[HTML][HTML] Nuclear envelope protein Lem2 is required for mouse development and regulates MAP and AKT kinases

O Tapia, LG Fong, MD Huber, SG Young, L Gerace - PloS one, 2015 - journals.plos.org
O Tapia, LG Fong, MD Huber, SG Young, L Gerace
PloS one, 2015journals.plos.org
The nuclear lamina, along with associated nuclear membrane proteins, is a nexus for
regulating signaling in the nucleus. Numerous human diseases arise from mutations in
lamina proteins, and experimental models for these disorders have revealed aberrant
regulation of various signaling pathways. Previously, we reported that the inner nuclear
membrane protein Lem2, which is expressed at high levels in muscle, promotes the
differentiation of cultured myoblasts by attenuating ERK signaling. Here, we have analyzed …
The nuclear lamina, along with associated nuclear membrane proteins, is a nexus for regulating signaling in the nucleus. Numerous human diseases arise from mutations in lamina proteins, and experimental models for these disorders have revealed aberrant regulation of various signaling pathways. Previously, we reported that the inner nuclear membrane protein Lem2, which is expressed at high levels in muscle, promotes the differentiation of cultured myoblasts by attenuating ERK signaling. Here, we have analyzed mice harboring a disrupted allele for the Lem2 gene (Lemd2). No gross phenotypic defects were seen in heterozygotes, although muscle regeneration induced by cardiotoxin was delayed. By contrast, homozygous Lemd2 knockout mice died by E11.5. Although many normal morphogenetic hallmarks were observed in E10.5 knockout embryos, most tissues were substantially reduced in size. This was accompanied by activation of multiple MAP kinases (ERK1/2, JNK, p38) and AKT. Knockdown of Lem2 expression in C2C12 myoblasts also led to activation of MAP kinases and AKT. These findings indicate that Lemd2 plays an essential role in mouse embryonic development and that it is involved in regulating several signaling pathways. Since increased MAP kinase and AKT/mTORC signaling is found in other animal models for diseases linked to nuclear lamina proteins, LEMD2 should be considered to be another candidate gene for human disease.
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