Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is the most frequent form of acute encephalopathy in early childhood in Japan. Magnetic resonance imaging provides useful hallmarks of diagnosing AESD. However, metabolomic profiles for AESD remain elusive. This study investigates whether measurement of amino acids in the cerebrospinal fluid (CSF) is useful for the diagnosis of AESD before onset.
Methods
In the first study, CSF samples were collected from patients (11 AESD and 17 controls) admitted to Kyushu University Hospital during 2011–2016. Amino acids in the CSF were analyzed using mass spectrometry. Cytometric bead arrays were used to measure cytokine and chemokine levels in the CSF. The second study was performed by recruiting patients (8 AESD patients and 10 controls) admitted during 2011–2024. CSF samples were stored at −20 °C for 1 month to 12 years.
Results
In the first study, glutamate levels in the CSF from AESD patients were higher than in controls and correlated with methionine, threonine, and tyrosine levels. A correlation map of cytokines and amino acids revealed that glutamate formed a cluster with IL-1β, IL-10, and IL-12 p70. In the second study, no difference in glutamate levels was observed between AESD and control groups.
Conclusions
CSF glutamate potentially serves as a useful marker for diagnosing AESD. The long-term storage of CSF samples was likely to cause a decay of glutamate in the CSF. Prospective studies using fresh CSF samples are necessary to validate the results in this study.
{"title":"Glutamate in cerebrospinal fluid as a diagnostic marker for acute encephalopathy in childhood","authors":"Kenta Kajiwara , Daiki Setoyama , Kanako Higashi , Tomoko Nomiyama , Yuko Ichimiya , Daichi Kumamoto , Satoshi Akamine , Yuri Sonoda , Pin Fee Chong , Ryuichi Takemoto , Wakato Matsuoka , Soichi Mizuguchi , Noriyuki Kaku , Takahiro A. Kato , Tomohiko Akahoshi , Yuya Kunisaki , Yasunari Sakai , Shouichi Ohga","doi":"10.1016/j.braindev.2025.104448","DOIUrl":"10.1016/j.braindev.2025.104448","url":null,"abstract":"<div><h3>Backgrounds</h3><div>Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is the most frequent form of acute encephalopathy in early childhood in Japan. Magnetic resonance imaging provides useful hallmarks of diagnosing AESD. However, metabolomic profiles for AESD remain elusive. This study investigates whether measurement of amino acids in the cerebrospinal fluid (CSF) is useful for the diagnosis of AESD before onset.</div></div><div><h3>Methods</h3><div>In the first study, CSF samples were collected from patients (11 AESD and 17 controls) admitted to Kyushu University Hospital during 2011–2016. Amino acids in the CSF were analyzed using mass spectrometry. Cytometric bead arrays were used to measure cytokine and chemokine levels in the CSF. The second study was performed by recruiting patients (8 AESD patients and 10 controls) admitted during 2011–2024. CSF samples were stored at −20 °C for 1 month to 12 years.</div></div><div><h3>Results</h3><div>In the first study, glutamate levels in the CSF from AESD patients were higher than in controls and correlated with methionine, threonine, and tyrosine levels. A correlation map of cytokines and amino acids revealed that glutamate formed a cluster with IL-1β, IL-10, and IL-12 p70. In the second study, no difference in glutamate levels was observed between AESD and control groups.</div></div><div><h3>Conclusions</h3><div>CSF glutamate potentially serves as a useful marker for diagnosing AESD. The long-term storage of CSF samples was likely to cause a decay of glutamate in the CSF. Prospective studies using fresh CSF samples are necessary to validate the results in this study.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104448"},"PeriodicalIF":1.3,"publicationDate":"2025-09-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145152058","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}
Pub Date : 2025-09-23DOI: 10.1016/j.braindev.2025.104444
Chi Hou , Yiru Zeng , Wenxiao Wu , Haixia Zhu , Wenlin Wu , Yang Tian, Lianfeng Chen, Wen-Xiong Chen, Yuanyuan Gao, Xiaojing Li
Purpose
To retrospectively analyze the clinical characteristics and relapse factors of children with relapsed anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis in south China.
Methods
Clinical data of children diagnosed with relapsed anti-NMDAR encephalitis in Guangzhou Women and Children's Medical Center from October 2014 to June 2022 were collected. Patients with monophasic disease course in the same follow-up period were randomly selected as controls. Statistical analysis was performed using SPSS IBM 28.0 and figures were graphed using GraphPad Prism 7.01.
Results
A total of 18 children diagnosed with relapsed anti-NMDAR encephalitis (male: female 5:13). The initial onset age was (9.8 ± 3.1) year-old. Relapse interval was 7.0 [interquartile range (IQR) 6.5–15.0] months. Compared with relapses, neurological symptoms were more extensive and mRS scores were higher at the first episode (P = 0.000 in both). Female gender was an independent risk factor for relapse [odds ratio (OR) =0.055, 95 % confidence interval (CI): 0.003–0.907, P = 0.043]. Compared to the patients with monophasic disease course, the relapsed ones were prone to leave neurological sequelae (P = 0.011) at the last follow-up.
Conclusions
Relapses often occur within 1 year after the first episode in children with anti-NMDAR encephalitis. Relapses were milder than first episodes. Female sex is an independent risk factor for relapses. The relapsed individuals were prone to leave neurological sequelae at the last follow-up.
{"title":"Relapse in pediatric anti-N-methyl-d-aspartate receptor encephalitis: A cohort study in one of the national children's medical center in China","authors":"Chi Hou , Yiru Zeng , Wenxiao Wu , Haixia Zhu , Wenlin Wu , Yang Tian, Lianfeng Chen, Wen-Xiong Chen, Yuanyuan Gao, Xiaojing Li","doi":"10.1016/j.braindev.2025.104444","DOIUrl":"10.1016/j.braindev.2025.104444","url":null,"abstract":"<div><h3>Purpose</h3><div>To retrospectively analyze the clinical characteristics and relapse factors of children with relapsed anti-<em>N</em>-methyl-<span>d</span>-aspartate receptor (NMDAR) encephalitis in south China.</div></div><div><h3>Methods</h3><div>Clinical data of children diagnosed with relapsed anti-NMDAR encephalitis in Guangzhou Women and Children's Medical Center from October 2014 to June 2022 were collected. Patients with monophasic disease course in the same follow-up period were randomly selected as controls. Statistical analysis was performed using SPSS IBM 28.0 and figures were graphed using GraphPad Prism 7.01.</div></div><div><h3>Results</h3><div>A total of 18 children diagnosed with relapsed anti-NMDAR encephalitis (male: female 5:13). The initial onset age was (9.8 ± 3.1) year-old. Relapse interval was 7.0 [interquartile range (IQR) 6.5–15.0] months. Compared with relapses, neurological symptoms were more extensive and mRS scores were higher at the first episode (<em>P</em> = 0.000 in both). Female gender was an independent risk factor for relapse [odds ratio (OR) =0.055, 95 % confidence interval (CI): 0.003–0.907, <em>P</em> = 0.043]. Compared to the patients with monophasic disease course, the relapsed ones were prone to leave neurological sequelae (<em>P</em> = 0.011) at the last follow-up.</div></div><div><h3>Conclusions</h3><div>Relapses often occur within 1 year after the first episode in children with anti-NMDAR encephalitis. Relapses were milder than first episodes. Female sex is an independent risk factor for relapses. The relapsed individuals were prone to leave neurological sequelae at the last follow-up.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104444"},"PeriodicalIF":1.3,"publicationDate":"2025-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145120888","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}
Small for gestational age (SGA) and developmental coordination disorder (DCD) are receiving increasing attention in pediatric development. Understanding the risk of DCD, particularly in relation to SGA, would support children's health and development. However, the relationship between SGA and DCD remains unveiled beyond single-cohort studies.
Objectives
This study aimed to integrate findings on DCD from different cohorts within the nationwide prospective Japanese Birth Cohort Consortium (JBiCC).
Study design and subjects
DCD was assessed in children aged 4 to 7 years from three birth cohorts participating in the JBiCC: the Hokkaido Study on Environment and Children's Health (Hokkaido Study), the Hamamatsu Birth Cohort for Mothers and Children (HBC Study), and the Tohoku Medical Megabank Project Birth and Three-Generation Cohort Study (TMM BirThree Cohort Study).
Outcome measures
DCD was assessed using either the Developmental Coordination Disorder Questionnaire Japanese Version (DCDQ-J) or the Ages and Stages Questionnaires Third Edition (ASQ-3). Logistic regression was used to assess the association between SGA and DCD in each cohort. Second, meta-analysis of the association between SGA and DCD defined by DCDQ-J, and individual patient data (IPD) meta-analysis of the association between SGA and DCDQ-J scores were conducted with two cohorts.
Results and conclusions
The analysis included 14,233 children in three cohorts. The individual cohort analyses did not explore statistical significance, except for the TMM BirThree Cohort Study. Meta-synthesis of the Hokkaido and HBC studies showed a β-coefficient of −2.63, 95 % CI [−5.22, −0.03]. IPD analysis of linear regression showed a β-coefficient of −2.76, 95 % CI [−5.38, −0.15]. Our results suggest that SGA may be a potential risk factor for DCD.
{"title":"Small for gestational age as a predictor of developmental coordination disorders: Exploring early risk from Japan birth cohort consortium","authors":"Hiroyoshi Iwata , Maki Tojo , Kenji J. Tsuchiya , Mami Ishikuro , Geng Chen , Satoshi Suyama , Akio Nakai , Naomi Tamura , Toshio Yoshikawa , Toyoki Yamagata , Tomoko Nishimura , Takeshi Yamaguchi , Keiko Yamazaki , Taku Obara , Kazue Ishitsuka , Naho Morisaki , Keitaro Makino , Shinichi Kuriyama , Reiko Kishi","doi":"10.1016/j.braindev.2025.104435","DOIUrl":"10.1016/j.braindev.2025.104435","url":null,"abstract":"<div><h3>Background</h3><div>Small for gestational age (SGA) and developmental coordination disorder (DCD) are receiving increasing attention in pediatric development. Understanding the risk of DCD, particularly in relation to SGA, would support children's health and development. However, the relationship between SGA and DCD remains unveiled beyond single-cohort studies.</div></div><div><h3>Objectives</h3><div>This study aimed to integrate findings on DCD from different cohorts within the nationwide prospective Japanese Birth Cohort Consortium (JBiCC).</div></div><div><h3>Study design and subjects</h3><div>DCD was assessed in children aged 4 to 7 years from three birth cohorts participating in the JBiCC: the Hokkaido Study on Environment and Children's Health (Hokkaido Study), the Hamamatsu Birth Cohort for Mothers and Children (HBC Study), and the Tohoku Medical Megabank Project Birth and Three-Generation Cohort Study (TMM BirThree Cohort Study).</div></div><div><h3>Outcome measures</h3><div>DCD was assessed using either the Developmental Coordination Disorder Questionnaire Japanese Version (DCDQ-J) or the Ages and Stages Questionnaires Third Edition (ASQ-3). Logistic regression was used to assess the association between SGA and DCD in each cohort. Second, meta-analysis of the association between SGA and DCD defined by DCDQ-J, and individual patient data (IPD) meta-analysis of the association between SGA and DCDQ-J scores were conducted with two cohorts.</div></div><div><h3>Results and conclusions</h3><div>The analysis included 14,233 children in three cohorts. The individual cohort analyses did not explore statistical significance, except for the TMM BirThree Cohort Study. Meta-synthesis of the Hokkaido and HBC studies showed a β-coefficient of −2.63, 95 % CI [−5.22, −0.03]. IPD analysis of linear regression showed a β-coefficient of −2.76, 95 % CI [−5.38, −0.15]. Our results suggest that SGA may be a potential risk factor for DCD.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104435"},"PeriodicalIF":1.3,"publicationDate":"2025-09-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145120899","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}
Febrile seizure (FS) is a worrisome event for parents. This study aimed to investigate how stress and perceived stigma are alleviated after experience FS in their children.
Methods
Parents who visited hospitals because of FS exhibited by their children were invited to participate this study. One hundred and sixty-one parents answered the questionnaire (1st Q); among them, 52 parents answered the same questionnaire (2nd Q) after 3 months. The questionnaire consisted of the Impact of Event Scale-Revised (IES-R) and the Parent Stigma Scale (PSS). The scores between the 1st Q and 2nd Q were compared with 52 participants who answered both questionnaires.
Results
The median value of the IES-R total score was 11, and 20 participants showed >24 in the 1st Q (n = 161). IES-R subscales of Intrusion and Hyperarousal symptoms and one item of PSS, “people who know your children have a FS treat your children differently”, decreased significantly (p<0.010, p = 0.013 and p = 0.038, respectively) from the 1st Q (n = 52) to 2nd Q (n = 52). Parents with children having a past history of FS showed significantly higher PSS scores than parents with children having first FS episode (p = 0.029) in the 1st Q (n = 161). Parents who had some knowledge about FS could deal significantly more appropriately with FS attacks compared with those who had no prior knowledge (p<0.001) in both the 1st Q (n = 161) and 2nd Q (n = 52).
Conclusions
Stress and stigma in parents were reduced to some extent 3 months after the FS attacks in their children. Recurrent FS can cause a transient perception of parental stigma. Appropriate education regarding correct management before experiencing the first episode of FS may be important.
{"title":"Alleviating stress and perceived stigma in parents after febrile seizure in their children","authors":"Takuya Tanabe , Mitsuru Kashiwagi , Motoko Ogino , Chizu Oba , Yuki Iai , Chihiro Yamamoto , Tetsuo Kubota , Hideaki Kanemura","doi":"10.1016/j.braindev.2025.104445","DOIUrl":"10.1016/j.braindev.2025.104445","url":null,"abstract":"<div><h3>Objectives</h3><div>Febrile seizure (FS) is a worrisome event for parents. This study aimed to investigate how stress and perceived stigma are alleviated after experience FS in their children.</div></div><div><h3>Methods</h3><div>Parents who visited hospitals because of FS exhibited by their children were invited to participate this study. One hundred and sixty-one parents answered the questionnaire (1st Q); among them, 52 parents answered the same questionnaire (2nd Q) after 3 months. The questionnaire consisted of the Impact of Event Scale-Revised (IES-R) and the Parent Stigma Scale (PSS). The scores between the 1st Q and 2nd Q were compared with 52 participants who answered both questionnaires.</div></div><div><h3>Results</h3><div>The median value of the IES-R total score was 11, and 20 participants showed >24 in the 1st Q (<em>n</em> = 161). IES-R subscales of Intrusion and Hyperarousal symptoms and one item of PSS, “people who know your children have a FS treat your children differently”, decreased significantly (<em>p</em><0.010, <em>p</em> = 0.013 and <em>p</em> = 0.038, respectively) from the 1st Q (<em>n</em> = 52) to 2nd Q (n = 52). Parents with children having a past history of FS showed significantly higher PSS scores than parents with children having first FS episode (<em>p</em> = 0.029) in the 1st Q (<em>n</em> = 161). Parents who had some knowledge about FS could deal significantly more appropriately with FS attacks compared with those who had no prior knowledge (<em>p</em><0.001) in both the 1st Q (<em>n</em> = 161) and 2nd Q (<em>n</em> = 52).</div></div><div><h3>Conclusions</h3><div>Stress and stigma in parents were reduced to some extent 3 months after the FS attacks in their children. Recurrent FS can cause a transient perception of parental stigma. Appropriate education regarding correct management before experiencing the first episode of FS may be important.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104445"},"PeriodicalIF":1.3,"publicationDate":"2025-09-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145105043","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}
Pub Date : 2025-09-18DOI: 10.1016/j.braindev.2025.104451
Christian Messina
{"title":"Decoding the pathophysiological role of fukutin in Fukuyama congenital muscular dystrophy","authors":"Christian Messina","doi":"10.1016/j.braindev.2025.104451","DOIUrl":"10.1016/j.braindev.2025.104451","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104451"},"PeriodicalIF":1.3,"publicationDate":"2025-09-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145093135","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}
Pub Date : 2025-09-17DOI: 10.1016/j.braindev.2025.104434
Mieko Hirose
Background
Children with autism spectrum disorder (ASD) often face difficulties in parent-child relationships and exhibit problematic behaviors. This study retrospectively examines the effects of standard Parent-Child Interaction Therapy (PCIT) for children with ASD and their caregivers in Japan, focusing on reducing parental stress and children's problematic behaviors.
Methods
Eight parent-child dyads with children aged 2.5–7 years with ASD underwent standard PCIT. Measures included the Eyberg Child Behavior Inventory (ECBI), Beck Depression Inventory-II (BDI-II), Parenting Stress Index-Short Form (PSI-SF), and Child Behavior Checklist for Ages 4–18 (CBCL/4–18), assessed pre- and post-treatments.
Results
For parents, ECBI problem score decreased from 16.8 (SD ± 4.4) to 3.0 (SD ± 4.1) pre- and post-treatment (p < 0.05, effect size 1.9). PSI-SF parent section scores dropped from 28.7 (SD ± 7.1) to 22.1 (SD ± 6.2) (p < 0.01, effect size 2.0). The total PSI-SF score from 58.1 (SD ± 10.9) to 45.0 (SD ± 11.1), (p < 0.01, effect size 1.7). For children, ECBI intensity scores decreased from 147.9 (SD ± 20.3) to 85.5 (SD ± 17.7) (p < 0.01, effect size 3.4). PSI-SF child section scores fell from 29.4 (SD ± 4.8) to 22.9 (SD ± 5.8) (p < 0.05, effect size of 1.2), and total CBCL from 70.3 (SD ± 5.9) to 62.0 (SD ± 8.4) (p < 0.05, effect size of 1.5).
Conclusion
The standard PCIT for children with ASD and their parents in Japan significantly reduces parental stress and children's problematic behaviors, improving parent-child interactions.
背景：自闭症谱系障碍（ASD）儿童在亲子关系中经常面临困难，并表现出问题行为。本研究回顾性研究了标准亲子互动疗法（PCIT）对日本自闭症儿童及其照顾者的影响，重点是减少父母的压力和儿童的问题行为。方法：8对2.5 ~ 7岁ASD患儿进行标准PCIT治疗。测量方法包括Eyberg儿童行为量表（ECBI）、Beck抑郁量表- ii （BDI-II）、父母压力指数简表（PSI-SF）和4-18岁儿童行为检查表（CBCL/4-18），对治疗前后进行评估。结果：家长的ECBI问题评分从治疗前后的16.8 （SD±4.4）降至3.0 (SD±4.1)(p)。结论：日本ASD儿童及其家长的标准PCIT显著降低了父母的压力和儿童的问题行为，改善了亲子互动。
{"title":"Parent-child interaction therapy as a therapeutic approach for children with autism spectrum disorder in Japan","authors":"Mieko Hirose","doi":"10.1016/j.braindev.2025.104434","DOIUrl":"10.1016/j.braindev.2025.104434","url":null,"abstract":"<div><h3>Background</h3><div>Children with autism spectrum disorder (ASD) often face difficulties in parent-child relationships and exhibit problematic behaviors. This study retrospectively examines the effects of standard Parent-Child Interaction Therapy (PCIT) for children with ASD and their caregivers in Japan, focusing on reducing parental stress and children's problematic behaviors.</div></div><div><h3>Methods</h3><div>Eight parent-child dyads with children aged 2.5–7 years with ASD underwent standard PCIT. Measures included the Eyberg Child Behavior Inventory (ECBI), Beck Depression Inventory-II (BDI-II), Parenting Stress Index-Short Form (PSI-SF), and Child Behavior Checklist for Ages 4–18 (CBCL/4–18), assessed pre- and post-treatments.</div></div><div><h3>Results</h3><div>For parents, ECBI problem score decreased from 16.8 (SD ± 4.4) to 3.0 (SD ± 4.1) pre- and post-treatment (<em>p</em> < 0.05, effect size 1.9). PSI-SF parent section scores dropped from 28.7 (SD ± 7.1) to 22.1 (SD ± 6.2) (<em>p</em> < 0.01, effect size 2.0). The total PSI-SF score from 58.1 (SD ± 10.9) to 45.0 (SD ± 11.1), (<em>p</em> < 0.01, effect size 1.7). For children, ECBI intensity scores decreased from 147.9 (SD ± 20.3) to 85.5 (SD ± 17.7) (<em>p</em> < 0.01, effect size 3.4). PSI-SF child section scores fell from 29.4 (SD ± 4.8) to 22.9 (SD ± 5.8) (<em>p</em> < 0.05, effect size of 1.2), and total CBCL from 70.3 (SD ± 5.9) to 62.0 (SD ± 8.4) (<em>p</em> < 0.05, effect size of 1.5).</div></div><div><h3>Conclusion</h3><div>The standard PCIT for children with ASD and their parents in Japan significantly reduces parental stress and children's problematic behaviors, improving parent-child interactions.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104434"},"PeriodicalIF":1.3,"publicationDate":"2025-09-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145088441","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}
Pub Date : 2025-09-16DOI: 10.1016/j.braindev.2025.104446
Saalim Shahid, Muhammad Mudasir Atif
{"title":"AESD and vitamin therapy: The need for biomarkers and follow-up","authors":"Saalim Shahid, Muhammad Mudasir Atif","doi":"10.1016/j.braindev.2025.104446","DOIUrl":"10.1016/j.braindev.2025.104446","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104446"},"PeriodicalIF":1.3,"publicationDate":"2025-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145082575","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}
Pub Date : 2025-09-16DOI: 10.1016/j.braindev.2025.104440
Christian Messina
{"title":"When details matter: Critical considerations in the study of meningitis","authors":"Christian Messina","doi":"10.1016/j.braindev.2025.104440","DOIUrl":"10.1016/j.braindev.2025.104440","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104440"},"PeriodicalIF":1.3,"publicationDate":"2025-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145082607","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}
Cerebral palsy (CP) is the most common motor disorder in childhood that causes lifelong disabilities. Studies suggest that administration of autologous cord blood (CB) or sibling cord blood (SCB) may improve gross motor function and brain connectivity in CP. In this pilot study, we evaluated the safety and efficacy of allogeneic SCB in Japanese children with CP.
Methods
A single-arm pilot study of a single intravenous dose of HLA-matched or partially matched (at least 4/6 HLA molecular matches) SCB was conducted in five Japanese patients with CP (5.0–6.3 years old). The primary endpoint was to measure the safety profiles and assessment of motor function and neurodevelopmental behaviors as the secondary endpoint.
Results
No serious side effects or concerning infusion reactions were observed. All patients showed a mean improvement of 4.89 ± 4.01 points in Gross Motor Function Measure-66 (GMFM-66) score at 6 months, which was better than predicted by age and severity of disease. In addition, GMFM-66 scores at 1 and 2 years was further improved by a mean of 6.49 ± 3.58 and 7.93 ± 4.26 points, respectively. One case showed improvement in the Developmental Quotient (DQ) overall and language/social scales and another case showed improvement in the Wechsler Intelligence Scale for Children, Fourth Edition (WISC-IV) overall, language. No significant correlations were found between GMFM-66 scores, DQ scales, WISC-IV scores and HLA concordance or number of cells administered.
Conclusion
Allogeneic SCB administration is safe and feasible in Japanese CP patients and may offer therapeutic potential in clinical practice. Future study is needed to clarify several unclear issues.
{"title":"Safety and feasibility of allogeneic sibling cord blood infusion in Japanese children with cerebral palsy: A single-center pilot study","authors":"Shiho Saitoh , Hiroaki Kikuchi , Takuzo Marukane , Terumasa Tsuno , Rina Hosoda , Nobuyasu Baba , Feifei Wang , Yumi Kuroiwa , Ryuhei Nagai , Masayuki Tsuda , Nagamasa Maeda , Yusuke Sagara , Mikiya Fujieda","doi":"10.1016/j.braindev.2025.104443","DOIUrl":"10.1016/j.braindev.2025.104443","url":null,"abstract":"<div><h3>Introduction</h3><div>Cerebral palsy (CP) is the most common motor disorder in childhood that causes lifelong disabilities. Studies suggest that administration of autologous cord blood (CB) or sibling cord blood (SCB) may improve gross motor function and brain connectivity in CP. In this pilot study, we evaluated the safety and efficacy of allogeneic SCB in Japanese children with CP.</div></div><div><h3>Methods</h3><div>A single-arm pilot study of a single intravenous dose of HLA-matched or partially matched (at least 4/6 HLA molecular matches) SCB was conducted in five Japanese patients with CP (5.0–6.3 years old). The primary endpoint was to measure the safety profiles and assessment of motor function and neurodevelopmental behaviors as the secondary endpoint.</div></div><div><h3>Results</h3><div>No serious side effects or concerning infusion reactions were observed. All patients showed a mean improvement of 4.89 ± 4.01 points in Gross Motor Function Measure-66 (GMFM-66) score at 6 months, which was better than predicted by age and severity of disease. In addition, GMFM-66 scores at 1 and 2 years was further improved by a mean of 6.49 ± 3.58 and 7.93 ± 4.26 points, respectively. One case showed improvement in the Developmental Quotient (DQ) overall and language/social scales and another case showed improvement in the Wechsler Intelligence Scale for Children, Fourth Edition (WISC-IV) overall, language. No significant correlations were found between GMFM-66 scores, DQ scales, WISC-IV scores and HLA concordance or number of cells administered.</div></div><div><h3>Conclusion</h3><div>Allogeneic SCB administration is safe and feasible in Japanese CP patients and may offer therapeutic potential in clinical practice. Future study is needed to clarify several unclear issues.</div></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":"47 5","pages":"Article 104443"},"PeriodicalIF":1.3,"publicationDate":"2025-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145082597","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}
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