Removal of maternal compensation reveals retinal-specific roles for Pomgnt1 in zebrafish

Maternal-zygotic pomgnt1 knockout zebrafish show retinal abnormalities by 2 mpf despite normal survival and no gross or locomotor deficits through 5 mpf.

• Why it matters: Clinical severity in POMGNT1 disorders varies, and zebrafish retinal degeneration previously seen in pomngt1 KOs may be shaped by maternal transcript compensation and other tissues.
• What they did: The study compared maternal-zygotic pomgnt1 KOs across multiple timepoints, measuring survival, morphology, locomotor activity, behavior, and retinal organization.
• The result: Maternal factors delay retinal pathology onset, and mutants developed a novel wall-hugging behavior pattern that may reflect visual impairment or anxiety, supporting retinal-specific Pomgnt1 vulnerability.

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