocytosed membrane components and receptors back to the cell surface. To define the. in vivo biological functions of EHD1, we have generated. Ehd1 knockout mice and previously reported a requirement of EHD1 for spermatogenesis. Here, we show that approximately 56% of the. Ehd1-null mice displayed gross ocular abnormalities, including anophthalmia, aphakia, microphthalmia and congenital cataracts. Histological characterization of ocular abnormalities showed pleiotropic defects that include a smaller or absent lens, persistence of lens stalk and hyaloid vasculature, and deformed optic cups. To test whether these profound ocular defects resulted from the loss of EHD1 in the lens or in non-lenticular tissues, we deleted the. Ehd1 gene selectively in the presumptive lens ectoderm using. Le-Cre. Conditional. Ehd1 deletion in the lens resulted in developmental defects that included thin epithelial layers, small lenses and absence of corneal endothelium + Ehd1 deletion in the lens also resulted in reduced lens epithelial proliferation, survival and expression of junctional proteins E-cadherin and ZO-1. Finally,. Le-Cre-mediated deletion of. Ehd1 in the lens led to defects in corneal endothelial differentiation. Taken together, these data reveal a unique role for EHD1 in early lens development and suggest a previously unknown link between the endocytic recycling pathway and regulation of key developmental processes including proliferation, differentiation and morphogenesis.
| Original language | English |
|---|---|
| Journal | Developmental Biology |
| DOIs | |
| State | Accepted/In press - Mar 17 2015 |
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All Science Journal Classification (ASJC) codes
- Developmental Biology
- Cell Biology
- Molecular Biology
Cite this
The endocytic recycling regulatory protein EHD1 Is required for ocular lens development. / Arya, Priyanka; Rainey, Mark A.; Bhattacharyya, Sohinee; Mohapatra, Bhopal C.; George, Manju; Kuracha, Murali R.; Storck, Matthew D.; Band, Vimla; Govindarajan, Venkatesh; Band, Hamid.
In: Developmental Biology, 17.03.2015.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - The endocytic recycling regulatory protein EHD1 Is required for ocular lens development
AU - Arya, Priyanka
AU - Rainey, Mark A.
AU - Bhattacharyya, Sohinee
AU - Mohapatra, Bhopal C.
AU - George, Manju
AU - Kuracha, Murali R.
AU - Storck, Matthew D.
AU - Band, Vimla
AU - Govindarajan, Venkatesh
AU - Band, Hamid
PY - 2015/3/17
Y1 - 2015/3/17
N2 - The C-terminal Eps15 homology domain-containing (EHD) proteins play a key role in endocytic recycling, a fundamental cellular process that ensures the return of endocytosed membrane components and receptors back to the cell surface. To define the. in vivo biological functions of EHD1, we have generated. Ehd1 knockout mice and previously reported a requirement of EHD1 for spermatogenesis. Here, we show that approximately 56% of the. Ehd1-null mice displayed gross ocular abnormalities, including anophthalmia, aphakia, microphthalmia and congenital cataracts. Histological characterization of ocular abnormalities showed pleiotropic defects that include a smaller or absent lens, persistence of lens stalk and hyaloid vasculature, and deformed optic cups. To test whether these profound ocular defects resulted from the loss of EHD1 in the lens or in non-lenticular tissues, we deleted the. Ehd1 gene selectively in the presumptive lens ectoderm using. Le-Cre. Conditional. Ehd1 deletion in the lens resulted in developmental defects that included thin epithelial layers, small lenses and absence of corneal endothelium + Ehd1 deletion in the lens also resulted in reduced lens epithelial proliferation, survival and expression of junctional proteins E-cadherin and ZO-1. Finally,. Le-Cre-mediated deletion of. Ehd1 in the lens led to defects in corneal endothelial differentiation. Taken together, these data reveal a unique role for EHD1 in early lens development and suggest a previously unknown link between the endocytic recycling pathway and regulation of key developmental processes including proliferation, differentiation and morphogenesis.
AB - The C-terminal Eps15 homology domain-containing (EHD) proteins play a key role in endocytic recycling, a fundamental cellular process that ensures the return of endocytosed membrane components and receptors back to the cell surface. To define the. in vivo biological functions of EHD1, we have generated. Ehd1 knockout mice and previously reported a requirement of EHD1 for spermatogenesis. Here, we show that approximately 56% of the. Ehd1-null mice displayed gross ocular abnormalities, including anophthalmia, aphakia, microphthalmia and congenital cataracts. Histological characterization of ocular abnormalities showed pleiotropic defects that include a smaller or absent lens, persistence of lens stalk and hyaloid vasculature, and deformed optic cups. To test whether these profound ocular defects resulted from the loss of EHD1 in the lens or in non-lenticular tissues, we deleted the. Ehd1 gene selectively in the presumptive lens ectoderm using. Le-Cre. Conditional. Ehd1 deletion in the lens resulted in developmental defects that included thin epithelial layers, small lenses and absence of corneal endothelium + Ehd1 deletion in the lens also resulted in reduced lens epithelial proliferation, survival and expression of junctional proteins E-cadherin and ZO-1. Finally,. Le-Cre-mediated deletion of. Ehd1 in the lens led to defects in corneal endothelial differentiation. Taken together, these data reveal a unique role for EHD1 in early lens development and suggest a previously unknown link between the endocytic recycling pathway and regulation of key developmental processes including proliferation, differentiation and morphogenesis.
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U2 - 10.1016/j.ydbio.2015.10.005
DO - 10.1016/j.ydbio.2015.10.005
M3 - Article
C2 - 26455409
AN - SCOPUS:84951735730
JO - Developmental Biology
JF - Developmental Biology
SN - 0012-1606
ER -