PMID- 17010201
OWN - NLM
STAT- MEDLINE
DCOM- 20061026
LR  - 20230507
IS  - 1471-213X (Electronic)
IS  - 1471-213X (Linking)
VI  - 6
DP  - 2006 Sep 29
TI  - Eye and neural defects associated with loss of GDF6.
PG  - 43
AB  - BACKGROUND: In Xenopus the bone morphogenetic protein growth and differentiation 
      factor 6 (GDF6) is expressed at the edge of the neural plate, and within the 
      anterior neural plate including the eye fields. Here we address the role of GDF6 
      in neural and eye development by morpholino knockdown experiments. RESULTS: We 
      show that depletion of GDF6 (BMP13) resulted in a reduction in eye size, loss of 
      laminar structure and a reduction in differentiated neural cell types within the 
      retina. This correlated with a reduction in staining for Smad1/5/8 
      phosphorylation indicating a decrease in GDF6 signalling through loss of 
      phosphorylation of these intracellular mediators of bone morphogenetic protein 
      (BMP) signalling. In addition, the Pax6 expression domain is reduced in size at 
      early optic vesicle stages. Neural cell adhesion molecule (NCAM) is generally 
      reduced in intensity along the neural tube, while in the retina and brain 
      discreet patches of NCAM expression are also lost. GDF6 knock down resulted in an 
      increase in cell death along the neural tube and within the retina as determined 
      by terminal deoxynucleotidyl transferase-mediated dUTP nick end labeling (TUNEL) 
      staining. CONCLUSION: Our data demonstrate that GDF6 has an important role in 
      neural differentiation in the eye as well as within the central nervous system, 
      and that GDF6 may act in some way to maintain cell survival within the ectoderm, 
      during the normal waves of programmed cell death.
FAU - Hanel, Meredith L
AU  - Hanel ML
AD  - UCD School of Biomolecular and Biomedical Science, Conway Institute, University 
      College Dublin, Belfield, Dublin 4, Ireland. meredith.hanel@ucd.ie
FAU - Hensey, Carmel
AU  - Hensey C
LA  - eng
GR  - WT_/Wellcome Trust/United Kingdom
GR  - 061410/GM/SP/JAT/WT_/Wellcome Trust/United Kingdom
GR  - 057990/2/99/2/WT_/Wellcome Trust/United Kingdom
PT  - Journal Article
PT  - Research Support, Non-U.S. Gov't
DEP - 20060929
PL  - England
TA  - BMC Dev Biol
JT  - BMC developmental biology
JID - 100966973
RN  - 0 (Bone Morphogenetic Proteins)
RN  - 0 (Eye Proteins)
RN  - 0 (Growth Differentiation Factor 6)
RN  - 0 (Homeodomain Proteins)
RN  - 0 (PAX6 Transcription Factor)
RN  - 0 (Paired Box Transcription Factors)
RN  - 0 (RNA, Messenger)
RN  - 0 (Repressor Proteins)
RN  - 0 (Xenopus Proteins)
SB  - IM
MH  - Animals
MH  - Apoptosis/genetics
MH  - Blotting, Western
MH  - Bone Morphogenetic Proteins/deficiency/*genetics/physiology
MH  - Embryo, Nonmammalian/abnormalities/embryology/metabolism
MH  - Eye Abnormalities/*genetics
MH  - Eye Proteins/genetics/metabolism
MH  - Gene Expression Regulation, Developmental/*genetics
MH  - Growth Differentiation Factor 6
MH  - Homeodomain Proteins/genetics/metabolism
MH  - Immunohistochemistry
MH  - In Situ Hybridization
MH  - In Situ Nick-End Labeling
MH  - Nervous System/embryology/metabolism/pathology
MH  - Nervous System Diseases/embryology/*genetics
MH  - PAX6 Transcription Factor
MH  - Paired Box Transcription Factors/genetics/metabolism
MH  - RNA, Messenger/genetics/metabolism
MH  - Repressor Proteins/genetics/metabolism
MH  - Retina/abnormalities/metabolism
MH  - Xenopus/embryology/*genetics
MH  - Xenopus Proteins/deficiency/genetics/physiology
PMC - PMC1609107
EDAT- 2006/10/03 09:00
MHDA- 2006/10/27 09:00
PMCR- 2006/09/29
CRDT- 2006/10/03 09:00
PHST- 2006/06/02 00:00 [received]
PHST- 2006/09/29 00:00 [accepted]
PHST- 2006/10/03 09:00 [pubmed]
PHST- 2006/10/27 09:00 [medline]
PHST- 2006/10/03 09:00 [entrez]
PHST- 2006/09/29 00:00 [pmc-release]
AID - 1471-213X-6-43 [pii]
AID - 10.1186/1471-213X-6-43 [doi]
PST - epublish
SO  - BMC Dev Biol. 2006 Sep 29;6:43. doi: 10.1186/1471-213X-6-43.