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Nomo1 deficiency causes autism-like behavior in zebrafish
Qi Zhang1, Fei Li1, Tingting Li1
1Translational Medical Center for Development and Disease, Shanghai Key Laboratory of Birth Defect Prevention and Control, NHC Key Laboratory of Neonatal Diseases, Institute of Pediatrics, Children's Hospital of Fudan University, National Children's Medical Center, 210013, Shanghai, China.
EMBO Reports
|January 22, 2024
Summary
NOMO1 gene deficiency in zebrafish causes neuropsychiatric behaviors and brain inflammation. Melatonin treatment improved behaviors and reduced inflammation, suggesting its therapeutic potential for related disorders.
Area of Science:
- Neuroscience
- Genetics
- Pharmacology
Background:
- Neuropsychiatric disorders present complex, overlapping symptoms, hindering diagnosis and treatment.
- NOMO1, located at 16p13.11, a region linked to neuropsychiatric diseases, is a candidate gene requiring functional investigation.
- Understanding NOMO1's role can elucidate pathogenic mechanisms and inform therapeutic strategies for co-morbid conditions.
Purpose of the Study:
- To investigate the function of the NOMO1 gene in vivo.
- To characterize the behavioral and neurological phenotypes of NOMO1-deficient zebrafish.
- To explore the therapeutic potential of melatonin for NOMO1 deficiency-related neuropsychiatric symptoms.
Main Methods:
- Generation of nomo1 knockout (nomo1-/-) zebrafish model.
- Assessment of brain and neuronal development, apoptosis, and inflammation pathways.
- Behavioral testing in adult nomo1-/- zebrafish, including locomotor activity, social interaction, and stereotypic behaviors.
- Measurement of melatonin levels and evaluation of melatonin treatment effects.
Main Results:
- Nomo1 mutants displayed abnormal brain development, activated apoptosis, and inflammation.
- Adult nomo1-/- zebrafish exhibited hyperactivity, social deficits, and repetitive behaviors.
- Reduced melatonin levels were observed, alongside affected habenular nucleus and pineal gland.
- Melatonin administration ameliorated locomotor hyperactivity, stereotypic behaviors, and brain inflammation.
Conclusions:
- NOMO1 deficiency leads to significant neurodevelopmental and behavioral abnormalities in zebrafish.
- Melatonin supplementation demonstrates efficacy in reversing key behavioral and inflammatory phenotypes.
- Melatonin may serve as a viable therapeutic intervention for neuropsychiatric disorders linked to NOMO1 deficiency.
