Pla2g6 Deficiency Induces Neuronal Precursor Apoptosis During Neurodevelopment

Phospholipase A2 group VI (PLA2G6) regulates phospholipid remodeling and cellular homeostasis, and its mutations cause neurodegenerative disorders, including neurodegeneration with brain iron accumulation and PLA2G6-associated parkinsonism (PARK14). Although many cases present in adulthood, a substantial subset shows early onset, indicating that PLA2G6 dysfunction can affect neuronal systems during developmental stages. However, whether PLA2G6 directly regulates early neurogenesis remains undefined. Here, using zebrafish embryos, we investigated the role of Pla2g6 during neural development through loss- and gain-of-function approaches. pla2g6 is dynamically expressed during embryogenesis, with enrichment in the developing central nervous system during neurogenesis. CRISPR/Cas9-mediated Pla2g6 deficiency did not alter neural progenitor formation but significantly reduced neuronal precursors. Expression of the disease-associated PLA2G6 D331Y variant phenocopied this effect, confirming that the observed phenotype results from loss of Pla2g6 function. The reduction in neuronal precursors occurred without changes in proliferation but was accompanied by a marked increase in apoptosis, identifying neuronal precursor cell death as the primary mechanism. Under oxidative stress conditions, Pla2g6 overexpression reduced neuronal apoptosis, whereas Pla2g6 deficiency markedly enhanced reactive oxygen species -induced apoptosis. These findings establish Pla2g6 as a regulator of oxidative stress-associated apoptotic signaling during neurogenesis. Together, these results define Pla2g6 as a stage-specific determinant of neuronal precursor survival, linking lipid homeostasis and oxidative stress control to early neural development. This study establishes a developmental framework for PLA2G6-associated disorders and positions impaired neuronal precursor survival as a contributing mechanism underlying disease onset.

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Publication Details

Journal
International Journal of Molecular Sciences
Published
2026-05-11
DOI
https://doi.org/10.3390/ijms27104280
Primary Topic
Neurological diseases and metabolism
Type
article
Field-Weighted Citation Impact
0.00

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article

Pla2g6 Deficiency Induces Neuronal Precursor Apoptosis During Neurodevelopment

Yi‐Chieh Chen, Yi‐Chuan Cheng, Yang-Jin Shen, Ting-Chen Hsu et al.
International Journal of Molecular Sciences
Neurological diseases and metabolism
article

Pla2g6 Deficiency Induces Neuronal Precursor Apoptosis During Neurodevelopment

Yi‐Chieh Chen, Yi‐Chuan Cheng, Yang-Jin Shen, Ting-Chen Hsu, Han-Fang Liu
article en

Abstract

Phospholipase A2 group VI (PLA2G6) regulates phospholipid remodeling and cellular homeostasis, and its mutations cause neurodegenerative disorders, including neurodegeneration with brain iron accumulation and PLA2G6-associated parkinsonism (PARK14). Although many cases present in adulthood, a substantial subset shows early onset, indicating that PLA2G6 dysfunction can affect neuronal systems during developmental stages. However, whether PLA2G6 directly regulates early neurogenesis remains undefined. Here, using zebrafish embryos, we investigated the role of Pla2g6 during neural development through loss- and gain-of-function approaches. pla2g6 is dynamically expressed during embryogenesis, with enrichment in the developing central nervous system during neurogenesis. CRISPR/Cas9-mediated Pla2g6 deficiency did not alter neural progenitor formation but significantly reduced neuronal precursors. Expression of the disease-associated PLA2G6 D331Y variant phenocopied this effect, confirming that the observed phenotype results from loss of Pla2g6 function. The reduction in neuronal precursors occurred without changes in proliferation but was accompanied by a marked increase in apoptosis, identifying neuronal precursor cell death as the primary mechanism. Under oxidative stress conditions, Pla2g6 overexpression reduced neuronal apoptosis, whereas Pla2g6 deficiency markedly enhanced reactive oxygen species -induced apoptosis. These findings establish Pla2g6 as a regulator of oxidative stress-associated apoptotic signaling during neurogenesis. Together, these results define Pla2g6 as a stage-specific determinant of neuronal precursor survival, linking lipid homeostasis and oxidative stress control to early neural development. This study establishes a developmental framework for PLA2G6-associated disorders and positions impaired neuronal precursor survival as a contributing mechanism underlying disease onset.

International Journal of Molecular SciencesVol. 27(10)
Chang Gung University (TW), Chang Gung Memorial Hospital (TW), Linkou Chang Gung Memorial Hospital (TW)
Chang Gung Memorial Hospital, Chang Gung University, Chang Gung Medical Foundation, National Science and Technology Council
Openalex Percentile: Top 6%
Neurological diseases and metabolism
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