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Fixed Action Patterns01:06

Fixed Action Patterns

A fixed action pattern (FAP) is a specific, hard-wired sequence of behaviors that occurs in response to an external stimulus, called a sign stimulus. The behavior is “fixed” because it is essentially unchangeable—proceeding similarly across individuals of a species every time it occurs.

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Related Experiment Video

Updated: Jun 19, 2026

Analysis of Skeletal Muscle Defects in Larval Zebrafish by Birefringence and Touch-evoke Escape Response Assays
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Analysis of Skeletal Muscle Defects in Larval Zebrafish by Birefringence and Touch-evoke Escape Response Assays

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ap4b1 -/- zebrafish demonstrate morphological and motor abnormalities.

Helena Rosengarten1,2, Angelica D'Amore1, Hyo-Min Kim1

  • 1Movement Disorders Program, Department of Neurology & The F.M. Kirby Neurobiology Center, Boston Children's Hospital, Harvard Medical School, 300 Longwood Avenue, Boston, MA 02115, United States.

Human Molecular Genetics
|April 23, 2025
PubMed
Summary

A new zebrafish model for Hereditary Spastic Paraplegia type 47 (SPG47) shows motor deficits and developmental issues, aiding research into AP-4 deficiency.

Keywords:
adaptor protein complex 4axon outgrowthhereditary spastic paraplegiazebrafish

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Area of Science:

  • Neuroscience
  • Genetics
  • Developmental Biology

Background:

  • Hereditary spastic paraplegia type 47 (SPG47) results from AP4B1 gene variants, causing neurodevelopmental and motor impairments.
  • Understanding the role of AP4B1 in neurodevelopment is crucial for SPG47 research.

Purpose of the Study:

  • To create and characterize a zebrafish model for SPG47.
  • To investigate the function of ap4b1 in zebrafish neurodevelopment and motor control.

Main Methods:

  • CRISPR/Cas9 gene-editing was used to generate ap4b1-/- zebrafish.
  • Evaluated motor function, behavior (locomotion, light-dark transitions, coiling), axonal length, and seizure susceptibility.

Main Results:

  • ap4b1-/- zebrafish displayed reduced motor neuron axonal length and impaired motor function.
  • Observed developmental defects including craniofacial abnormalities and spinal curvature.
  • Larvae showed altered activity patterns and reduced survival under stress.

Conclusions:

  • The ap4b1-/- zebrafish model effectively recapitulates key SPG47 phenotypes.
  • This model serves as a valuable platform for studying AP-4 deficiency and SPG47 mechanisms.