OC29 is preferentially expressed in the presumptive sensory organ region of the otocyst

Nishida, Akiko; Kobuke, Kazuhiro; Kojima, Ken; Ito, Juichi; Honjo, Tasuku; Tashiro, Kei

doi:10.1002/dvdy.20180

Cited by 5 publications

(7 citation statements)

References 46 publications

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“…In contrast, Gasp2 expression was readily detected in a variety of sites in day 9.5 postcoitum embryos ( Fig. 2 B and C), including the retina, otocyst, and neural tube, consistent with previously reported information on rat embryos (22). One notable difference between our results and the results of the prior study, however, was that we also observed prominent expression of Gasp2 in the posterior region of the embryo (Fig.…”

Section: Significancesupporting

confidence: 92%

Regulation of GDF-11 and myostatin activity by GASP-1 and GASP-2

Lee

2013

Proc. Natl. Acad. Sci. U.S.A.

103

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Myostatin (MSTN) and growth and differentiation factor-11 (GDF-11) are highly related TGF-β family members that have distinct biological functions. MSTN is expressed primarily in skeletal muscle and acts to limit muscle growth. GDF-11 is expressed more widely and plays multiple roles, including regulating axial skeletal patterning during development. Several MSTN and GDF-11 binding proteins have been identified, including GDF-associated serum protein-1 (GASP-1) and GASP-2, which are capable of inhibiting the activities of these ligands. Here, we show that GASP-1 and GASP-2 act by blocking the initial signaling event (namely, the binding of the ligand to the type II receptor). Moreover, we show that mice lacking Gasp1 and Gasp2 have phenotypes consistent with overactivity of MSTN and GDF-11. Specifically, we show that Gasp2 −/− mice have posteriorly directed transformations of the axial skeleton, which contrast with the anteriorly directed transformations seen in Gdf11 −/− mice. We also show that both Gasp1 −/− and Gasp2 −/− mice have reductions in muscle weights, a shift in fiber type from fast glycolytic type IIb fibers to fast oxidative type IIa fibers, and impaired muscle regeneration ability, which are the reverse of what are seen in Mstn −/− mice. All of these findings suggest that both GASP-1 and GASP-2 are important modulators of GDF-11 and MSTN activity in vivo.

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Section: Significancesupporting

confidence: 92%

Regulation of GDF-11 and myostatin activity by GASP-1 and GASP-2

Lee

2013

Proc. Natl. Acad. Sci. U.S.A.

103

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“…Growth factor-binding proteins may control the action of growth factors not only by inhibiting their action but also by serving as a reservoir for growth factors, may localize their action in the vicinity of the binding proteins and thus may help to establish growth factor gradients in the extracellular space through physical association. It may be relevant in this respect that WFIKKN1 is expressed in the developing inner ear, mainly in the BMP4-positive presumptive cristae, and it was suggested that WFIKKN1 might be involved in the early development of the inner-ear sensory organ by controlling the action of BMP4 [4]. …”

Section: Discussionmentioning

confidence: 99%

“…In human fetal tissues, the expression of WFIKKN1 was highest in lung, skeletal muscle and liver, whereas WFIKKN2 expression was found in brain, skeletal muscle, kidney and thymus [2]. It was shown that OC29 (the rat orthologue of human WFIKKN1) is also expressed in the presumptive cristae of the developing inner ear [4].…”

Section: Expression Of Wfikkn Proteinsmentioning

confidence: 97%

Biological functions of the WAP domain-containing multidomain proteins WFIKKN1 and WFIKKN2

Kondás

Szláma

Nagy

et al. 2011

Biochemical Society Transactions

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WFIKKN1 and WFIKKN2 are two closely related multidomain proteins consisting of a WAP (whey acidic protein)-, a follistatin-, an immunoglobulin-, two Kunitz-type protease inhibitor-domains and an NTR domain (netrin domain). Recent experiments have shown that both WFIKKN1 and WFIKKN2 bind myostatin and GDF11 (growth and differentiation factor 11) with high affinity and are potent antagonists of these growth factors. Structure-function studies on WFIKKN proteins have revealed that their interactions with GDF8 and GDF11 are mediated primarily by the follistatin and NTR domains.

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“…1). In contrast, Gasp2 expression decreased after E10.5, 7) and in adult tissue, its expression was limited to testis. 5) These distinct tissue expression patterns during development may contribute to differential regulation of myostatin and GDF-11 activities in specific tissues including muscle, tooth, palate, and ear.…”

Section: Resultsmentioning

confidence: 82%

Roles of GASP-1 and GDF-11 in Dental and Craniofacial Development

Lee

2015

Journal of Oral Medicine and Pain

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Purpose Growth and differentiation factor-11 (GDF-11) is a TGF-β family member that plays important regulatory roles in development of multiple tissues which include axial skeletal patterning, palatal closure, and tooth formation. Proteins that have been identified as GDF-11 inhibitors include GDF-associated serum protein-1 (GASP-1) and GASP-2. Recently, we found that mice genetically engineered to lack both Gasp1 and Gdf11 have an increased frequency of cleft palate. The goal of this study was to investigate the roles of GDF-11 and its inhibitors, GASP-1 and GASP-2, during dental and craniofacial development and growth. Methods Mouse genetic studies were used in this study. Homozygous knockout mice for Gasp1 (Gasp1−/−) and Gasp2 (Gasp2−/−) were viable and fertile, but Gdf11 homozygous knockout (Gdf11−/−) mice died within 24 hours after birth. The effect of either Gasp1 or Gasp2 deletion in Gdf11−/− mice during embryogenesis was evaluated in Gasp1−/−;Gdf11−/− and Gasp2−/−;Gdf11−/− mouse embryos at 18.5 days post-coitum (E18.5). For the analysis of adult tissues, we used Gasp1−/−;Gdf11+/− and Gasp2−/−;Gdf11+/− mice to evaluate the potential haploinsufficiency of Gdf11 in Gasp1−/− and Gasp2−/− mice. Results Although Gasp2 expression decreased after E10.5, Gasp1 expression was readily detected in various ectodermal tissues at E17.5, including hair follicles, epithelium in nasal cavity, retina, and developing tooth buds. Interestingly, Gasp1−/−;Gdf11−/− mice had abnormal formation of lower incisors: tooth buds for lower incisors were under-developed or missing. Although Gdf11+/− mice were viable and had mild transformations of the axial skeleton, no specific defects in the craniofacial development have been observed in Gdf11+/− mice. However, loss of Gasp1 in Gdf11+/− mice occasionally resulted in small and abnormally shaped auricles. Conclusions These findings suggest that both GASP-1 and GDF-11 play important roles in dental and craniofacial development both during embryogenesis and in adult tissues.

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OC29 is preferentially expressed in the presumptive sensory organ region of the otocyst

Cited by 5 publications

References 46 publications

Regulation of GDF-11 and myostatin activity by GASP-1 and GASP-2

Regulation of GDF-11 and myostatin activity by GASP-1 and GASP-2

Biological functions of the WAP domain-containing multidomain proteins WFIKKN1 and WFIKKN2

Roles of GASP-1 and GDF-11 in Dental and Craniofacial Development

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