PMID- 23482490
OWN - NLM
STAT- MEDLINE
DCOM- 20130514
LR  - 20211021
IS  - 1477-9129 (Electronic)
IS  - 0950-1991 (Print)
IS  - 0950-1991 (Linking)
VI  - 140
IP  - 7
DP  - 2013 Apr
TI  - Lethal giant larvae 2 regulates development of the ciliated organ Kupffer's 
      vesicle.
PG  - 1550-9
LID - 10.1242/dev.087130 [doi]
AB  - Motile cilia perform crucial functions during embryonic development and 
      throughout adult life. Development of organs containing motile cilia involves 
      regulation of cilia formation (ciliogenesis) and formation of a luminal space 
      (lumenogenesis) in which cilia generate fluid flows. Control of ciliogenesis and 
      lumenogenesis is not yet fully understood, and it remains unclear whether these 
      processes are coupled. In the zebrafish embryo, lethal giant larvae 2 (lgl2) is 
      expressed prominently in ciliated organs. Lgl proteins are involved in 
      establishing cell polarity and have been implicated in vesicle trafficking. Here, 
      we identified a role for Lgl2 in development of ciliated epithelia in Kupffer's 
      vesicle, which directs left-right asymmetry of the embryo; the otic vesicles, 
      which give rise to the inner ear; and the pronephric ducts of the kidney. Using 
      Kupffer's vesicle as a model ciliated organ, we found that depletion of Lgl2 
      disrupted lumen formation and reduced cilia number and length. Immunofluorescence 
      and time-lapse imaging of Kupffer's vesicle morphogenesis in Lgl2-deficient 
      embryos suggested cell adhesion defects and revealed loss of the adherens 
      junction component E-cadherin at lateral membranes. Genetic interaction 
      experiments indicate that Lgl2 interacts with Rab11a to regulate E-cadherin and 
      mediate lumen formation that is uncoupled from cilia formation. These results 
      uncover new roles and interactions for Lgl2 that are crucial for both 
      lumenogenesis and ciliogenesis and indicate that these processes are genetically 
      separable in zebrafish.
FAU - Tay, Hwee Goon
AU  - Tay HG
AD  - Department of Cell and Developmental Biology, State University of New York, 
      Upstate Medical University, Syracuse, NY 13210, USA.
FAU - Schulze, Sabrina K
AU  - Schulze SK
FAU - Compagnon, Julien
AU  - Compagnon J
FAU - Foley, Fiona C
AU  - Foley FC
FAU - Heisenberg, Carl-Philipp
AU  - Heisenberg CP
FAU - Yost, H Joseph
AU  - Yost HJ
FAU - Abdelilah-Seyfried, Salim
AU  - Abdelilah-Seyfried S
FAU - Amack, Jeffrey D
AU  - Amack JD
LA  - eng
GR  - R01 HL095690/HL/NHLBI NIH HHS/United States
GR  - R01HL095690/HL/NHLBI NIH HHS/United States
PT  - Journal Article
PT  - Research Support, N.I.H., Extramural
PT  - Research Support, Non-U.S. Gov't
PL  - England
TA  - Development
JT  - Development (Cambridge, England)
JID - 8701744
RN  - 0 (Zebrafish Proteins)
RN  - 0 (llgl2 protein, zebrafish)
SB  - IM
MH  - Animals
MH  - Animals, Genetically Modified
MH  - Body Patterning/genetics
MH  - Cell Polarity/genetics
MH  - Cilia/genetics/metabolism/*physiology
MH  - Embryo, Nonmammalian
MH  - Embryonic Development/genetics/physiology
MH  - Gene Expression Regulation, Developmental
MH  - Kupffer Cells/metabolism/*physiology
MH  - Larva/genetics/metabolism
MH  - Morphogenesis/*genetics/physiology
MH  - *Zebrafish/embryology/genetics
MH  - Zebrafish Proteins/genetics/metabolism/*physiology
PMC - PMC3596994
EDAT- 2013/03/14 06:00
MHDA- 2013/05/15 06:00
PMCR- 2014/04/01
CRDT- 2013/03/14 06:00
PHST- 2013/03/14 06:00 [entrez]
PHST- 2013/03/14 06:00 [pubmed]
PHST- 2013/05/15 06:00 [medline]
PHST- 2014/04/01 00:00 [pmc-release]
AID - 140/7/1550 [pii]
AID - 10.1242/dev.087130 [doi]
PST - ppublish
SO  - Development. 2013 Apr;140(7):1550-9. doi: 10.1242/dev.087130.