[article]
| Titre : |
Distinct early development trajectories in Nf1(±) and Tsc2(±) mouse models of autism |
| Type de document : |
texte imprimé |
| Auteurs : |
Helena FERREIRA, Auteur ; Sofia SANTOS, Auteur ; João MARTINS, Auteur ; Miguel CASTELO-BRANCO, Auteur ; Joana GONÇALVES, Auteur |
| Langues : |
Anglais (eng) |
| Mots-clés : |
Animals Disease Models, Animal Tuberous Sclerosis Complex 2 Protein/genetics Male Female Mice Vocalization, Animal/physiology Autism Spectrum Disorder/genetics/physiopathology Neurofibromatosis 1/genetics/physiopathology Neurofibromin 1/genetics Animals, Newborn Tuberous Sclerosis/genetics Sex Characteristics Maternal Deprivation Mice, Inbred C57BL Asd Developmental milestones Neurofibromatosis type 1 Tuberous sclerosis complex 2 Ultrasonic vocalizations following the European Union Council Directive (2010/63/EU), National Regulations, and ORBEA board of ICNAS (1/2017). Consent for publication: Not applicable. Competing interests: The authors declare no competing interests. |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Autism spectrum disorder (ASD) is a neurodevelopmental condition characterized by deficits in social communication and interaction, and repetitive behaviors. Males are three times more likely to be diagnosed with ASD than females, and sex-dependent alterations in behavior and communication have been reported both in clinical and animal research. Animal models are useful for understanding ASD-related manifestations and their associated neurobiological mechanisms. However, even though ASD is diagnosed during childhood, relatively few animal studies have focused on neonatal development. METHODS: Here, we performed a detailed analysis of neonatal developmental milestones and maternal separation-induced ultrasonic vocalizations (USVs) in two genetic animal models of ASD, neurofibromatosis type 1 (Nf1(±)) and tuberous sclerosis complex 2 (Tsc2(±)). RESULTS: Nf1(±) and Tsc2(±) mice display strikingly distinct developmental profiles regarding motor, strength, and coordination skills. Nf1(±) mouse pups mostly show genotype-related differences, whereas Tsc2(±) mouse pups mainly present sexual dimorphisms. Furthermore, we found several differences regarding the number of USVs, frequency modulation, and temporal and spectral profile. Importantly, Nf1(±) animals tend to present sex- and genotype-dependent differences earlier than the Tsc2(±) mouse pups, suggesting distinct developmental curves between these two animal models. CONCLUSIONS: This study provides a nuanced understanding of how these two ASD models differ in their developmental trajectories. It underscores the importance of studying sex differences and early-life developmental markers, as these could offer crucial insights into ASD's progression and neurobiology. The distinct profiles of these models may help guide more targeted therapeutic strategies in the future. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-025-09624-6 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576 |
in Journal of Neurodevelopmental Disorders > 17 (2025)
[article] Distinct early development trajectories in Nf1(±) and Tsc2(±) mouse models of autism [texte imprimé] / Helena FERREIRA, Auteur ; Sofia SANTOS, Auteur ; João MARTINS, Auteur ; Miguel CASTELO-BRANCO, Auteur ; Joana GONÇALVES, Auteur. Langues : Anglais ( eng) in Journal of Neurodevelopmental Disorders > 17 (2025)
| Mots-clés : |
Animals Disease Models, Animal Tuberous Sclerosis Complex 2 Protein/genetics Male Female Mice Vocalization, Animal/physiology Autism Spectrum Disorder/genetics/physiopathology Neurofibromatosis 1/genetics/physiopathology Neurofibromin 1/genetics Animals, Newborn Tuberous Sclerosis/genetics Sex Characteristics Maternal Deprivation Mice, Inbred C57BL Asd Developmental milestones Neurofibromatosis type 1 Tuberous sclerosis complex 2 Ultrasonic vocalizations following the European Union Council Directive (2010/63/EU), National Regulations, and ORBEA board of ICNAS (1/2017). Consent for publication: Not applicable. Competing interests: The authors declare no competing interests. |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Autism spectrum disorder (ASD) is a neurodevelopmental condition characterized by deficits in social communication and interaction, and repetitive behaviors. Males are three times more likely to be diagnosed with ASD than females, and sex-dependent alterations in behavior and communication have been reported both in clinical and animal research. Animal models are useful for understanding ASD-related manifestations and their associated neurobiological mechanisms. However, even though ASD is diagnosed during childhood, relatively few animal studies have focused on neonatal development. METHODS: Here, we performed a detailed analysis of neonatal developmental milestones and maternal separation-induced ultrasonic vocalizations (USVs) in two genetic animal models of ASD, neurofibromatosis type 1 (Nf1(±)) and tuberous sclerosis complex 2 (Tsc2(±)). RESULTS: Nf1(±) and Tsc2(±) mice display strikingly distinct developmental profiles regarding motor, strength, and coordination skills. Nf1(±) mouse pups mostly show genotype-related differences, whereas Tsc2(±) mouse pups mainly present sexual dimorphisms. Furthermore, we found several differences regarding the number of USVs, frequency modulation, and temporal and spectral profile. Importantly, Nf1(±) animals tend to present sex- and genotype-dependent differences earlier than the Tsc2(±) mouse pups, suggesting distinct developmental curves between these two animal models. CONCLUSIONS: This study provides a nuanced understanding of how these two ASD models differ in their developmental trajectories. It underscores the importance of studying sex differences and early-life developmental markers, as these could offer crucial insights into ASD's progression and neurobiology. The distinct profiles of these models may help guide more targeted therapeutic strategies in the future. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-025-09624-6 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576 |
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Faire une suggestion Affiner la rechercheDistinct early development trajectories in Nf1(±) and Tsc2(±) mouse models of autism / Helena FERREIRA in Journal of Neurodevelopmental Disorders, 17 (2025)
