PUBLICATION

Prep1.1 has essential genetic functions in hindbrain development and cranial neural crest cell differentiation

Authors

Deflorian, G., Tiso, N., Ferretti, E., Meyer, D., Blasi, F., Bortolussi, M., and Argenton, F.

ID

ZDB-PUB-040109-2

Date

2004

Source

Development (Cambridge, England) 131(3): 613-627 (Journal)

Registered Authors

Argenton, Francesco, Bortolussi, Marino, Deflorian, Gianluca, Meyer, Dirk, Tiso, Natascia

Keywords

none

MeSH Terms

Gastrula/metabolism
Branchial Region/embryology
Branchial Region/metabolism
Cell Differentiation/physiology*
DNA-Binding Proteins*
Zebrafish/embryology
Zebrafish/metabolism
Head/abnormalities
Head/physiology
Zebrafish Proteins/metabolism
Cartilage/abnormalities
Cartilage/metabolism
Neural Crest/embryology*
Neural Crest/metabolism
Animals
Homeodomain Proteins/genetics*
Homeodomain Proteins/metabolism
Protein Transport/physiology
Rhombencephalon/embryology*
Rhombencephalon/metabolism
Transcription Factors
Cell Nucleus/metabolism
Embryo, Nonmammalian/metabolism*

(all 23)

PubMed

14711874 Full text @ Development

Abstract

In this study we analysed the function of the Meinox gene prep1.1 during zebrafish development. Meinox proteins form heterotrimeric complexes with Hox and Pbx members, increasing the DNA binding specificity of Hox proteins in vitro and in vivo. However, a role for a specific Meinox protein in the regulation of Hox activity in vivo has not been demonstrated. In situ hybridization showed that prep1.1 is expressed maternally and ubiquitously up to 24 hours post-fertilization (hpf), and restricted to the head from 48 hpf onwards. Morpholino-induced prep1.1 loss-of-function caused significant apoptosis in the CNS. Hindbrain segmentation and patterning was affected severely, as revealed by either loss or defective expression of several hindbrain markers (foxb1.2/mariposa, krox20, pax2.1 and pax6.1), including anteriorly expressed Hox genes (hoxb1a, hoxa2 and hoxb2), the impaired migration of facial nerve motor neurons, and the lack of reticulospinal neurons (RSNs) except Mauthner cells. Furthermore, the heads of prep1.1 morphants lacked all pharyngeal cartilages. This was not caused by the absence of neural crest cells or their impaired migration into the pharyngeal arches, as shown by expression of dlx2 and snail1, but by the inability of these cells to differentiate into chondroblasts. Our results indicate that prep1.1 has a unique genetic function in craniofacial chondrogenesis and, acting as a member of Meinox-Pbc-Hox trimers, it plays an essential role in hindbrain development.

Genes / Markers

Figures

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Expression

Gene	Fish	Conditions	Stage	Qualifier	Anatomy	Assay	Figure
GFP	ml1Tg	standard conditions	Protruding-mouth to Days 21-29		pharyngeal arch 1	IFL	Fig. 7
GFP	ml1Tg	standard conditions	Protruding-mouth to Days 21-29		telencephalon anterior region	IFL	Fig. 7
GFP	ml1Tg + MO2-pknox1.1	standard conditions	Protruding-mouth to Days 21-29	Not Detected	pharyngeal arch 1	IFL	Fig. 7
GFP	ml1Tg + MO2-pknox1.1	standard conditions	Protruding-mouth to Days 21-29		telencephalon anterior region	IFL	Fig. 7
pknox1.1	WT	standard conditions	Protruding-mouth		brain	ISH	Fig. 1
pknox1.1	WT	standard conditions	Long-pec		head	ISH	Fig. 1
pknox1.1	WT	standard conditions	Prim-5		head	ISH	Fig. 1
pknox1.1	WT	standard conditions	Protruding-mouth		otic vesicle	ISH	Fig. 1
pknox1.1	WT	standard conditions	Adult		ovary	RTPCR	Fig. 1
pknox1.1	WT	standard conditions	Prim-5		post-vent region	ISH	Fig. 1

1 - 10 of 28

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO1-pbx4	standard conditions	Prim-5	pericardium edematous, abnormal	Fig. 3
WT + MO1-pbx4	standard conditions	Day 5	eye decreased size, abnormal	Fig. 3
WT + MO1-pbx4	standard conditions	Day 5	head decreased size, abnormal	Fig. 3
WT + MO1-pbx4	standard conditions	Day 5	pericardium edematous, abnormal	Fig. 3
WT + MO1-pbx4	standard conditions	Day 5	swim bladder inflation arrested, abnormal	Fig. 3
WT + MO1-pknox1.1	standard conditions	Prim-5	brain morphology, abnormal	Fig. 3
WT + MO1-pknox1.1	standard conditions	Prim-5	cell death increased occurrence, abnormal	Fig. 3
WT + MO1-pknox1.1	standard conditions	Prim-5	pericardium edematous, abnormal	Fig. 3
WT + MO1-pknox1.1	standard conditions	Prim-5	swim bladder aplastic, abnormal	Fig. 3
WT + MO1-pknox1.1	standard conditions	Day 5	eye decreased size, abnormal	Fig. 3

1 - 10 of 30

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
ml1Tg	Tg(-1.8gsc:GFP)	Transgenic Insertion

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
pbx4	MO1-pbx4	MRPHLNO
pknox1.1	MO1-pknox1.1	MRPHLNO
pknox1.1	MO2-pknox1.1	MRPHLNO

Fish

Fish
ml1Tg
ml1Tg + MO2-pknox1.1
WT
WT + MO1-pbx4
WT + MO1-pknox1.1
WT + MO2-pknox1.1

Antibodies

Orthology

Gene	Orthology
pknox1.1

Engineered Foreign Genes

Marker	Marker Type	Name
GFP	EFG	GFP

Mapping

Deflorian et al., 2004 ZDB-PUB-040109-2 PMID:14711874

Prep1.1 has essential genetic functions in hindbrain development and cranial neural crest cell differentiation

Deflorian et al., 2004

ZDB-PUB-040109-2
PMID:14711874