Partial FAM19A5 deficiency in mice leads to disrupted spine maturation, hyperactivity, and an altered fear response

Shahapal, Anu; Park, Sumi; Yoo, Sangjin; Ma, Shi-Xun; Lee, Jongha; Kwak, Hoyun; Hwang, Jong-Ik; Seong, Jae Young

doi:10.1371/journal.pone.0327493

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Partial FAM19A5 deficiency in mice leads to disrupted spine maturation, hyperactivity, and an altered fear response

Shahapal, Anu;
Park, Sumi;
Yoo, Sangjin;
Ma, Shi-Xun;
Lee, Jongha;
... Seong, Jae Young;
외 2명

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초록

The FAM19A5 polypeptide, encoded by the TAFA5 gene, is evolutionarily conserved among vertebral species. This protein is predominantly expressed in the brain, highlighting its crucial role in the central nervous system. Here, we investigated the potential roles of FAM19A5 in brain development and behavior using a FAM19A5-LacZ KI mouse model. This model exhibited a partial reduction in the FAM19A5 protein level. FAM19A5-LacZ KI mice displayed no significant alterations in gross brain structure but alterations in dendritic spine distribution, with a bias toward immature forms. These mice also had lower body weights. Behavioral tests revealed that compared with their wild-type littermates, FAM19A5-LacZ KI male mice displayed hyperactivity and a delayed innate fear response. These findings suggest that FAM19A5 plays a role in regulating spine maturation and maintenance, thereby contributing to neural connectivity and behavior.

키워드

Isoflurane; Any-maze 6.36 Software; Cfx96 Touchtm Real-time Pcr Detection System; Chemidoc Mp Imaging System; Graphpad Prism 5 Software; Imagej; Revertaid First Strand Cdna Synthesis Kit; Ssoadvanced Universal Sybr Green Supermix; Tcs Sp8; Zen Software; Adipocytokine; Fam19a5; Isoflurane; Unclassified Drug; Altered Fear Response; Anesthesia; Animal Experiment; Animal Model; Animal Tissue; Anxiety; Article; Body Weight; Brain Development; Brain Size; Brain Tissue; Cell Culture; Central Nervous System; Coimmunoprecipitation; Confocal Microscopy; Connectome; Controlled Study; Dendritic Spine; Elevated Plus Maze Test; Enzyme Linked Immunosorbent Assay; Experimental Behavioral Test; Fam19a5 Deficiency; Fear; Female; Food Intake; Gene Expression; Genetic Disorder; Genetic Transfection; Genotype; Hanging Wire Test; Hek293 Cell Line; Hippocampus; Human; Human Cell; Hyperactivity; Locomotion; Male; Marble Burying Test; Maturation; Mouse; Nestlet Shredding Test; Nonhuman; Novel Object Recognition Test; Open Field Test; Optical Density; Pavlovian Fear Conditioning Test; Phenotype; Polyacrylamide Gel Electrophoresis; Protein Expression; Protein Stability; Real Time Polymerase Chain Reaction; Rotarod Test; Spine; Spine Maturation; Tail Suspension Test; Unconditioned Innate Fear Response Test; Western Blotting; Y-maze Test; Animal; Animal Behavior; Brain; Genetics; Hyperkinesia; Metabolism; Pathology; Pathophysiology; Physiology; Animals; Behavior, Animal; Brain; Dendritic Spines; Fear; Hyperkinesis; Male; Mice; ALZHEIMERS-DISEASE; FAMILY

제목: Partial FAM19A5 deficiency in mice leads to disrupted spine maturation, hyperactivity, and an altered fear response

저자: Shahapal, Anu; Park, Sumi; Yoo, Sangjin; Ma, Shi-Xun; Lee, Jongha; Kwak, Hoyun; Hwang, Jong-Ik; Seong, Jae Young

DOI: 10.1371/journal.pone.0327493

발행일: 2025-08-05

유형: Article

저널명: PLoS One

권: 20

호: 8