7 recherche sur le mot-clé 'Ultrasonic vocalizations'

Courtship and distress ultrasonic vocalizations are altered in a mouse model of Angelman syndrome / Caleigh D. GUOYNES in Journal of Neurodevelopmental Disorders, 17 (2025)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 17 (2025) Titre : Courtship and distress ultrasonic vocalizations are altered in a mouse model of Angelman syndrome Type de document : texte imprimé Auteurs : Caleigh D. GUOYNES, Auteur ; Grace PAVALKO, Auteur ; Michael S. SIDOROV, Auteur Langues : Anglais (eng) Mots-clés : Angelman syndrome  Behavior  Ube3a  Ultrasonic vocalizations  protocols were approved by and performed in accordance with the relevant  guidelines and regulations of the Institutional Animal Care and Use Committee of  Children’s National Hospital and were in compliance with the US National Research  Council’s Guide for the Care and Use of Laboratory Animals, the US Public Health  Service’s Policy on Humane Care and Use of Laboratory Animals, and Guide for the  Care and Use of Laboratory Animals. Consent for publication: Not applicable.  Competing interests: The authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: Angelman syndrome (AS) is a single-gene neurodevelopmental disorder caused by loss of function of the maternal copy of the UBE3A gene. Nearly all individuals with AS lack speech, resulting in major impacts on daily life for patients and caregivers. To evaluate new therapies for AS, it is crucial to have a mouse model that characterizes meaningful clinical features. Vocalizations are used in many contexts in mice, including pup retrieval, social interactions, courtship, and distress. Previous work in the Ube3a(m−/p+) mouse model of AS found abnormalities in the number of ultrasonic vocalizations (USVs) mice produced during pup isolation and same-sex social interactions. Here, we evaluated Ube3a(m−/p+) vocalizations during courtship and distress. Quantifying USVs in these contexts enables comparison of USVs in social (courtship) and non-social (distress) settings. In addition, we assessed the utility of incorporating USV testing into existing Ube3a(m−/p+) mouse behavioral assessments used to evaluate potential AS treatments. METHODS: We used multiple behavioral paradigms to evaluate courtship vocalizations and tail suspension tests to evaluate distress vocalizations in adult wild-type (WT) and Ube3a(m−/p+) littermates, and quantified USV properties using the program DeepSqueak. A subset of mice also performed an established Ube3a(m−/p+) behavioral battery that included rotarod, open field, marble burying, and nest building. We used principal component analysis to evaluate the value of USV testing in this cohort in the context of other behaviors. RESULTS: In both social courtship and nonsocial distress behavioral paradigms, Ube3a(m−/p+) mice made fewer USVs compared to WT mice. Spectral properties of USVs were abnormal in Ube3a(m−/p+) mice on courtship tests but mostly typical on distress tests. Including USVs in the Ube3a(m−/p+) mouse behavior battery increased the distance between Ube3a(m−/p+) and WT clusters in principal component space. CONCLUSIONS: Ube3a(m−/p+) mice have disrupted USV production in social and nonsocial contexts. Spectral properties of USVs are most impacted in the social courtship context. Adding USVs to the Ube3a(m−/p+) behavior battery may improve sensitivity to detect group differences and changes in communication. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s11689-025-09648-y. En ligne : https://dx.doi.org/10.1186/s11689-025-09648-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576 [article] Courtship and distress ultrasonic vocalizations are altered in a mouse model of Angelman syndrome [texte imprimé] / Caleigh D. GUOYNES, Auteur ; Grace PAVALKO, Auteur ; Michael S. SIDOROV, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 17 (2025) Mots-clés : Angelman syndrome  Behavior  Ube3a  Ultrasonic vocalizations  protocols were approved by and performed in accordance with the relevant  guidelines and regulations of the Institutional Animal Care and Use Committee of  Children’s National Hospital and were in compliance with the US National Research  Council’s Guide for the Care and Use of Laboratory Animals, the US Public Health  Service’s Policy on Humane Care and Use of Laboratory Animals, and Guide for the  Care and Use of Laboratory Animals. Consent for publication: Not applicable.  Competing interests: The authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: Angelman syndrome (AS) is a single-gene neurodevelopmental disorder caused by loss of function of the maternal copy of the UBE3A gene. Nearly all individuals with AS lack speech, resulting in major impacts on daily life for patients and caregivers. To evaluate new therapies for AS, it is crucial to have a mouse model that characterizes meaningful clinical features. Vocalizations are used in many contexts in mice, including pup retrieval, social interactions, courtship, and distress. Previous work in the Ube3a(m−/p+) mouse model of AS found abnormalities in the number of ultrasonic vocalizations (USVs) mice produced during pup isolation and same-sex social interactions. Here, we evaluated Ube3a(m−/p+) vocalizations during courtship and distress. Quantifying USVs in these contexts enables comparison of USVs in social (courtship) and non-social (distress) settings. In addition, we assessed the utility of incorporating USV testing into existing Ube3a(m−/p+) mouse behavioral assessments used to evaluate potential AS treatments. METHODS: We used multiple behavioral paradigms to evaluate courtship vocalizations and tail suspension tests to evaluate distress vocalizations in adult wild-type (WT) and Ube3a(m−/p+) littermates, and quantified USV properties using the program DeepSqueak. A subset of mice also performed an established Ube3a(m−/p+) behavioral battery that included rotarod, open field, marble burying, and nest building. We used principal component analysis to evaluate the value of USV testing in this cohort in the context of other behaviors. RESULTS: In both social courtship and nonsocial distress behavioral paradigms, Ube3a(m−/p+) mice made fewer USVs compared to WT mice. Spectral properties of USVs were abnormal in Ube3a(m−/p+) mice on courtship tests but mostly typical on distress tests. Including USVs in the Ube3a(m−/p+) mouse behavior battery increased the distance between Ube3a(m−/p+) and WT clusters in principal component space. CONCLUSIONS: Ube3a(m−/p+) mice have disrupted USV production in social and nonsocial contexts. Spectral properties of USVs are most impacted in the social courtship context. Adding USVs to the Ube3a(m−/p+) behavior battery may improve sensitivity to detect group differences and changes in communication. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s11689-025-09648-y. En ligne : https://dx.doi.org/10.1186/s11689-025-09648-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576

Distinct early development trajectories in Nf1(±) and Tsc2(±) mouse models of autism / Helena FERREIRA in Journal of Neurodevelopmental Disorders, 17 (2025)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 17 (2025) 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 [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

Socio-affective communication in Tph2-deficient rat pups: communal nesting aggravates growth retardation despite ameliorating maternal affiliation deficits / Judith R. HOMBERG ; Laura BOREGGIO ; Marta C F SAMINA ; Rogério C.R. CASTRO ; Sharon M. KOLK ; Natalia ALENINA ; Michael BADER ; Jinye DAI ; Markus WÖHR in Molecular Autism, 15 (2024)  

Public ISBD [article]  inMolecular Autism > 15 (2024) . - 50 Titre : Socio-affective communication in Tph2-deficient rat pups: communal nesting aggravates growth retardation despite ameliorating maternal affiliation deficits Type de document : texte imprimé Auteurs : Judith R. HOMBERG, Auteur ; Laura BOREGGIO, Auteur ; Marta C F SAMINA, Auteur ; Rogério C.R. CASTRO, Auteur ; Sharon M. KOLK, Auteur ; Natalia ALENINA, Auteur ; Michael BADER, Auteur ; Jinye DAI, Auteur ; Markus WÖHR, Auteur Article en page(s) : 50 Langues : Anglais (eng) Mots-clés : Animals  *Tryptophan Hydroxylase/deficiency/genetics/metabolism  Female  Male  *Maternal Behavior  Rats  Vocalization, Animal  Social Behavior  Nesting Behavior  Growth Disorders/etiology  Animal Communication  Animals, Newborn  Behavior, Animal  Autism  Neurodevelopmental disorders  Serotonin  Tryptophan hydroxylase 2  Ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : BACKGROUND: A lack of serotonin (also known as 5-hydroxytryptamine, 5-HT) in the brain due to deficiency of the rate-limiting enzyme in 5-HT synthesis, tryptophan hydroxylase 2 (TPH2), was recently reported to result in impaired maternal affiliation across species, including mice, rats, and monkeys. In rodents, this was reflected in a lack of preference for maternal odors and reduced levels of isolation-induced ultrasonic vocalizations (USV), possibly contributing to a severe growth retardation phenotype. METHODS: Here, we tested whether growth retardation, maternal affiliation deficits, and/or impairments in socio-affective communication caused by Tph2 deficiency can be rescued through early social enrichment in rats. To this aim, we compared male and female Tph2(-/-) knockout and Tph2(+/-) heterozygous rat pups to Tph2(+/+) wildtype littermate controls, with litters being randomly assigned to standard nesting (SN; one mother with her litter) or communal nesting (CN; two mothers with their two litters). RESULTS: Our results show that Tph2 deficiency causes severe growth retardation, together with moderate impairments in somatosensory reflexes and thermoregulatory capabilities, partially aggravated by CN. Tph2 deficiency further led to deficits in socio-affective communication, as evidenced by reduced emission of isolation-induced USV, associated with changes in acoustic features, clustering of subtypes, and temporal organization. Although CN did not rescue the impairments in socio-affective communication, CN ameliorated the maternal affiliation deficit caused by Tph2 deficiency in the homing test. To close the communicative loop between mother and pup, we assessed maternal preference and showed that mothers display a preference for Tph2(+/+) controls over Tph2(-/-) pups, particularly under CN conditions. This is consistent with the aggravated growth phenotype in Tph2(-/-) pups exposed to the more competitive CN environment. CONCLUSION: Together, this indicates that CN aggravates growth retardation despite ameliorating maternal affiliation deficits in Tph2-deficient rat pups, possibly due to reduced and acoustically altered isolation-induced USV, hindering efficient socio-affective communication between mother and pup. En ligne : https://dx.doi.org/10.1186/s13229-024-00629-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=555 [article] Socio-affective communication in Tph2-deficient rat pups: communal nesting aggravates growth retardation despite ameliorating maternal affiliation deficits [texte imprimé] / Judith R. HOMBERG, Auteur ; Laura BOREGGIO, Auteur ; Marta C F SAMINA, Auteur ; Rogério C.R. CASTRO, Auteur ; Sharon M. KOLK, Auteur ; Natalia ALENINA, Auteur ; Michael BADER, Auteur ; Jinye DAI, Auteur ; Markus WÖHR, Auteur . - 50. Langues : Anglais (eng) in Molecular Autism > 15 (2024) . - 50 Mots-clés : Animals  *Tryptophan Hydroxylase/deficiency/genetics/metabolism  Female  Male  *Maternal Behavior  Rats  Vocalization, Animal  Social Behavior  Nesting Behavior  Growth Disorders/etiology  Animal Communication  Animals, Newborn  Behavior, Animal  Autism  Neurodevelopmental disorders  Serotonin  Tryptophan hydroxylase 2  Ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : BACKGROUND: A lack of serotonin (also known as 5-hydroxytryptamine, 5-HT) in the brain due to deficiency of the rate-limiting enzyme in 5-HT synthesis, tryptophan hydroxylase 2 (TPH2), was recently reported to result in impaired maternal affiliation across species, including mice, rats, and monkeys. In rodents, this was reflected in a lack of preference for maternal odors and reduced levels of isolation-induced ultrasonic vocalizations (USV), possibly contributing to a severe growth retardation phenotype. METHODS: Here, we tested whether growth retardation, maternal affiliation deficits, and/or impairments in socio-affective communication caused by Tph2 deficiency can be rescued through early social enrichment in rats. To this aim, we compared male and female Tph2(-/-) knockout and Tph2(+/-) heterozygous rat pups to Tph2(+/+) wildtype littermate controls, with litters being randomly assigned to standard nesting (SN; one mother with her litter) or communal nesting (CN; two mothers with their two litters). RESULTS: Our results show that Tph2 deficiency causes severe growth retardation, together with moderate impairments in somatosensory reflexes and thermoregulatory capabilities, partially aggravated by CN. Tph2 deficiency further led to deficits in socio-affective communication, as evidenced by reduced emission of isolation-induced USV, associated with changes in acoustic features, clustering of subtypes, and temporal organization. Although CN did not rescue the impairments in socio-affective communication, CN ameliorated the maternal affiliation deficit caused by Tph2 deficiency in the homing test. To close the communicative loop between mother and pup, we assessed maternal preference and showed that mothers display a preference for Tph2(+/+) controls over Tph2(-/-) pups, particularly under CN conditions. This is consistent with the aggravated growth phenotype in Tph2(-/-) pups exposed to the more competitive CN environment. CONCLUSION: Together, this indicates that CN aggravates growth retardation despite ameliorating maternal affiliation deficits in Tph2-deficient rat pups, possibly due to reduced and acoustically altered isolation-induced USV, hindering efficient socio-affective communication between mother and pup. En ligne : https://dx.doi.org/10.1186/s13229-024-00629-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=555

Autistic-relevant behavioral phenotypes of a mouse model of cyclin-dependent kinase-like 5 deficiency disorder / Oceane COIFFARD ; Celeste FERRAGUTO ; Athanasios MANOLIS ; Elisabetta CIANI ; Susanna PIETROPAOLO in Autism Research, 17-9 (September 2024)  

Public ISBD [article]  inAutism Research > 17-9 (September 2024) . - p.1742-1759 Titre : Autistic-relevant behavioral phenotypes of a mouse model of cyclin-dependent kinase-like 5 deficiency disorder Type de document : texte imprimé Auteurs : Oceane COIFFARD, Auteur ; Celeste FERRAGUTO, Auteur ; Athanasios MANOLIS, Auteur ; Elisabetta CIANI, Auteur ; Susanna PIETROPAOLO, Auteur Article en page(s) : p.1742-1759 Langues : Anglais (eng) Mots-clés : acoustic startle  CDD  mouse pups  neurodevelopmental disorders  social behaviors  social interaction  ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : Abstract Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a neurodevelopmental disease caused by mutations in the X-linked CDKL5 gene and characterized by early-onset epilepsy, intellectual disability, and autistic features. To date, the etiological mechanisms underlying CDD are largely unknown and no effective therapies are available. The Cdkl5 knock-out (KO) mouse has been broadly employed in preclinical studies on CDD; Cdkl5-KO mice display neurobehavioral abnormalities recapitulating most CDD symptoms, including alterations in motor, sensory, cognitive, and social abilities. However, most available preclinical studies have been carried out on adult Cdkl5-KO mice, so little is known about the phenotypic characteristics of this model earlier during development. Furthermore, major autistic-relevant phenotypes, for example, social and communication deficits, have been poorly investigated and mostly in male mutants. Here, we assessed the autistic-relevant behavioral phenotypes of Cdkl5-KO mice during the first three post-natal weeks and in adulthood. Males and females were tested, the latter including both heterozygous and homozygous mutants. Cdkl5 mutant pups showed qualitative and quantitative alterations in ultrasonic communication, detected first at 2 weeks of age and confirmed later in adulthood. Increased levels of anxiety-like behaviors were observed in mutants at 3 weeks and in adulthood, when stereotypies, reduced social interaction and memory deficits were also observed. These behavioral effects of the mutation were evident in both sexes, being more marked and varied in homozygous than heterozygous females. These findings provide novel evidence for the autistic-relevant behavioral profile of the Cdkl5 mouse model, thus supporting its use in future preclinical studies investigating CDD pathology and autism spectrum disorders. En ligne : https://doi.org/10.1002/aur.3226 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=535 [article] Autistic-relevant behavioral phenotypes of a mouse model of cyclin-dependent kinase-like 5 deficiency disorder [texte imprimé] / Oceane COIFFARD, Auteur ; Celeste FERRAGUTO, Auteur ; Athanasios MANOLIS, Auteur ; Elisabetta CIANI, Auteur ; Susanna PIETROPAOLO, Auteur . - p.1742-1759. Langues : Anglais (eng) in Autism Research > 17-9 (September 2024) . - p.1742-1759 Mots-clés : acoustic startle  CDD  mouse pups  neurodevelopmental disorders  social behaviors  social interaction  ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : Abstract Cyclin-dependent kinase-like 5 (CDKL5) deficiency disorder (CDD) is a neurodevelopmental disease caused by mutations in the X-linked CDKL5 gene and characterized by early-onset epilepsy, intellectual disability, and autistic features. To date, the etiological mechanisms underlying CDD are largely unknown and no effective therapies are available. The Cdkl5 knock-out (KO) mouse has been broadly employed in preclinical studies on CDD; Cdkl5-KO mice display neurobehavioral abnormalities recapitulating most CDD symptoms, including alterations in motor, sensory, cognitive, and social abilities. However, most available preclinical studies have been carried out on adult Cdkl5-KO mice, so little is known about the phenotypic characteristics of this model earlier during development. Furthermore, major autistic-relevant phenotypes, for example, social and communication deficits, have been poorly investigated and mostly in male mutants. Here, we assessed the autistic-relevant behavioral phenotypes of Cdkl5-KO mice during the first three post-natal weeks and in adulthood. Males and females were tested, the latter including both heterozygous and homozygous mutants. Cdkl5 mutant pups showed qualitative and quantitative alterations in ultrasonic communication, detected first at 2 weeks of age and confirmed later in adulthood. Increased levels of anxiety-like behaviors were observed in mutants at 3 weeks and in adulthood, when stereotypies, reduced social interaction and memory deficits were also observed. These behavioral effects of the mutation were evident in both sexes, being more marked and varied in homozygous than heterozygous females. These findings provide novel evidence for the autistic-relevant behavioral profile of the Cdkl5 mouse model, thus supporting its use in future preclinical studies investigating CDD pathology and autism spectrum disorders. En ligne : https://doi.org/10.1002/aur.3226 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=535

17-beta estradiol increases parvalbumin levels in Pvalb heterozygous mice and attenuates behavioral phenotypes with relevance to autism core symptoms / Federica FILICE in Molecular Autism, 9 (2018)  

Public ISBD [article]  inMolecular Autism > 9 (2018) . - 15p. Titre : 17-beta estradiol increases parvalbumin levels in Pvalb heterozygous mice and attenuates behavioral phenotypes with relevance to autism core symptoms Type de document : texte imprimé Auteurs : Federica FILICE, Auteur ; Emanuel LAUBER, Auteur ; K.J. VORCKEL, Auteur ; M. WOHR, Auteur ; Beat SCHWALLER, Auteur Article en page(s) : 15p. Langues : Anglais (eng) Mots-clés : 17-beta estradiol  Asd  Estradiol treatment  Excitation/inhibition balance  Parvalbumin  Social behavior  Ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : Background: Autism spectrum disorder (ASD) is a group of neurodevelopmental disorders characterized by two core symptoms: impaired social interaction and communication, and restricted, repetitive behaviors and interests. The pathophysiology of ASD is not yet fully understood, due to a plethora of genetic and environmental risk factors that might be associated with or causal for ASD. Recent findings suggest that one putative convergent pathway for some forms of ASD might be the downregulation of the calcium-binding protein parvalbumin (PV). PV-deficient mice (PV-/-, PV+/-), as well as Shank1-/-, Shank3-/-, and VPA mice, which show behavioral deficits relevant to all human ASD core symptoms, are all characterized by lower PV expression levels. Methods: Based on the hypothesis that PV expression might be increased by 17-beta estradiol (E2), PV+/- mice were treated with E2 from postnatal days 5-15 and ASD-related behavior was tested between postnatal days 25 and 31. Results: PV expression levels were significantly increased after E2 treatment and, concomitantly, sociability deficits in PV+/- mice in the direct reciprocal social interaction and the 3-chamber social approach assay, as well as repetitive behaviors, were attenuated. E2 treatment of PV+/+ mice did not increase PV levels and had detrimental effects on sociability and repetitive behavior. In PV-/- mice, E2 obviously did not affect PV levels; tested behaviors were not different from the ones in vehicle-treated PV-/- mice. Conclusion: Our results suggest that the E2-linked amelioration of ASD-like behaviors is specifically occurring in PV+/- mice, indicating that PV upregulation is required for the E2-mediated rescue of ASD-relevant behavioral impairments. En ligne : http://dx.doi.org/10.1186/s13229-018-0199-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354 [article] 17-beta estradiol increases parvalbumin levels in Pvalb heterozygous mice and attenuates behavioral phenotypes with relevance to autism core symptoms [texte imprimé] / Federica FILICE, Auteur ; Emanuel LAUBER, Auteur ; K.J. VORCKEL, Auteur ; M. WOHR, Auteur ; Beat SCHWALLER, Auteur . - 15p. Langues : Anglais (eng) in Molecular Autism > 9 (2018) . - 15p. Mots-clés : 17-beta estradiol  Asd  Estradiol treatment  Excitation/inhibition balance  Parvalbumin  Social behavior  Ultrasonic vocalizations Index. décimale : PER Périodiques Résumé : Background: Autism spectrum disorder (ASD) is a group of neurodevelopmental disorders characterized by two core symptoms: impaired social interaction and communication, and restricted, repetitive behaviors and interests. The pathophysiology of ASD is not yet fully understood, due to a plethora of genetic and environmental risk factors that might be associated with or causal for ASD. Recent findings suggest that one putative convergent pathway for some forms of ASD might be the downregulation of the calcium-binding protein parvalbumin (PV). PV-deficient mice (PV-/-, PV+/-), as well as Shank1-/-, Shank3-/-, and VPA mice, which show behavioral deficits relevant to all human ASD core symptoms, are all characterized by lower PV expression levels. Methods: Based on the hypothesis that PV expression might be increased by 17-beta estradiol (E2), PV+/- mice were treated with E2 from postnatal days 5-15 and ASD-related behavior was tested between postnatal days 25 and 31. Results: PV expression levels were significantly increased after E2 treatment and, concomitantly, sociability deficits in PV+/- mice in the direct reciprocal social interaction and the 3-chamber social approach assay, as well as repetitive behaviors, were attenuated. E2 treatment of PV+/+ mice did not increase PV levels and had detrimental effects on sociability and repetitive behavior. In PV-/- mice, E2 obviously did not affect PV levels; tested behaviors were not different from the ones in vehicle-treated PV-/- mice. Conclusion: Our results suggest that the E2-linked amelioration of ASD-like behaviors is specifically occurring in PV+/- mice, indicating that PV upregulation is required for the E2-mediated rescue of ASD-relevant behavioral impairments. En ligne : http://dx.doi.org/10.1186/s13229-018-0199-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354

Age-specific autistic-like behaviors in heterozygous Fmr1-KO female mice / Manon GAUDUCHEAU in Autism Research, 10-6 (June 2017)  

Permalink

Serotonin Disturbance in Mouse Models of Autism Spectrum Disorders / Kota TAMADA

Permalink