International Journal of Developmental Neuroscience最新文献

{"title":"Long-term effect of indomethacin on a rat model of neonatal hypoxia ischemic encephalopathy through behavioral tests","authors":"Tugay Tepe, Mehmet Satar, Mustafa Ozdemir, Hacer Yapicioglu Yildizdas, Ferda Ozlü, Seyda Erdogan, Tugba Toyran, Kübra Akillioglu, Seda Köse, Cagri Avci","doi":"10.1002/jdn.10305","DOIUrl":"10.1002/jdn.10305","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Many medical experts prescribe indomethacin because of its anti-inflammatory, analgesic, tocolytic, and duct closure effects. This article presents an evaluation of the enduring impact of indomethacin on neonatal rats with hypoxic–ischemic (HI) insults, employing behavioral tests as a method of assessment.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>The experiment was conducted on male Wistar-Albino rats weighing 10 to 15 g, aged between seven and 10 days. The rats were divided into three groups using a random allocation method as follows: hypoxic ischemic encephalopathy (HIE) group, HIE treated with indomethacin group (INDO), and Sham group. A left common carotid artery ligation and hypoxia model was applied in both the HIE and INDO groups. The INDO group was treated with 4 mg/kg intraperitoneal indomethacin every 24 h for 3 days, while the Sham and HIE groups were given dimethylsulfoxide (DMSO). After 72 h, five rats from each group were sacrificed and brain tissue samples were stained with 2,3,5-Triphenyltetrazolium chloride (TCC) for infarct-volume measurement. Seven rats from each group were taken to the behavioral laboratory in the sixth postnatal week (PND42) and six from each group were sacrificed for the Evans blue (EB) experiment for blood–brain barrier (BBB) integrity evaluation. The open field (OF) test and Morris water maze (MWM) tests were performed. After behavioral tests, brain tissue were obtained and stained with TCC to assess the infarct volume.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The significant increase in the time spent in the central area and the frequency of crossing to the center in the INDO group compared with the HIE group indicated that indomethacin decreased anxiety-like behavior (<i>p</i> < 0.001, <i>p</i> < 0.05). However, the MWM test revealed that indomethacin did not positively affect learning and memory performance (<i>p</i> > 0.05). Additionally, indomethacin significantly reduced infarct volume and neuropathological grading in adolescence (<i>p</i> < 0.05), although not statistically significant in the early period. Moreover, the EB experiment demonstrated that indomethacin effectively increased BBB integrity (<i>p</i> < 0.05).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>In this study, we have shown for the first time that indomethacin treatment can reduce levels of anxiety-like behavior and enhance levels of exploratory behavior in a neonatal rat model with HIE. It is necessary to determine whether nonsteroidal anti-inflammatory agents, such as indomethacin, should be used for adjuvant therapy in newborns with HIE.</p>\u0000 ","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"84 1","pages":"22-34"},"PeriodicalIF":1.8,"publicationDate":"2023-10-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41235133","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}

{"title":"Ginkgolide B promotes spontaneous recovery and enhances endogenous netrin-1 after neonatal hypoxic–ischemic brain damage","authors":"Aiming Chen,&nbsp;Jun Hua,&nbsp;Jun Yuan,&nbsp;Yajuan Feng,&nbsp;Fengzhan Chen,&nbsp;Yongqin Zhou,&nbsp;Ting Han,&nbsp;Weiwei Jiang,&nbsp;Huiping Chen","doi":"10.1002/jdn.10301","DOIUrl":"10.1002/jdn.10301","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>Perinatal hypoxic–ischemic encephalopathy (HIE) is a condition that can lead to long-term cognitive, motor, and behavioral impairments in newborns. Although brain hypothermia therapy is currently the standard treatment for HIE, it does not provide complete neuroprotection. As a result, there is a need to explore additional therapies to enhance treatment outcomes. This study aims to investigate the potential role of Ginkgolide B (GB) in promoting neuroplasticity and facilitating spontaneous recovery after HIE.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>In this study, we employed a neonatal rat model of HIE to investigate the effects of GB on spontaneous recovery. GB treatment was initiated 24 h after hypoxia and administered continuously for a duration of 14 days. We evaluated several outcome measures after the treatment period, including spontaneous behavioral recovery and brain repair. Additionally, we quantified the levels of netrin-1 in both plasma and the peri-ischemic zone after the occurrence of HIE.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>We found that GB treatment significantly facilitated spontaneous behavioral recovery in the HIE pups. Furthermore, cognitive function was restored, and brain tissue repair had a noticeable acceleration. We observed increased cell proliferation in the subventricular, stratum, and subgranular zones. Of particular interest, we observed elevated levels of netrin-1 in both plasma and the ischemic penumbra following GB treatment.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>Our findings suggest that GB promotes neuroplasticity and enhances spontaneous recovery in newborns affected by HIE. The observed upregulation of netrin-1 may be crucial in mediating these effects. These results highlight the promising potential of GB as a post-HIE therapy, particularly in enhancing spontaneous recovery and improving long-term outcomes.</p>\u0000 </section>\u0000 </div>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"740-752"},"PeriodicalIF":1.8,"publicationDate":"2023-09-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41126503","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}

{"title":"The effect of the home environment on children with autism spectrum disorder","authors":"Gul Kahveci,&nbsp;Emrah Caylak,&nbsp;Donay Nisa Kara","doi":"10.1002/jdn.10304","DOIUrl":"10.1002/jdn.10304","url":null,"abstract":"<p>The estimated prevalence of autism spectrum disorders (ASD) is 1% worldwide. Autistic individuals typically have a high level of sensitivity to the various environmental stimuli (smell, noise, light). These stimuli have a positive or negative influence on the person–environment interaction, and an excess of stimuli may create inappropriate or unanticipated behavioral responses (such as a crisis) effecting their well-being. The Model of Competence, which provides an explanation of the interaction between the individual and the environment, was selected as the conceptual framework to direct this study. The purpose of this study is to investigate the opinions and experiences of mothers regarding the influence of the characteristics of the home environment on autistic individuals. A qualitative interpretative description design was utilized for this study's framework. Participants in the study were mothers who have autistic children. The study with focus groups was continued until data saturation was reached. There was a thematic investigation carried out. The findings show that the factors that have an effect on autistic individuals can be categorized into several subsets like sensory, routines, and physical environment. Despite the fact that the home setting is often a secure and consistent environment, these aspects were identified as crucial. Consequently, one should give some thought to the consequences that this could have in other settings where it would be harder to exercise control. The identification of these factors and the impact they have enables a better understanding of the interaction between an autistic individual and their environment and serves to guide professionals in their interventions.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"84 1","pages":"14-21"},"PeriodicalIF":1.8,"publicationDate":"2023-09-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41126504","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}

{"title":"Increased expression levels of DLX5 inhibit the development of the nervous system","authors":"Tingting Liao,&nbsp;Xia Xu,&nbsp;Junzi Wu,&nbsp;Yi Xie,&nbsp;Jianying Yan","doi":"10.1002/jdn.10300","DOIUrl":"10.1002/jdn.10300","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Introduction</h3>\u0000 \u0000 <p>Preeclampsia is a hypertensive disorder of pregnancy. DLX5 plays an important role in the migration and differentiation of subglobus pallidus precursor cells.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>We established a zebrafish line expressing high levels of DLX5 and investigated changes in behavior and development of the nervous system.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The ratios of brain volume area to whole body area at 96 hpf zebrafish in the experimental group (gRNA + CasRx) were significantly lower than the WT group and the negative control group (casRx) (<i>P</i> &lt; 0.01). Behavioral trajectory distances and movement speeds exhibited by the 6th day of development in zebrafish in the experimental group (gRNA + CasRx) were significantly shorter (<i>P</i> &lt; 0.01) and lower (<i>P</i> &lt; 0.05) than the negative control group (gRNA + CasRx), respectively.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Data suggested that the increased expression levels of DLX5 can inhibit brain volume development and behavioral activities in zebrafish. Maybe the high expression levels of DLX5 in the pathological state of preeclampsia can inhibit the development of the nervous system in offspring.</p>\u0000 </section>\u0000 </div>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"728-739"},"PeriodicalIF":1.8,"publicationDate":"2023-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jdn.10300","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41112208","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}

{"title":"Ap4s1 truncation leads to axonal defects in a zebrafish model of spastic paraplegia 52","authors":"Yiduo Li,&nbsp;Cuizhen Zhang,&nbsp;Gang Peng","doi":"10.1002/jdn.10303","DOIUrl":"10.1002/jdn.10303","url":null,"abstract":"<p>Biallelic mutations in AP4S1, the σ4 subunit of the adaptor protein complex 4 (AP-4), lead to autosomal recessive spastic paraplegia 52 (SPG52). It is a subtype of AP-4-associated hereditary spastic paraplegia (AP-4-HSP), a complex childhood-onset neurogenetic disease characterized by progressive spastic paraplegia of the lower limbs. This disease has so far lacked effective treatment, in part due to a lack of suitable animal models. Here, we used CRISPR/Cas9 technology to generate a truncation mutation in the <i>ap4s1</i> gene in zebrafish. The <i>ap4s1</i> truncation led to motor impairment, delayed neurodevelopment, and distal axonal degeneration. This animal model is useful for further research into AP-4 and AP-4-HSP.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"753-764"},"PeriodicalIF":1.8,"publicationDate":"2023-09-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41118892","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}

{"title":"Adaptive, behavioral, and cognitive outcomes in individuals with fragile X syndrome with varying autism severity","authors":"Ramkumar Aishworiya,&nbsp;Ye Eun Tak,&nbsp;Matthew Dominic Ponzini,&nbsp;Hazel Maridith Barlahan Biag,&nbsp;Maria Jimena Salcedo-Arellano,&nbsp;Kyoungmi Kim,&nbsp;Flora Tassone,&nbsp;Andrea Schneider,&nbsp;Angela John Thurman,&nbsp;Leonard Abbeduto,&nbsp;David Hessl,&nbsp;Jamie Leah Randol,&nbsp;Francois V. Bolduc,&nbsp;Sarah Lippe,&nbsp;Paul Hagerman,&nbsp;Randi Hagerman","doi":"10.1002/jdn.10299","DOIUrl":"10.1002/jdn.10299","url":null,"abstract":"<p>This study aimed to determine the association between severity of autism spectrum disorder (ASD) and cognitive, behavioral, and molecular measures in individuals with fragile X syndrome (FXS). Study inclusion criteria included individuals with FXS and (1) age 6–40 years, (2) full-scale IQ &lt; 84, and (3) language ≥3-word phrases. ASD symptom severity was determined by Autism Diagnostic Observation Schedule-2 (ADOS-2). Other measures identified non-verbal IQ, adaptive skills, and aberrant behaviors. Molecular measures included blood <i>FMR1</i> and <i>CYFIP1</i> mRNA levels, FMRP and MMP9 levels. Analysis of variance (ANOVA) and Spearman's correlations were used to compare ASD severity groups. Data from 54 individuals was included with no/mild (<i>N</i> = 7), moderate (<i>N</i> = 18), and severe (<i>N</i> = 29) ASD. Individuals with high ASD severity had lower adaptive behavior scores (47.48 ± 17.49) than the no/mild group (69.00 ± 20.45, <i>p</i> = 0.0366); they also had more challenging behaviors, lethargy, and stereotypic behaviors. <i>CYFIP1</i> mRNA expression levels positively correlated with the ADOS-2 comparison score(<i>r</i>² = 0.33, <i>p</i> = 0.0349), with no significant correlations with other molecular markers. In conclusion, autism symptom severity is associated with more adverse cognitive and adaptive skills and specific behaviors in FXS, whereas <i>CYFIP1</i> mRNA expression levels may be a potential biomarker for severity of ASD in FXS.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"715-727"},"PeriodicalIF":1.8,"publicationDate":"2023-09-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jdn.10299","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41148056","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}

{"title":"Postnatal development of the hippocampal GABAergic system in rats genetically prone to audiogenic seizures","authors":"Andrey P. Ivlev,&nbsp;Alexandra A. Naumova","doi":"10.1002/jdn.10298","DOIUrl":"10.1002/jdn.10298","url":null,"abstract":"<p>Epileptogenesis can be associated with altered genetic control of the GABAergic system. Here we analyzed Krushinsky–Molodkina (KM) rats genetically prone to audiogenic epilepsy. KM rats express fully formed audiogenic seizures (AGSs) not early, then they reach 3 months. At the age of 1–2 months, KM rats either do not express AGS or demonstrate an incomplete pattern of seizure. Such long-term development of AGS susceptibility makes KM rats an especially convenient model to investigate the mechanisms and dynamics of the development of inherited epilepsy. The analysis of the GABAergic system of the hippocampus of KM rats was done during postnatal development at the 15th, 60th, and 120th postnatal days. Wistar rats of corresponding ages were used as a control. In the hippocampus of KM pups, we observed a decrease in the expression of glutamic acid decarboxylase 67 (GAD67) and parvalbumin (PV), which points to a decrease in the activity of GABAergic neurons. Analysis of the 2-month-old KM rats showed an increase in GAD67 and PV expression while synapsin I and vesicular GABA transporter (VGAT) were decreased. In adult KM rats, the expression of GAD67, PV, and synapsin I was upregulated. Altogether, the obtained data indicate significant alterations in GABAergic transmission in the hippocampus of audiogenic KM rats during the first postnatal months.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"703-714"},"PeriodicalIF":1.8,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10484280","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}

{"title":"Efficacy and safety of V1a receptor antagonists in autism spectrum disorder: A meta-analysis","authors":"Sneha Kimi,&nbsp;Rituparna Maiti,&nbsp;Anand Srinivasan,&nbsp;Biswa Ranjan Mishra,&nbsp;Debasish Hota","doi":"10.1002/jdn.10297","DOIUrl":"10.1002/jdn.10297","url":null,"abstract":"<p>This meta-analysis has evaluated the efficacy and safety of V_1a receptor antagonists in ASD compared to placebo. The reviewers extracted data from four relevant clinical trials after a literature search on databases and clinical trial registries. Quality assessment was done using the risk of bias assessment tool, and the random-effects model was used to estimate effect size. Subgroup analysis, meta-regression and sensitivity analysis were done. PRISMA guidelines were followed in the selection, analysis and reporting of findings. V_1a receptor antagonists did not reduce Vineland II Adaptive behaviour composite score significantly (SMD: 0.14; 95% CI: −0.06–0.35; <i>p</i> = 0.16; PI: −0.44–0.73), communication domain subscale score and socialization domain subscale score. The change in daily living skills domain subscale score was significant and favourable for V_1a receptor antagonists (SMD: 0.15; 95% CI: 0.03–0.26; <i>p</i> = 0.01). The subgroup analysis revealed a significant improvement in Vineland II Adaptive behaviour composite score with doses &lt;10 mg (SMD: 0.45; 95% CI: 0.11–0.78; <i>p</i> = 0.009). Meta-regression does not show a significant association between SMD of ASD symptom score reduction with the duration and dose of V_1a receptor antagonist therapy. Treatment-emergent adverse effects were not serious and dose dependent. Low doses (&lt;10 mg) of V_1a receptor antagonist may be effective in reducing the core symptoms of ASD compared to placebo; however, future active-controlled clinical trials are necessary to generate conclusive evidence.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"84 1","pages":"3-13"},"PeriodicalIF":1.8,"publicationDate":"2023-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10466704","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}

{"title":"Haloperidol alters neurotrophic factors and epigenetic parameters in an animal model of schizophrenia induced by ketamine","authors":"Samira S. Valvassori,&nbsp;Richard T. da Rosa,&nbsp;Gustavo C. Dal-Pont,&nbsp;Roger B. Varela,&nbsp;Gustavo A. Mastella,&nbsp;Thiani Daminelli,&nbsp;Gabriel R. Fries,&nbsp;João Quevedo,&nbsp;Alexandra I. Zugno","doi":"10.1002/jdn.10296","DOIUrl":"10.1002/jdn.10296","url":null,"abstract":"<p>This study aimed to evaluate Haloperidol's (Hal) effects on the behavioral, neurotrophic factors, and epigenetic parameters in an animal model of schizophrenia (SCZ) induced by ketamine (Ket). Injections of Ket or saline were administered intraperitoneal (once a day) between the 1st and 14th days of the experiment. Water or Hal was administered via gavage between the 8th and 14th experimental days. Thirty minutes after the last injection, the animals were subjected to behavioral analysis. The activity of DNA methyltransferase (DNMT), histone deacetylase (HDAC), and histone acetyltransferase and levels of brain-derived neurotrophic factor (BDNF), nerve growth factor (NGF), neurotrophin-3 (NT-3), and glial-derived neurotrophic factor (GDNF) were evaluated in the frontal cortex, hippocampus, and striatum. Ket increased the covered distance and time spent in the central area of the open field, and Hal did not reverse these behavioral alterations. Significant increases in the DNMT and HDAC activities were detected in the frontal cortex and striatum from rats that received Ket, Hal, or a combination thereof. Besides, Hal per se increased the activity of DNMT and HDAC in the hippocampus of rats. Hal per se or the association of Ket plus Hal decreased BDNF, NGF, NT-3, and GDNF, depending on the brain region and treatment regimen. The administration of Hal can alter the levels of neurotrophic factors and the activity of epigenetic enzymes, which can be a factor in the development of effect collateral in SCZ patients. However, the precise mechanisms involved in these alterations are still unclear.</p>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 8","pages":"691-702"},"PeriodicalIF":1.8,"publicationDate":"2023-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10439680","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}

{"title":"Brain structural changes in alternating hemiplegia of childhood using single-case voxel-based morphometry analysis","authors":"Elly Arizono,&nbsp;Noriko Sato,&nbsp;Yoko Shigemoto,&nbsp;Yukio Kimura,&nbsp;Emiko Chiba,&nbsp;Hiroyuki Maki,&nbsp;Hiroshi Matsuda,&nbsp;Eri Takeshita,&nbsp;Yuko Shimizu-Motohashi,&nbsp;Masayuki Sasaki,&nbsp;Kazuhiro Saito","doi":"10.1002/jdn.10295","DOIUrl":"10.1002/jdn.10295","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background and purpose</h3>\u0000 \u0000 <p>Alternating hemiplegia of childhood (AHC) is a rare neurodevelopmental disease caused by <i>ATP1A3</i> mutations. Using voxel-based morphometry (VBM) analysis, we compared an AHC patient cohort with controls. Additionally, with single-case VBM analysis, we assessed the associations between clinical severity and brain volume in patients with AHC.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Materials and methods</h3>\u0000 \u0000 <p>To investigate structural brain changes in gray matter (GM) and white matter (WM) volumes between 9 patients with AHC and 20 age-matched controls, VBM analysis was performed using three-dimensional T1-weighted magnetic resonance imaging. Single-case VBM analysis was also performed on nine patients with AHC to investigate the associations between the respective volumes of GM/WM differences and the motor level, cognitive level, and status epilepticus severity in patients with AHC.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Compared with controls, patients with AHC showed significant GM volume reductions in both hippocampi and diffuse cerebellum, and there were WM reductions in both cerebral hemispheres. In patients with AHC, cases with more motor dysfunction, the less GM/WM volume of cerebellum was shown. Three of the six cases with cognitive dysfunction showed a clear GM volume reduction in the insulae. Five of the six cases with status epilepticus showed the GM volume reduction in hippocampi. One case had severe status epilepticus without motor dysfunction and showed no cerebellar atrophy.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>With single-case VBM analysis, we could show the association between region-specific changes in brain volume and the severity of various clinical symptoms even in a small sample of subjects.</p>\u0000 </section>\u0000 </div>","PeriodicalId":13914,"journal":{"name":"International Journal of Developmental Neuroscience","volume":"83 7","pages":"665-673"},"PeriodicalIF":1.8,"publicationDate":"2023-08-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10395542","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}