PUBLICATION

Genetic compensation between Pax3 and Pax7 in zebrafish appendicular muscle formation

Authors

Nord, H., Kahsay, A., Dennhag, N., Pedrosa Domellöf, F., von Hofsten, J.

ID

ZDB-PUB-210827-6

Date

2021

Source

Developmental Dynamics : an official publication of the American Association of Anatomists 251(9): 1423-1438 (Journal)

Registered Authors

Nord, Hanna, von Hofsten, Jonas

Keywords

appendicular myogenesis, limb development, muscle regeneration

MeSH Terms

Paired Box Transcription Factors*/genetics
Animals
Mammals
PAX3 Transcription Factor/genetics
Zebrafish*/genetics
Muscle Development/genetics
Muscle, Skeletal
PAX7 Transcription Factor/genetics

(all 8)

PubMed

34435397 Full text @ Dev. Dyn.

Abstract

Background Migrating muscle progenitors delaminate from the somite and subsequently form muscle tissue in distant anatomical regions such as the paired appendages, or limbs. In amniotes, this process requires a signaling cascade including the transcription factor paired box 3 (Pax3).

Results In this study we found that, unlike in mammals, pax3a/3b double mutant zebrafish develop near to normal appendicular muscle. By analyzing numerous mutant combinations of pax3a, pax3b and pax7a and pax7b, we determined that there is a feed-back system and a compensatory mechanism between Pax3 and Pax7 in this developmental process, even though Pax7 alone is not required for appendicular myogenesis. pax3a/3b/7a/7b quadruple mutant developed muscle-less pectoral fins.

Conclusions We found that Pax3 and Pax7 are redundantly required during appendicular myogenesis in zebrafish, where Pax7 is able to activate the same developmental programs as Pax3 in the pre-migratory progenitor cells. This article is protected by copyright. All rights reserved.

Genes / Markers

Figures

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Expression

Fish	Conditions	Stage	Anatomy	Assay	Figure
i150Tg	standard conditions	Prim-5	middle migratory muscle precursor stream	IFL	Fig. 6
i150Tg	perforation: somite 10	Day 5	myotome	IFL	Fig. 8
i150Tg	perforation: somite 10	Days 7-13	myotome	IFL	Fig. 8
i150Tg	perforation: somite 10	Day 6	myotome	IFL	Fig. 8
i150Tg	standard conditions	Long-pec	pectoral fin middle migratory muscle precursor stream	IFL	Fig. 6
i150Tg	standard conditions	20-25 somites	somite 2	IFL	Fig. 6
i150Tg	standard conditions	Prim-5	somite 2	IFL	Fig. 6
i150Tg	standard conditions	20-25 somites	somite 3	IFL	Fig. 6
i150Tg	standard conditions	Prim-5	somite 3	IFL	Fig. 6
i150Tg	standard conditions	20-25 somites	somite 4	IFL	Fig. 6

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
i150Tg	control	Day 5	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
i150Tg	control	Day 6	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
i150Tg	control	Days 7-13	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
i150Tg	perforation: somite 10	Day 5	myotome EGFP expression increased amount, abnormal	Fig. 8
i150Tg	perforation: somite 10	Day 5	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
i150Tg	perforation: somite 10	Day 6	myotome EGFP expression increased amount, abnormal	Fig. 8
i150Tg	perforation: somite 10	Day 6	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
i150Tg	perforation: somite 10	Days 7-13	myotome EGFP expression increased amount, abnormal	Fig. 8
i150Tg	perforation: somite 10	Days 7-13	somite 10 skeletal muscle tissue regeneration normal process quality, normal	Fig. 8
pax3a^umu5/umu5	standard conditions	Day 5	pectoral fin musculature functionality, normal	Fig. 2

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
i135Tg	Tg(-2.2 mylpfa:GFP)	Transgenic Insertion
i150Tg	TgBAC(pax3a:EGFP)	Transgenic Insertion
umu3		Small Deletion	pax7a
umu4		Indel	pax7b
umu5		Insertion	pax3a
umu6		Small Deletion	pax3b

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Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
pax3a	CRISPR1-pax3a	CRISPR
pax3b	CRISPR1-pax3b	CRISPR

Fish

Fish
i150Tg
pax3a^umu5/umu5
pax3a^umu5/umu5; i150Tg
pax3a^umu5/umu5; pax7b^umu4/umu4
pax3b^umu6/umu6
pax3b^umu6/umu6; i150Tg
pax3b^umu6/umu6; pax3a^umu5/umu5
pax3b^umu6/umu6; pax3a^umu5/umu5; i150Tg
pax3b^umu6/umu6; pax3a^umu5/umu5; pax7b^umu4/umu4
pax3b^umu6/umu6; pax7a^umu3/umu3

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Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP
GFP	EFG	GFP

Mapping

Nord et al., 2021 ZDB-PUB-210827-6 PMID:34435397

Genetic compensation between Pax3 and Pax7 in zebrafish appendicular muscle formation

Nord et al., 2021

ZDB-PUB-210827-6
PMID:34435397