PMID:15843411

Citation	Zakin, L, Reversade, B, Kuroda, H, Lyons, KM and De Robertis, EM (2005) Sirenomelia in Bmp7 and Tsg compound mutant mice: requirement for Bmp signaling in the development of ventral posterior mesoderm. Development 132:2489-99
Abstract	Sirenomelia or mermaid-like phenotype is one of the principal human congenital malformations that can be traced back to the stage of gastrulation. Sirenomelia is characterized by the fusion of the two hindlimbs into a single one. In the mouse, sirens have been observed in crosses between specific strains and as the consequence of mutations that increase retinoic acid levels. We report that the loss of bone morphogenetic protein 7 (Bmp7) in combination with a half dose or complete loss of twisted gastrulation (Tsg) causes sirenomelia in the mouse. Tsg is a Bmp- and chordin-binding protein that has multiple effects on Bmp metabolism in the extracellular space; Bmp7 is one of many Bmps and is shown here to bind to Tsg. In Xenopus, co-injection of Tsg and Bmp7 morpholino oligonucleotides (MO) has a synergistic effect, greatly inhibiting formation of ventral mesoderm and ventral fin tissue. In the mouse, molecular marker studies indicate that the sirenomelia phenotype is associated with a defect in the formation of ventroposterior mesoderm. These experiments demonstrate that dorsoventral patterning of the mouse posterior mesoderm is regulated by Bmp signaling, as is the case in other vertebrates. Sirens result from a fusion of the hindlimb buds caused by a defect in the formation of ventral mesoderm.
Links	PubMed Online version:10.1242/dev.01822
Keywords	Animals; Blotting, Western/veterinary; Body Patterning/physiology; Bone Morphogenetic Protein 7; Bone Morphogenetic Proteins/genetics; Bone Morphogenetic Proteins/metabolism; Ectromelia/embryology; Ectromelia/genetics; Ectromelia/veterinary; Gene Expression Regulation, Developmental; Hindlimb/pathology; Histological Techniques/veterinary; In Situ Hybridization/veterinary; Mesoderm/physiology; Mice; Mutation/genetics; Oligonucleotides, Antisense; Proteins/genetics; Proteins/metabolism; Reverse Transcriptase Polymerase Chain Reaction/veterinary; Rodent Diseases/embryology; Rodent Diseases/genetics; Signal Transduction; Transforming Growth Factor beta/genetics; Transforming Growth Factor beta/metabolism; Xenopus; Xenopus Proteins
edit table

Annotations

Gene product	Qualifier	GO ID	GO term name	Evidence Code	with/from	Aspect	Notes	Status
edit table

References

See Help:References for how to manage references in GONUTS.