Developmental Neurobiology最新文献_第10页

Loss of mitochondrial Chchd10 or Chchd2 in zebrafish leads to an ALS-like phenotype and Complex I deficiency independent of the mitochondrial integrated stress response 斑马鱼线粒体Chchd10或Chchd2的缺失会导致als样表型和独立于线粒体综合应激反应的复合物I缺陷

IF 3 4区医学

Developmental Neurobiology Pub Date : 2023-02-16 DOI: 10.1002/dneu.22909

Virginie Petel Légaré, Christian J. Rampal, Tyler J. N. Gurberg, Mari J. Aaltonen, Alexandre Janer, Lorne Zinman, Eric A. Shoubridge, Gary A. B. Armstrong

{"title":"Loss of mitochondrial Chchd10 or Chchd2 in zebrafish leads to an ALS-like phenotype and Complex I deficiency independent of the mitochondrial integrated stress response","authors":"Virginie Petel Légaré, Christian J. Rampal, Tyler J. N. Gurberg, Mari J. Aaltonen, Alexandre Janer, Lorne Zinman, Eric A. Shoubridge, Gary A. B. Armstrong","doi":"10.1002/dneu.22909","DOIUrl":"10.1002/dneu.22909","url":null,"abstract":"Mutations in CHCHD10 and CHCHD2, encoding two paralogous mitochondrial proteins, have been identified in cases of amyotrophic lateral sclerosis, frontotemporal lobar degeneration, and Parkinson's disease. Their role in disease is unclear, though both have been linked to mitochondrial respiration and mitochondrial stress responses. Here, we investigated the biological roles of these proteins during vertebrate development using knockout (KO) models in zebrafish. We demonstrate that loss of either or both proteins leads to motor impairment, reduced survival and compromised neuromuscular junction integrity in larval zebrafish. Compensation by Chchd10 was observed in the chchd2−/− model, but not by Chchd2 in the chchd10−/− model. The assembly of mitochondrial respiratory chain Complex I was impaired in chchd10−/− and chchd2−/− zebrafish larvae, but unexpectedly not in a double chchd10−/− and chchd2−/− model, suggesting that reduced mitochondrial Complex I cannot be solely responsible for the observed phenotypes, which are generally more severe in the double KO. We observed transcriptional activation markers of the mitochondrial integrated stress response (mt-ISR) in the double chchd10−/− and chchd2−/− KO model, suggesting that this pathway is involved in the restoration of Complex I assembly in our double KO model. The data presented here demonstrates that the Complex I assembly defect in our single KO models arises independently of the mt-ISR. Furthermore, this study provides evidence that both proteins are required for normal vertebrate development.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"83 1-2","pages":"54-69"},"PeriodicalIF":3.0,"publicationDate":"2023-02-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/dneu.22909","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9292321","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 1

Cover Image, Volume 82, Issue 7–8 封面图片，第82卷，第7-8期

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-11-22 DOI: 10.1002/dneu.22908

引用次数: 0

Excitatory and inhibitory neuron imbalance in the intrauterine growth restricted fetal guinea pig brain: Relevance to the developmental origins of schizophrenia and autism 宫内生长受限的豚鼠胎儿脑中的兴奋性和抑制性神经元失衡:与精神分裂症和自闭症的发育起源相关

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-11-14 DOI: 10.1002/dneu.22907

Angela Cumberland, Nadia Hale, Aminath Azhan, Courtney P. Gilchrist, Ginevra Chincarini, Mary Tolcos

{"title":"Excitatory and inhibitory neuron imbalance in the intrauterine growth restricted fetal guinea pig brain: Relevance to the developmental origins of schizophrenia and autism","authors":"Angela Cumberland, Nadia Hale, Aminath Azhan, Courtney P. Gilchrist, Ginevra Chincarini, Mary Tolcos","doi":"10.1002/dneu.22907","DOIUrl":"10.1002/dneu.22907","url":null,"abstract":"Neurodevelopmental disorders such as schizophrenia and autism are thought to involve an imbalance of excitatory and inhibitory signaling in the brain. Intrauterine growth restriction (IUGR) is a risk factor for these disorders, with IUGR onset occurring during critical periods of neurodevelopment. The aim of this study was to determine the impact of IUGR on excitatory and inhibitory neurons of the fetal neocortex and hippocampus. Fetal brains (n = 2) were first collected from an unoperated pregnant guinea pig at mid-gestation (32 days of gestation [dg]; term ∼67 dg) to visualize excitatory (Ctip2) and inhibitory (calretinin [CR] and somatostatin [SST]) neurons via immunohistochemistry. Chronic placental insufficiency (CPI) was then induced via radial artery ablation at 30 dg in another cohort of pregnant guinea pigs (n = 8) to generate IUGR fetuses (52 dg; n = 8); control fetuses (52 dg; n = 7) were from sham surgeries with no radial artery ablation. At 32 dg, Ctip2- and CR-immunoreactive (IR) cells had populated the cerebral cortex, whereas SST-IR cells had not, suggesting these neurons were yet to complete migration. At 52 dg, in IUGR versus control fetuses, there was a reduction in SST-IR cell density in the cerebral cortex (p = .0175) and hilus of the dentate gyrus (p = .0035) but not the striatum (p > .05). There was no difference between groups in the density of Ctip2-IR (cortex) or CR-IR (cortex, hippocampus) neurons (p > 0.05). Thus, we propose that an imbalance in inhibitory (SST-IR) and excitatory (Ctip2-IR) neurons in the IUGR fetal guinea pig brain could lead to excitatory/inhibitory dysfunction commonly seen in neurodevelopmental disorders such as autism and schizophrenia.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"83 1-2","pages":"40-53"},"PeriodicalIF":3.0,"publicationDate":"2022-11-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/dneu.22907","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9344297","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

microRNA-124 regulates Notch and NeuroD1 to mediate transition states of neuronal development microRNA-124调节Notch和NeuroD1介导神经元发育的过渡状态

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-11-07 DOI: 10.1002/dneu.22902

Kalin D. Konrad, Jia L. Song

{"title":"microRNA-124 regulates Notch and NeuroD1 to mediate transition states of neuronal development","authors":"Kalin D. Konrad, Jia L. Song","doi":"10.1002/dneu.22902","DOIUrl":"10.1002/dneu.22902","url":null,"abstract":"MicroRNAs regulate gene expression by destabilizing target mRNA and/or inhibiting translation in animal cells. The ability to mechanistically dissect miR-124′s function during specification, differentiation, and maturation of neurons during development within a single system has not been accomplished. Using the sea urchin embryo, we take advantage of the manipulability of the embryo and its well-documented gene regulatory networks (GRNs). We incorporated NeuroD1 as part of the sea urchin neuronal GRN and determined that miR-124 inhibition resulted in aberrant gut contractions, swimming velocity, and neuronal development. Inhibition of miR-124 resulted in an increased number of cells expressing transcription factors (TFs) associated with progenitor neurons and a concurrent decrease of mature and functional neurons. Results revealed that in the early blastula/gastrula stages, miR-124 regulates undefined factors during neuronal specification and differentiation. In the late gastrula/larval stages, miR-124 regulates Notch and NeuroD1 during the transition between neuronal differentiation and maturation. Overall, we have improved the neuronal GRN and identified miR-124 to play a prolific role in regulating various transitions of neuronal development.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"83 1-2","pages":"3-27"},"PeriodicalIF":3.0,"publicationDate":"2022-11-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10440801/pdf/nihms-1909457.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10043228","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 2

Family income buffers the relationship between childhood adverse experiences and putamen volume 家庭收入缓冲了童年不良经历与壳核体积之间的关系

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-10-31 DOI: 10.1002/dneu.22906

Max P. Herzberg, Laura Hennefield, Katherine R. Luking, Ashley F. P. Sanders, Alecia C. Vogel, Sridhar Kandala, Rebecca Tillman, Joan Luby, Deanna M. Barch

{"title":"Family income buffers the relationship between childhood adverse experiences and putamen volume","authors":"Max P. Herzberg, Laura Hennefield, Katherine R. Luking, Ashley F. P. Sanders, Alecia C. Vogel, Sridhar Kandala, Rebecca Tillman, Joan Luby, Deanna M. Barch","doi":"10.1002/dneu.22906","DOIUrl":"10.1002/dneu.22906","url":null,"abstract":"Adverse experiences and family income in childhood have been associated with altered brain development. While there is a large body of research examining these associations, it has primarily used cross-sectional data sources and studied adverse experiences and family income in isolation. However, it is possible that low family income and adverse experiences represent dissociable and potentially interacting profiles of risk. To address this gap in the literature, we examined brain structure as a function of adverse experiences in childhood and family income in 158 youths with up to five waves of MRI data. Specifically, we assessed the interactive effect of these two risk factors on six regions of interest: hippocampus, putamen, amygdala, nucleus accumbens, caudate, and thalamus. Adverse experiences and family income interacted to predict putamen volume (B = 0.086, p = 0.011) but only in participants with family income one standard deviation below the mean (slope estimate = −0.11, p = 0.03). These results suggest that adverse experiences in childhood result in distinct patterns of brain development across the socioeconomic gradient. Given previous findings implicating the role of the putamen in psychopathology-related behaviors, these results emphasize the importance of considering life events and socioeconomic context when evaluating markers of risk. Future research should include interactive effects of environmental exposures and family income to better characterize risk for psychopathology in diverse samples.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"83 1-2","pages":"28-39"},"PeriodicalIF":3.0,"publicationDate":"2022-10-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/dneu.22906","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9291288","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Microglia in motor neuron disease: Signaling evidence from last 10 years 运动神经元疾病中的小胶质细胞:近10年的信号证据

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-10-29 DOI: 10.1002/dneu.22905

Min-Jia Wang, Lu Kang, Yao-Zheng Wang, Bi-Ru Yang, Chun Zhang, Yu-Feng Lu, Liang Kang

引用次数: 7

Terminal field volume of the glossopharyngeal nerve in adult rats reverts to prepruning size following microglia depletion with PLX5622 用PLX5622去除小胶质细胞后，成年大鼠舌咽神经终末场体积恢复到修剪前的大小

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-10-29 DOI: 10.1002/dneu.22904

Andrew J. Riquier, Suzanne I. Sollars

{"title":"Terminal field volume of the glossopharyngeal nerve in adult rats reverts to prepruning size following microglia depletion with PLX5622","authors":"Andrew J. Riquier, Suzanne I. Sollars","doi":"10.1002/dneu.22904","DOIUrl":"10.1002/dneu.22904","url":null,"abstract":"Programmed reduction of synapses is a hallmark of the developing brain, with sensory systems emerging as useful models with which to study this pruning. The central projections (terminal field) of the gustatory glossopharyngeal nerve (GL) of the rat are a prime example of developmental pruning, undergoing an approximate 66% reduction in volume from postnatal day 15 (P15) to P25. Later in adulthood, developmental GL pruning can be experimentally reversed, expanding to preweaning volumes, suggesting mature volumes may be actively maintained throughout the life span. Microglia are central nervous system glia cells that perform pruning and maintenance functions in other sensory systems, including other gustatory nerves. To determine their role in GL pruning, we depleted microglia from Sprague–Dawley rat brains from P1 to P40 using daily intraperitoneal injections of the colony-stimulating factor 1 receptor inhibitor PLX5622. This prevented GL developmental pruning, resulting in preweaning terminal field volumes and innervation patterns persisting through P40, 2 weeks after pruning is normally completed. These findings show microglia are necessary for developmental GL pruning. Ceasing PLX5622 treatments at P40 allowed microglia repopulation, and within 4 weeks the GL terminal field had reduced to control volumes, indicating that pruning can occur outside of the typical developmental period. Conversely, when microglia were depleted in adult rats, GL terminal fields expanded, reverting to sizes comparable to the neonatal rat. These data indicate that microglia are required for GL pruning and may continue to maintain the GL terminal field at a reduced size into adulthood.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"82 7-8","pages":"613-624"},"PeriodicalIF":3.0,"publicationDate":"2022-10-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/d4/16/DNEU-82-613.PMC9790758.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10620840","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

Large-scale waves of activity in the neonatal mouse brain in vivo occur almost exclusively during sleep cycles 活体新生小鼠大脑的大规模活动波几乎只发生在睡眠周期

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-10-17 DOI: 10.1002/dneu.22901

Dennis R. Tabuena, Randy Huynh, Jenna Metcalf, Thomas Richner, Albrecht Stroh, Bingni W. Brunton, William J. Moody, Curtis R. Easton

{"title":"Large-scale waves of activity in the neonatal mouse brain in vivo occur almost exclusively during sleep cycles","authors":"Dennis R. Tabuena, Randy Huynh, Jenna Metcalf, Thomas Richner, Albrecht Stroh, Bingni W. Brunton, William J. Moody, Curtis R. Easton","doi":"10.1002/dneu.22901","DOIUrl":"10.1002/dneu.22901","url":null,"abstract":"Spontaneous electrical activity plays major roles in the development of cortical circuitry. This activity can occur highly localized regions or can propagate over the entire cortex. Both types of activity coexist during early development. To investigate how different forms of spontaneous activity might be temporally segregated, we used wide-field trans-cranial calcium imaging over an entire hemisphere in P1–P8 mouse pups. We found that spontaneous waves of activity that propagate to cover the majority of the cortex (large-scale waves; LSWs) are generated at the end of the first postnatal week, along with several other forms of more localized activity. We further found that LSWs are segregated into sleep cycles. In contrast, cortical activity during wake states is more spatially restricted and the few large-scale forms of activity that occur during wake can be distinguished from LSWs in sleep based on their initiation in the motor cortex and their correlation with body movements. This change in functional cortical circuitry to a state that is permissive for large-scale activity may temporally segregate different forms of activity during critical stages when activity-dependent circuit development occurs over many spatial scales. Our data also suggest that LSWs in early development may be a functional precursor to slow sleep waves in the adult, which play critical roles in memory consolidation and synaptic rescaling.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"82 7-8","pages":"596-612"},"PeriodicalIF":3.0,"publicationDate":"2022-10-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9419421","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 1

Sex differences in myelination of the zebra finch vocal control system emerge relatively late in development 斑胸草雀声音控制系统髓鞘形成的性别差异在发育中相对较晚出现

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-10-07 DOI: 10.1002/dneu.22900

Adriana Diez, Shenghan Wang, Nicole Carfagnini, Scott A. MacDougall-Shackleton

{"title":"Sex differences in myelination of the zebra finch vocal control system emerge relatively late in development","authors":"Adriana Diez, Shenghan Wang, Nicole Carfagnini, Scott A. MacDougall-Shackleton","doi":"10.1002/dneu.22900","DOIUrl":"10.1002/dneu.22900","url":null,"abstract":"The role of myelination in the development of motor control is widely known, but its role in the development of cognitive abilities is less understood. Here, we examined sex differences in the development of myelination of structures and tracts that support song learning and production in songbirds. We collected brains from 63 young male and female zebra finches (Taeniopygia guttata) over four stages of development that correspond to different stages of song learning. Using a myelination marker (myelin basic protein), we measured the development of myelination in three different nuclei of the vocal control system (HVC, RA, and lateral magnocellular nucleus of the anterior nidopallium [LMAN]) and two tracts (HVC-RA and lamina mesopallium ventralis [LMV]). We found that the myelination of the vocal control nuclei and tracts is sex related and male biased. In males, the patterns of myelination were age-dependent, asynchronous in rate and progression and associated with the development of song learning and production. In females, myelination of vocal control nuclei was low or absent and did not significantly change with age. Sex differences in myelination of the HVC-RA tract were large and emerged late in development well after sex differences in the size of vocal control brain regions are established. Myelination of this tract in males coincides with the age of song crystallization. Overall, the changes in myelination in the vocal control areas and tracts measured are region-, age-, and sex-specific and are consistent with sex differences in song development.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"82 7-8","pages":"581-595"},"PeriodicalIF":3.0,"publicationDate":"2022-10-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10627152","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0

The cortical hem lacks stem cell potential despite expressing SOX9 and HOPX 尽管皮质边缘表达SOX9和HOPX，但缺乏干细胞潜能

IF 3 4区医学

Developmental Neurobiology Pub Date : 2022-09-06 DOI: 10.1002/dneu.22899

Alessia Caramello, Christophe Galichet, Miriam Llorian Sopena, Robin Lovell-Badge, Karine Rizzoti

{"title":"The cortical hem lacks stem cell potential despite expressing SOX9 and HOPX","authors":"Alessia Caramello, Christophe Galichet, Miriam Llorian Sopena, Robin Lovell-Badge, Karine Rizzoti","doi":"10.1002/dneu.22899","DOIUrl":"10.1002/dneu.22899","url":null,"abstract":"The adult dentate gyrus (DG) of rodents hosts a neural stem cell (NSC) niche capable of generating new neurons throughout life. The embryonic origin and molecular mechanisms underlying formation of DG NSCs are still being investigated. We performed a bulk transcriptomic analysis on mouse developing archicortex conditionally deleted for Sox9, a SoxE transcription factor controlling both gliogenesis and NSC formation, and identified Hopx, a recently identified marker of both prospective adult DG NSCs and astrocytic progenitors, as being downregulated. We confirm SOX9 is required for HOPX expression in the embryonic archicortex. In particular, we found that both NSC markers are highly expressed in the cortical hem (CH), while only weakly in the adjacent dentate neuroepithelium (DNE), suggesting a potential CH embryonic origin for DG NSCs. However, we demonstrate both in vitro and in vivo that the embryonic CH, as well as its adult derivatives, lacks stem cell potential. Instead, deletion of Sox9 in the DNE affects both HOPX expression and NSC formation in the adult DG. We conclude that HOPX expression in the CH is involved in astrocytic differentiation downstream of SOX9, which we previously showed regulates DG development by inducing formation of a CH-derived astrocytic scaffold. Altogether, these results suggest that both proteins work in a dose-dependent manner to drive either astrocytic differentiation in CH or NSC formation in DNE.","PeriodicalId":11300,"journal":{"name":"Developmental Neurobiology","volume":"82 7-8","pages":"565-580"},"PeriodicalIF":3.0,"publicationDate":"2022-09-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9826121/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10628860","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

引用次数: 0