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Patient-derived AMOTL1 mutations lead to defective cell migration and tissue development.

Jiaqian Luo1, Ruxin Jin1, Fang Geng2,3

  • 1Institute of Pediatrics, Children's Hospital of Fudan University, and Shanghai Key Laboratory of Medical Epigenetics, International Co-laboratory of Medical Epigenetics and Metabolism, Institutes of Biomedical Sciences, Shanghai Medical College, Fudan University, Shanghai, China.

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Summary

Mutations in Angiomotin-like 1 (AMOTL1) disrupt its interaction with Tankyrase, causing protein buildup and inhibiting cell migration, leading to congenital defects.

Keywords:
Angiomotin-like 1RNF146TNKS1/2proteasomes

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Area of Science:

  • Molecular Biology
  • Developmental Biology
  • Cell Biology

Background:

  • Angiomotin-like 1 (AMOTL1) is crucial for organogenesis and development by regulating cell junctions, polarity, and migration.
  • Two hotspot mutations (Arg157 and Pro160) in AMOTL1 are linked to congenital defects in multiple families.
  • The pathogenic mechanism of these AMOTL1 mutations remained unclear.

Purpose of the Study:

  • To elucidate the molecular and cellular mechanisms underlying congenital defects caused by AMOTL1 mutations.
  • To investigate the impact of Arg157 and Pro160 mutations on AMOTL1 protein stability and function.
  • To explore the consequences of AMOTL1 mutations in a zebrafish model of embryonic development.

Main Methods:

  • Investigated the interaction of AMOTL1 mutants (R157C, P160L) with Tankyrase 1/2 (TNKS1/2) and Ring finger protein 146 (RNF146).
  • Assessed the effects of mutations on AMOTL1 poly ADP-ribosylation, ubiquitination, and proteasomal degradation.
  • Analyzed the impact of AMOTL1 mutants on cell junction and focal adhesion integrity, and cell migration.
  • Examined craniofacial, cardiac, and skeletal muscle development in zebrafish embryos expressing the R157C mutant.

Main Results:

  • R157C and P160L AMOTL1 mutants failed to interact with TNKS1/2 and RNF146.
  • Mutants were resistant to PARylation, ubiquitination, and proteasomal degradation, leading to cytoplasmic accumulation.
  • Accumulated AMOTL1 mutants disrupted cell junctions and focal adhesions, inhibiting cell migration velocity and persistence.
  • Expression of R157C mutant in zebrafish caused craniofacial, cardiac, and skeletal muscle defects.

Conclusions:

  • AMOTL1 mutations impair Tankyrase binding, leading to protein stabilization and disruption of cellular processes.
  • These molecular and cellular defects underlie the observed congenital abnormalities.
  • The study clarifies the pathogenic mechanism of AMOTL1 mutations in human development.