Pub Date : 2023-09-01DOI: 10.1016/j.braindev.2023.05.001
Claire E. Manley , Kerri Walter , Serena Micheletti , Matthew Tietjen , Emily Cantillon , Elisa M. Fazzi , Peter J. Bex , Lotfi B. Merabet
Individuals with cerebral visual impairment (CVI) have difficulties identifying common objects, especially when presented as cartoons or abstract images. In this study, participants were shown a series of images of ten common objects, each from five possible categories ranging from abstract black & white line drawings to color photographs. Fifty individuals with CVI and 50 neurotypical controls verbally identified each object and success rates and reaction times were collected. Visual gaze behavior was recorded using an eye tracker to quantify the extent of visual search area explored and number of fixations. A receiver operating characteristic (ROC) analysis was also carried out to compare the degree of alignment between the distribution of individual eye gaze patterns and image saliency features computed by the graph-based visual saliency (GBVS) model. Compared to controls, CVI participants showed significantly lower success rates and longer reaction times when identifying objects. In the CVI group, success rate improved moving from abstract black & white images to color photographs, suggesting that object form (as defined by outlines and contours) and color are important cues for correct identification. Eye tracking data revealed that the CVI group showed significantly greater visual search areas and number of fixations per image, and the distribution of eye gaze patterns in the CVI group was less aligned with the high saliency features of the image compared to controls. These results have important implications in helping to understand the complex profile of visual perceptual difficulties associated with CVI.
{"title":"Object identification in cerebral visual impairment characterized by gaze behavior and image saliency analysis","authors":"Claire E. Manley , Kerri Walter , Serena Micheletti , Matthew Tietjen , Emily Cantillon , Elisa M. Fazzi , Peter J. Bex , Lotfi B. Merabet","doi":"10.1016/j.braindev.2023.05.001","DOIUrl":"10.1016/j.braindev.2023.05.001","url":null,"abstract":"<div><p><span>Individuals with cerebral visual impairment (CVI) have difficulties identifying common objects, especially when presented as cartoons or abstract images. In this study, participants were shown a series of images of ten common objects, each from five possible categories ranging from abstract black & white line drawings to color </span>photographs<span>. Fifty individuals with CVI and 50 neurotypical controls verbally identified each object and success rates and reaction times were collected. Visual gaze behavior was recorded using an eye tracker to quantify the extent of visual search area explored and number of fixations. A receiver operating characteristic (ROC) analysis was also carried out to compare the degree of alignment between the distribution of individual eye gaze patterns and image saliency features computed by the graph-based visual saliency (GBVS) model. Compared to controls, CVI participants showed significantly lower success rates and longer reaction times when identifying objects. In the CVI group, success rate improved moving from abstract black & white images to color photographs, suggesting that object form (as defined by outlines and contours) and color are important cues for correct identification. Eye tracking data revealed that the CVI group showed significantly greater visual search areas and number of fixations per image, and the distribution of eye gaze patterns in the CVI group was less aligned with the high saliency features of the image compared to controls. These results have important implications in helping to understand the complex profile of visual perceptual difficulties associated with CVI.</span></p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10524860/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9984500","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}
Pub Date : 2023-09-01DOI: 10.1016/j.braindev.2023.06.002
Yuko Shimizu-Motohashi, Hirofumi Komaki
{"title":"Reply to letter to the editor “Regarding nusinersen and other therapeutic strategies for improved motor function”","authors":"Yuko Shimizu-Motohashi, Hirofumi Komaki","doi":"10.1016/j.braindev.2023.06.002","DOIUrl":"10.1016/j.braindev.2023.06.002","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9992579","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 : 2023-09-01DOI: 10.1016/j.braindev.2023.05.004
Jessica Rossi , Marco Russo , Giuseppe Gobbi , Alessandra Terracciano , Roberta Zuntini , Stefano Giuseppe Caraffi , Antonio Novelli , Livia Garavelli , Franco Valzania , Romana Rizzi
Introduction
Raynaud-Claes syndrome is a very rare X-linked condition, characterized by intellectual disability, impaired language development, brain abnormalities, facial dysmorphisms and drug-resistant epilepsy. It is caused by loss-of-function variants in the CLCN4 gene, which encodes the 2Cl−/H + exchanger ClC-4, prominently expressed in the hippocampus and cerebellum. Different genotypic variants have been described, each exhibiting specific phenotypic characteristics. The loss-of-function variant p.Gly544Arg in the CLCN4 gene has been described in only two male probands, but there are no reports on phenotypic characterization in females.
Case presentation
We present a 30-year-old Italian woman with early-onset drug-resistant epilepsy, developmental and epileptic encephalopathy, developmental delay, absence of verbal language development, behavioral impairment with autistic features, and clusters of seizures during catamenial periods. The interictal EEG showed slight inconstant slowing of the background rhythm, with abnormal frontal predominant mu like rhythm and generalized spike and polyspike wave discharges, which increased in frequency during drowsiness. A brain MRI showed slight cranio-encephalic asymmetry and a smaller size of the left hippocampus. The whole exome sequencing (WES) revealed a de novo heterozygous c.1630G > A variant in the CLCN4 gene, resulting in the amino acid substitution p.Gly544Arg (rs587777161), consistent with Raynaud-Claes syndrome.
Discussion and conclusion
Our patient is the first case of a de novo p.Gly544Arg variant of the CLCN4 gene in a female proband, confirming that female patients with Raynaud-Claes syndrome can be as severely affected as the male counterparts. Our case expands the phenotypic characterization of different genotypic CLCN4 variants, which can become crucial in the future for early diagnosis if targeted therapy becomes available.
{"title":"Developmental and epileptic encephalopathy in a young Italian woman with a de novo missense variant in the CLCN4 gene: A case report","authors":"Jessica Rossi , Marco Russo , Giuseppe Gobbi , Alessandra Terracciano , Roberta Zuntini , Stefano Giuseppe Caraffi , Antonio Novelli , Livia Garavelli , Franco Valzania , Romana Rizzi","doi":"10.1016/j.braindev.2023.05.004","DOIUrl":"10.1016/j.braindev.2023.05.004","url":null,"abstract":"<div><h3>Introduction</h3><p>Raynaud-Claes syndrome is a very rare X-linked condition, characterized by intellectual disability, impaired language development, brain abnormalities, facial dysmorphisms and drug-resistant epilepsy. It is caused by loss-of-function variants in the <em>CLCN4</em> gene, which encodes the 2Cl−/H + exchanger <em>ClC-4</em>, prominently expressed in the hippocampus and cerebellum. Different genotypic variants have been described, each exhibiting specific phenotypic characteristics. The loss-of-function variant p.Gly544Arg in the <em>CLCN4</em> gene has been described in only two male probands, but there are no reports on phenotypic characterization in females.</p></div><div><h3>Case presentation</h3><p>We present a 30-year-old Italian woman with early-onset drug-resistant epilepsy, developmental and epileptic encephalopathy, developmental delay, absence of verbal language development, behavioral impairment with autistic features, and clusters of seizures during catamenial periods. The interictal EEG showed slight inconstant slowing of the background rhythm, with abnormal frontal predominant mu like rhythm and generalized spike and polyspike wave discharges, which increased in frequency during drowsiness. A brain MRI showed slight cranio-encephalic asymmetry and a smaller size of the left hippocampus. The whole exome sequencing (WES) revealed a <em>de novo</em> heterozygous c.1630G > A variant in the <em>CLCN4</em> gene, resulting in the amino acid substitution p.Gly544Arg (rs587777161), consistent with Raynaud-Claes syndrome.</p></div><div><h3>Discussion and conclusion</h3><p>Our patient is the first case of a de novo p.Gly544Arg variant of the <em>CLCN4</em> gene in a female proband, confirming that female patients with Raynaud-Claes syndrome can be as severely affected as the male counterparts. Our case expands the phenotypic characterization of different genotypic <em>CLCN4</em> variants, which can become crucial in the future for early diagnosis if targeted therapy becomes available.</p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10008597","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}
Variants in the GNB1 gene, which encodes the β1 subunit of a trimeric G protein, can cause moderate to severe psychomotor retardation. Acute encephalopathies have also been observed in patients with central nervous system abnormalities; however, severe neurological sequelae have not previously been reported.
Case presentations
Patient 1 was a Japanese female with a de novo GNB1 variant (c.284 T > C). At 8 months old she contracted influenza A and developed generalized convulsions. In the acute phase, brain magnetic resonance imaging (MRI) findings indicated acute encephalopathy; diffuse cerebral atrophy was present 1 month later. Although multidisciplinary treatment was administered, she had severe neurological sequelae including spastic tetraplegia, severe intellectual disabilities, and refractory epilepsy. Patient 2 was a Japanese male with a de novo GNB1 variant (c.239 T > C). He experienced an unexplained respiratory arrest aged 17 years; refractory convulsions developed. Brain MRI at 1 month showed bilateral basal ganglia high intensities; at 3 months, diffuse cerebral cortex and white matter atrophy was observed. Despite multidisciplinary treatment, he developed severe spastic tetraplegia and mental regression.
Discussion
We report two patients with GNB1 variants who had acute lesions on brain MRI and unexpected disease courses. In such patients with acute neurological deterioration, multidisciplinary treatment is required; patients should also be carefully observed for progression to acute encephalopathy.
{"title":"Atypical clinical course in two patients with GNB1 variants who developed acute encephalopathy","authors":"Megumi Tsuji , Azusa Ikeda , Yu Tsuyusaki , Mizue Iai , Kenji Kurosawa , Kenjiro Kosaki , Tomohide Goto","doi":"10.1016/j.braindev.2023.06.005","DOIUrl":"10.1016/j.braindev.2023.06.005","url":null,"abstract":"<div><h3>Introduction</h3><p>Variants in the <em>GNB1</em> gene, which encodes the β1 subunit of a trimeric G protein, can cause moderate to severe psychomotor retardation. Acute encephalopathies have also been observed in patients with central nervous system abnormalities; however, severe neurological sequelae have not previously been reported.</p></div><div><h3>Case presentations</h3><p>Patient 1 was a Japanese female with a <em>de novo GNB1</em> variant (c.284 T > C). At 8 months old she contracted influenza A and developed generalized convulsions. In the acute phase, brain magnetic resonance imaging (MRI) findings indicated acute encephalopathy; diffuse cerebral atrophy was present 1 month later. Although multidisciplinary treatment was administered, she had severe neurological sequelae including spastic tetraplegia, severe intellectual disabilities, and refractory epilepsy. Patient 2 was a Japanese male with a <em>de novo GNB1</em> variant (c.239 T > C). He experienced an unexplained respiratory arrest aged 17 years; refractory convulsions developed. Brain MRI at 1 month showed bilateral basal ganglia high intensities; at 3 months, diffuse cerebral cortex and white matter atrophy was observed. Despite multidisciplinary treatment, he developed severe spastic tetraplegia and mental regression.</p></div><div><h3>Discussion</h3><p>We report two patients with <em>GNB1</em> variants who had acute lesions on brain MRI and unexpected disease courses. In such patients with acute neurological deterioration, multidisciplinary treatment is required; patients should also be carefully observed for progression to acute encephalopathy.</p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10357451","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 : 2023-09-01DOI: 10.1016/j.braindev.2023.07.001
Yoshihiro Watanabe, Mao Odaka, Hirotaka Motoi, Yoshitaka Oyama, Kentaro Shiga, Shuichi Ito
{"title":"Reply to the letter “Regarding investigation of prognostic factors for HHV 6/7-associated acute encephalopathy”","authors":"Yoshihiro Watanabe, Mao Odaka, Hirotaka Motoi, Yoshitaka Oyama, Kentaro Shiga, Shuichi Ito","doi":"10.1016/j.braindev.2023.07.001","DOIUrl":"10.1016/j.braindev.2023.07.001","url":null,"abstract":"","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9986856","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}
Cerebellitis is a rare complication of clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS); however, MERS with cerebellitis is associated with a higher risk of neurological sequelae in comparison to MERS alone. Although the disease is difficult to diagnose by conventional MRI in the early disease phase, arterial spin labeling (ASL), a noninvasive MRI perfusion technique using magnetically-labeled arterial blood water protons, is considered promising.
Case report
We experienced three cases of MERS with cerebellitis. Diffusion-weighted imaging showed a high-intensity lesion at the splenium of the corpus callosum. ASL showed increased blood flow in the cerebellum in all three cases, despite cerebellar symptoms being inapparent or difficult to notice in the early phase of disease in all cases. Patients received methylprednisolone pulse therapy and intravenous immunoglobulin from the early phase of the disease and recovered without neurological sequelae.
Discussion
ASL magnetic response imaging simultaneously showed an area of hyperperfusion in the cerebellum. At the same time, the apparent diffusion coefficient of the splenial lesion was decreased in all three cases. The successful diagnosis of cerebellitis in the acute phase led to early therapeutic intervention, which may be important for this condition. We report the usefulness of ASL and review the relevant literature on MERS with cerebellitis.
{"title":"Usefulness of arterial spin labeling imaging, which contributed to the early detection of cerebellitis complicated by clinically mild encephalitis/encephalopathy with a reversible splenial lesion: Lessons from three cases","authors":"Nanako Nishiguchi , Tatsuharu Sato , Kazuhiko Hashimoto , Takuya Hayashida , Kouhei Haraguchi , Reiko Ideguchi , Hiroyuki Moriuchi","doi":"10.1016/j.braindev.2023.05.003","DOIUrl":"10.1016/j.braindev.2023.05.003","url":null,"abstract":"<div><h3>Background</h3><p><span><span>Cerebellitis is a rare complication of clinically mild encephalitis/encephalopathy with a reversible splenial lesion (MERS); however, MERS with cerebellitis is associated with a higher risk of neurological sequelae in comparison to MERS alone. Although the disease is difficult to diagnose by conventional MRI in the early disease phase, </span>arterial spin labeling (ASL), a noninvasive </span>MRI perfusion<span> technique using magnetically-labeled arterial blood water protons, is considered promising.</span></p></div><div><h3>Case report</h3><p><span><span>We experienced three cases of MERS with cerebellitis. Diffusion-weighted imaging showed a high-intensity lesion at the splenium of the </span>corpus callosum<span>. ASL showed increased blood flow in the cerebellum in all three cases, despite cerebellar symptoms being inapparent or difficult to notice in the early phase of disease in all cases. Patients received </span></span>methylprednisolone<span> pulse therapy and intravenous immunoglobulin from the early phase of the disease and recovered without neurological sequelae.</span></p></div><div><h3>Discussion</h3><p>ASL magnetic response imaging simultaneously showed an area of hyperperfusion in the cerebellum. At the same time, the apparent diffusion coefficient of the splenial lesion was decreased in all three cases. The successful diagnosis of cerebellitis in the acute phase led to early therapeutic intervention, which may be important for this condition. We report the usefulness of ASL and review the relevant literature on MERS with cerebellitis.</p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9987042","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}
Transcutaneous auricular vagus nerve stimulation (taVNS) was performed in two patients suffering structural focal epilepsy with preserved intellectual ability to show the feasibility of taVNS for specific patient groups.
Case Presentations
Patient 1 was a 24-year-old woman with frontal lobe epilepsy who had weekly hyperkinetic seizures despite multiple anti-seizure medications. Patient 2 was a 27-year-old woman with parietal lobe epilepsy and focal cortical dysplasia in the vicinity of the lipoma in the corpus callosum. She experienced weekly focal-impaired awareness seizures even with anti-seizure medication. taVNS was applied to the left earlobe of both patients at 1.5 mA, 25 Hz, 250 μs pulse width, and 30 s stimulation with 30 s rest for 4 h per day. Over an 8-week baseline and 20 weeks of stimulation, the rate of reduction in seizure frequency was evaluated, along with quality-of-life using the Short-Form 36-Item Health survey.
Results
At baseline, we measured up to 11 and 12 focal seizures per week in Patient 1 and 2, respectively, with both patients achieving seizure freedom after 4 and 20 weeks taVNS, respectively. Patient 1 and 2 were observed for 18 and 14 months, respectively, including the clinical trial and follow-up observation period. Quality-of-life ratings increased in both patients, and no significant adverse events occurred during the study period. During the maintenance period after 20 weeks, seizures remained absent in Patient 1, and seizures remained reduced in Patient 2.
Conclusion
Our results demonstrate that taVNS may be a promising tool for structural focal epilepsy with preserved cognitive function. A multicenter double-blind clinical trial is needed to confirm the role of taVNS as an anti-seizure tool.
{"title":"Transcutaneous auricular vagus nerve stimulation therapy in patients with cognitively preserved structural focal epilepsy: A case series report","authors":"Hideaki Shiraishi , Kiyoshi Egawa , Kaoru Murakami , Midori Nakajima , Yuki Ueda , Sachiko Nakakubo , Masashi Narugami , Shuhei Kimura , Takeru Goto , Yasuyoshi Hiramatsu , Masaaki Murakami","doi":"10.1016/j.braindev.2023.08.007","DOIUrl":"10.1016/j.braindev.2023.08.007","url":null,"abstract":"<div><h3>Objective</h3><p>Transcutaneous auricular vagus nerve stimulation (taVNS) was performed in two patients suffering structural focal epilepsy with preserved intellectual ability to show the feasibility of taVNS for specific patient groups.</p></div><div><h3>Case Presentations</h3><p>Patient 1 was a 24-year-old woman with frontal lobe epilepsy who had weekly hyperkinetic seizures despite multiple anti-seizure medications. Patient 2 was a 27-year-old woman with parietal lobe epilepsy and focal cortical dysplasia in the vicinity of the lipoma in the corpus callosum. She experienced weekly focal-impaired awareness seizures even with anti-seizure medication. taVNS was applied to the left earlobe of both patients at 1.5 mA, 25 Hz, 250 μs pulse width, and 30 s stimulation with 30 s rest for 4 h per day. Over an 8-week baseline and 20 weeks of stimulation, the rate of reduction in seizure frequency was evaluated, along with quality-of-life using the Short-Form 36-Item Health survey.</p></div><div><h3>Results</h3><p>At baseline, we measured up to 11 and 12 focal seizures per week in Patient 1 and 2, respectively, with both patients achieving seizure freedom after 4 and 20 weeks taVNS, respectively. Patient 1 and 2 were observed for 18 and 14 months, respectively, including the clinical trial and follow-up observation period. Quality-of-life ratings increased in both patients, and no significant adverse events occurred during the study period. During the maintenance period after 20 weeks, seizures remained absent in Patient 1, and seizures remained reduced in Patient 2.</p></div><div><h3>Conclusion</h3><p>Our results demonstrate that taVNS may be a promising tool for structural focal epilepsy with preserved cognitive function. A multicenter double-blind clinical trial is needed to confirm the role of taVNS as an anti-seizure tool.</p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S0387760423001481/pdfft?md5=9834d2b662cc41890282c8221cd358f3&pid=1-s2.0-S0387760423001481-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10135747","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}
Pub Date : 2023-08-30DOI: 10.1016/j.braindev.2023.08.002
Yoko Matsumoto , Yuji Yoshii , Akiyo Ikutomo , Mariko Yagi , Mio Nishimura , Yoko Kawasaki , Amanda Sarafian , Heakyung Kim , David P. Roye Jr. , Hiroko Matsumoto
Background
Pediatric stroke is a rare medical condition that often leads to long-lasting motor and cognitive impairments. Although therapies for adults after a stroke are well described, treatments for motor deficits following a pediatric stroke are yet to be investigated. We report a case of pediatric stroke in the chronic phase, in which a combination of novel treatments resulted in a significant improvement in physical function.
Case report
A seven-year-old girl with a left hemispheric cerebral infarction lost almost all right upper extremity motor function. Following onabotulinumtoxinA treatment, she underwent hand-arm bimanual intensive therapy augmented with a hybrid assistive limb for 90 h over 15 days. Evaluation after the training revealed significant improvements in physical function, daily activities, and occupational performance.
Conclusions
This report highlights the importance of innovative combinations of techniques in the treatment of pediatric stroke.
{"title":"Improvement in a post-stroke pediatric patient with hemiplegia: Use of a hand-arm bimanual intensive therapy with hybrid assistive limb","authors":"Yoko Matsumoto , Yuji Yoshii , Akiyo Ikutomo , Mariko Yagi , Mio Nishimura , Yoko Kawasaki , Amanda Sarafian , Heakyung Kim , David P. Roye Jr. , Hiroko Matsumoto","doi":"10.1016/j.braindev.2023.08.002","DOIUrl":"10.1016/j.braindev.2023.08.002","url":null,"abstract":"<div><h3>Background</h3><p>Pediatric stroke is a rare medical condition that often leads to long-lasting motor and cognitive impairments. Although therapies for adults after a stroke are well described, treatments for motor deficits following a pediatric stroke are yet to be investigated. We report a case of pediatric stroke in the chronic phase, in which a combination of novel treatments resulted in a significant improvement in physical function.</p></div><div><h3>Case report</h3><p>A seven-year-old girl with a left hemispheric cerebral infarction lost almost all right upper extremity motor function. Following onabotulinumtoxinA treatment, she underwent hand-arm bimanual intensive therapy augmented with a hybrid assistive limb for 90 h over 15 days. Evaluation after the training revealed significant improvements in physical function, daily activities, and occupational performance.</p></div><div><h3>Conclusions</h3><p>This report highlights the importance of innovative combinations of techniques in the treatment of pediatric stroke.</p></div>","PeriodicalId":56137,"journal":{"name":"Brain & Development","volume":null,"pages":null},"PeriodicalIF":1.7,"publicationDate":"2023-08-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S0387760423001286/pdfft?md5=1c09cddd2c3f243da7e2b551113a0639&pid=1-s2.0-S0387760423001286-main.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10132033","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}