The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.

M. Sylva; V.S.W. Li; A.A. Buffing; J.H. van Es; M. van den Born; S. Velden; Q. Gunst; J.H. Koolstra; A.F. Moorman; H. Clevers; M.J. van den Hoff

The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.

Translated title of the contribution: The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.

M. Sylva, V.S.W. Li, A.A. Buffing, J.H. van Es, M. van den Born, S. Velden, Q. Gunst, J.H. Koolstra, A.F. Moorman, H. Clevers, M.J. van den Hoff

Research output: Contribution to journal › Article › Academic › peer-review

Abstract

Follistatin-like 1 (Fstl1) is a secreted protein of the BMP inhibitor class. During development, expression of Fstl1 is already found in cleavage stage embryos and becomes gradually restricted to mesenchymal elements of most organs during subsequent development. Knock down experiments in chicken and zebrafish demonstrated a role as a BMP antagonist in early development. To investigate the role of Fstl1 during mouse development, a conditional Fstl1 KO allele as well as a Fstl1-GFP reporter mouse were created. KO mice die at birth from respiratory distress and show multiple defects in lung development. Also, skeletal development is affected. Endochondral bone development, limb patterning as well as patterning of the axial skeleton are perturbed in the absence of Fstl1. Taken together, these observations show that Fstl1 is a crucial regulator in BMP signalling during mouse development.

Translated title of the contribution	The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.
Original language	Undefined/Unknown
Pages (from-to)	e22616
Number of pages	1
Journal	PLoS ONE [E]
Volume	6
Issue number	8
Publication status	Published - 2011

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http://www.ncbi.nlm.nih.gov/pubmed/21826198

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title = "The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.",

abstract = "Follistatin-like 1 (Fstl1) is a secreted protein of the BMP inhibitor class. During development, expression of Fstl1 is already found in cleavage stage embryos and becomes gradually restricted to mesenchymal elements of most organs during subsequent development. Knock down experiments in chicken and zebrafish demonstrated a role as a BMP antagonist in early development. To investigate the role of Fstl1 during mouse development, a conditional Fstl1 KO allele as well as a Fstl1-GFP reporter mouse were created. KO mice die at birth from respiratory distress and show multiple defects in lung development. Also, skeletal development is affected. Endochondral bone development, limb patterning as well as patterning of the axial skeleton are perturbed in the absence of Fstl1. Taken together, these observations show that Fstl1 is a crucial regulator in BMP signalling during mouse development.",

author = "M. Sylva and V.S.W. Li and A.A. Buffing and {van Es}, J.H. and {van den Born}, M. and S. Velden and Q. Gunst and J.H. Koolstra and A.F. Moorman and H. Clevers and {van den Hoff}, M.J.",

year = "2011",

language = "Undefined/Unknown",

volume = "6",

pages = "e22616",

journal = "PLoS ONE [E]",

issn = "1932-6203",

publisher = "Public Library of Science",

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TY - JOUR

T1 - The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.

AU - Sylva, M.

AU - Li, V.S.W.

AU - Buffing, A.A.

AU - van Es, J.H.

AU - van den Born, M.

AU - Velden, S.

AU - Gunst, Q.

AU - Koolstra, J.H.

AU - Moorman, A.F.

AU - Clevers, H.

AU - van den Hoff, M.J.

PY - 2011

Y1 - 2011

N2 - Follistatin-like 1 (Fstl1) is a secreted protein of the BMP inhibitor class. During development, expression of Fstl1 is already found in cleavage stage embryos and becomes gradually restricted to mesenchymal elements of most organs during subsequent development. Knock down experiments in chicken and zebrafish demonstrated a role as a BMP antagonist in early development. To investigate the role of Fstl1 during mouse development, a conditional Fstl1 KO allele as well as a Fstl1-GFP reporter mouse were created. KO mice die at birth from respiratory distress and show multiple defects in lung development. Also, skeletal development is affected. Endochondral bone development, limb patterning as well as patterning of the axial skeleton are perturbed in the absence of Fstl1. Taken together, these observations show that Fstl1 is a crucial regulator in BMP signalling during mouse development.

AB - Follistatin-like 1 (Fstl1) is a secreted protein of the BMP inhibitor class. During development, expression of Fstl1 is already found in cleavage stage embryos and becomes gradually restricted to mesenchymal elements of most organs during subsequent development. Knock down experiments in chicken and zebrafish demonstrated a role as a BMP antagonist in early development. To investigate the role of Fstl1 during mouse development, a conditional Fstl1 KO allele as well as a Fstl1-GFP reporter mouse were created. KO mice die at birth from respiratory distress and show multiple defects in lung development. Also, skeletal development is affected. Endochondral bone development, limb patterning as well as patterning of the axial skeleton are perturbed in the absence of Fstl1. Taken together, these observations show that Fstl1 is a crucial regulator in BMP signalling during mouse development.

M3 - Article

SN - 1932-6203

VL - 6

SP - e22616

JO - PLoS ONE [E]

JF - PLoS ONE [E]

IS - 8

ER -

The BMP antagonist follistatin-like 1 is required for skeletal and lung organogenesis.

Abstract

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