A postnatal role for embryonic myosin revealed by MYH3 mutations that alter TGFÎ2 signaling and cause autosomal dominant spondylocarpotarsal synostosis

نویسندگان

  • Dorothy K. Grange
  • Jennifer Zieba
  • Wenjuan Zhang
  • Jessica X. Chong
  • Kimberly N. Forlenza
  • Jorge H. Martin
  • Kelly Heard
  • Merlin G. Butler
  • Tjitske Kleefstra
  • Ralph S. Lachman
  • Deborah Nickerson
  • Michael Regnier
  • Daniel H. Cohn
  • Michael Bamshad
  • Deborah Krakow
چکیده

A postnatal role for embryonic myosin revealed by MYH3 mutations that alter TGFβ signaling and cause autosomal dominant spondylocarpotarsal synostosis.

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A postnatal role for embryonic myosin revealed by MYH3 mutations that alter TGFβ signaling and cause autosomal dominant spondylocarpotarsal synostosis

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Spondylocarpotarsal synostosis (SCT) is an autosomal recessive disorder characterized by progressive vertebral fusions and caused by loss of function mutations in Filamin B (FLNB). FLNB acts as a signaling scaffold by linking the actin cytoskleteon to signal transduction systems, yet the disease mechanisms for SCT remain unclear. Employing a Flnb knockout mouse, we found morphologic and molecul...

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Urolithiasis in a child with Spondylocarpotarsal Synostosis Syndrome: A Co-Incidence.

Spondylocarpotarsal synostosis syndrome (SSS) is an autosomal recessive condition which is characterized by short stature, a carpotarsal coalition and a vertebral fusion, but without any rib anomaly. We are presenting a 7- year- old boy, who had uroliathiasis with the spondylocarpotarsal synostosis syndrome. This association, to the best of our knowledge, has not been reported so far.

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Background: We examine the GA-repeat core promoters of MECOM and GABRA3 in human embryonic kidney-293 cell line and show that those GA-repeats have promoter activity,and those different alleles of the repeats can significantly alter gene expression.We propose a novel role for GA-repeat core promoters to regulate gene expression in the genes involved in development and evolution. Materials and M...

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تاریخ انتشار 2017