Annals of the Child Neurology Society最新文献

{"title":"Tacrolimus-Related Neurotoxicity of the Pons in Children: Review of the Literature and a Case Report","authors":"Amy Hill,&nbsp;Mohamed Bilal Haradwala,&nbsp;Jean-Baptiste Le Pichon","doi":"10.1002/cns3.70000","DOIUrl":"https://doi.org/10.1002/cns3.70000","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Introduction</h3>\u0000 \u0000 <p>Tacrolimus is a potent immunosuppressive agent effective in preventing solid organ transplant rejection. It is widely used following allogeneic liver, kidney, heart, and bone marrow transplantation. Tacrolimus-related neurotoxicity, which can present in up to one-third of patients, manifests with a broad clinical spectrum. Neuroradiological features are classically reported as bilateral and symmetrical lesions involving the parietal and occipital lobes, similar to posterior reversible encephalopathy syndrome. Tacrolimus-related toxicity can also affect other parts of the brain, including the brainstem, although isolated brainstem involvement is rare.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>This report describes a patient who had tacrolimus-related neurotoxicity with an isolated brainstem lesion in which symptoms resolved with only a brief hold of the tacrolimus. A literature review identified four other pediatric patients who had tacrolimus-associated neurotoxicity with isolated brainstem involvement.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Discussion</h3>\u0000 \u0000 <p>Tacrolimus-associated neurotoxicity with pontine lesions in children is rare. In previously reported patients, tacrolimus was discontinued and neurological symptoms resolved. Our patient developed tacrolimus-associated clinical changes and pontine lesions that improved following a brief hold of the tacrolimus treatment. This girl highlights tacrolimus-associated neurotoxicity isolated to the brainstem in pediatric patients and demonstrates that tacrolimus may be safely restarted with careful monitoring and follow-up.</p>\u0000 </section>\u0000 </div>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"41-45"},"PeriodicalIF":0.0,"publicationDate":"2025-03-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.70000","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688783","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Quantifying age-related changes in mirror overflow in children and adolescents with attention-deficit/hyperactivity disorder","authors":"Beatrice Ojuri,&nbsp;Deana Crocetti,&nbsp;Evan Bucklin,&nbsp;Stewart H. Mostofsky,&nbsp;Joshua B. Ewen","doi":"10.1002/cns3.20100","DOIUrl":"https://doi.org/10.1002/cns3.20100","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>Children with attention-deficit/hyperactivity disorder (ADHD) show excessive mirror overflow (particularly in the nondominant hand); however, patterns of age-related decrease of overflow remain unclear. This study aimed to quantify age-related changes in mirror overflow in youth with and without ADHD.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Average mirror overflow was examined during left-hand finger tapping (LHFT; nondominant finger tapping) and right-hand finger tapping (RHFT; dominant finger tapping) using electronic finger twitch transducers in a cross-sectional sample of youth with ADHD (<i>n</i> = 77) and typically developing (TD) youth (<i>n</i> = 75) ages 8–18 years. Effects of age and ADHD diagnosis on LHFT, RHFT, and a summed “total” overflow (TOF) across hands were examined across the sample age range and within childhood (8–12 years) and adolescence (13–18 years).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>ADHD youth showed a decrease in overflow with age, including a large effect for TOF, with a very large age effect for LHFT but a more moderate age effect for RHFT. TD youth showed a moderate decrease in overflow with age for TOF, with a large decrease for LHFT but no significant decrease for RHFT. Additionally, we found that large effects of ADHD-related excessive overflow in childhood diminished in adolescence.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>Findings suggest that mirror overflow in ADHD youth diminishes into adolescence but does not resolve completely, suggesting ADHD-associated increased mirror overflow may reflect both a developmentally resolving effect and a somewhat persistent atypicality. Future studies with larger and longitudinal samples would provide additional insight into mechanisms contributing to excessive mirror overflow and its relationship to both clinical and neurobiological aspects of ADHD-associated disinhibition.</p>\u0000 </section>\u0000 </div>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"16-25"},"PeriodicalIF":0.0,"publicationDate":"2025-03-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20100","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688821","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Anti-NMDA Receptor Encephalitis and Mycoplasma pneumoniae Infection With Demyelination","authors":"Leah Loerinc,&nbsp;Jenny Lin,&nbsp;David S. Wolf,&nbsp;Grace Gombolay","doi":"10.1002/cns3.70001","DOIUrl":"https://doi.org/10.1002/cns3.70001","url":null,"abstract":"&lt;p&gt;While most children with anti-&lt;i&gt;N&lt;/i&gt;-methyl-&lt;span&gt;d&lt;/span&gt;-aspartate (NMDA) receptor encephalitis (NMDARE) have normal brain magnetic resonance imaging (MRI) [&lt;span&gt;1&lt;/span&gt;], 3% have demyelinating lesions on MRI [&lt;span&gt;2&lt;/span&gt;]. We describe a patient who had NMDARE and MRI lesions resembling multiple sclerosis (MS).&lt;/p&gt;&lt;p&gt;This 16-year-old girl with a history of major depressive disorder presented with 1 month of altered behavior with hyper-religiosity and insomnia. She was admitted to an inpatient psychiatric facility and was started on antipsychotic and antidepressant medications without improvement and was transferred to our facility. On presentation, she was awake but would not regard. She was nonverbal and did not follow commands, had full strength and normal reflexes, and withdrew to noxious stimuli bilaterally.&lt;/p&gt;&lt;p&gt;Brain MRI with contrast revealed multifocal T2/fluid-attenuated inversion recovery (FLAIR) hyperintense lesions with some enhancement (Figure 1), meeting the McDonald imaging criteria for MS [&lt;span&gt;3&lt;/span&gt;]. While some demyelinating syndromes such as myelin oligodendrocyte glycoprotein (MOG) antibody disease (MOGAD) can present with psychosis, psychosis is not a typical MS presentation, so additional evaluation was pursued.&lt;/p&gt;&lt;p&gt;She subsequently developed acute respiratory distress and was transferred to the intensive care unit. Serum &lt;i&gt;Mycoplasma pneumoniae&lt;/i&gt; IgM and IgG were positive by immunofluorescent assay. Routine bloodwork including blood counts, chemistry panels, inflammatory markers, and nutritional labs were unrevealing. Cerebrospinal fluid studies were unremarkable except for an elevated IgG index (2.5). Oligoclonal bands were negative. Serum testing for anti-MOG and aquaporin-4 antibodies were negative for MOGAD and neuromyelitis optica spectrum disorder (NMOSD), respectively. Anti-NMDA antibodies were positive in the cerebrospinal fluid (1:80) and serum (1:160), consistent with a diagnosis of NMDARE.&lt;/p&gt;&lt;p&gt;She was treated with high dose of intravenous steroids, plasmapheresis, intravenous immunoglobulin, and rituximab. She received azithromycin to treat an acute &lt;i&gt;M. pneumoniae&lt;/i&gt; infection. She improved during the next year on maintenance intravenous immunoglobulin and rituximab. On follow-up imaging 1 year later, most lesions had improved or resolved except for one persistent lesion.&lt;/p&gt;&lt;p&gt;NMDARE is a common cause of pediatric encephalitis and can present with psychiatric symptoms, seizures, movement disorders, or altered consciousness [&lt;span&gt;4&lt;/span&gt;]. Definitive diagnosis includes at least one characteristic symptom and positive anti-NMDA autoantibodies [&lt;span&gt;4&lt;/span&gt;]. We assessed for autoimmune encephalitis in this patient due to the atypical clinical presentation for a demyelinating disease. Demyelinating features can occur in 3% of patients with NMDARE, with some meeting criteria for MOGAD or NMOSD. However, overlap between MS and NMDARE is not common [&lt;span&gt;2&lt;/span&gt;]. While coexistent MS an","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"59-61"},"PeriodicalIF":0.0,"publicationDate":"2025-02-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.70001","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688779","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Neonatal Hyperammonemia Due to Ureaplasma Sepsis","authors":"Catherine A. Kronfol,&nbsp;Aaron W. Hocher,&nbsp;E. Steve Roach","doi":"10.1002/cns3.20107","DOIUrl":"https://doi.org/10.1002/cns3.20107","url":null,"abstract":"&lt;p&gt;We describe a baby with severe hyperammonemia who was initially suspected to have an inborn error of metabolism but instead had &lt;i&gt;Ureaplasma&lt;/i&gt; sepsis. Hyperammonemia from &lt;i&gt;Ureaplasma&lt;/i&gt; infection is well-documented in immunocompromised adults, but the phenomenon has not been described in neonates, in whom hyperammonemia is usually assumed to represent a hereditary metabolic disease.&lt;/p&gt;&lt;p&gt;This 36-week gestation baby was transferred from another hospital because of metabolic acidosis, respiratory distress, and suspected seizures. His mother's pregnancy was complicated by maternal diabetes, premature rupture of membranes, and a 3-day history of vaginal bleeding. He was born limp, lethargic, and cyanotic, with Apgar scores of 3 and 7. On day 2 of life, he required intubation because of apnea and metabolic acidosis. Abnormal facial movements and posturing were initially suspected to represent seizures, so he was loaded with phenobarbital and levetiracetam. He also received empiric antibiotics and antiviral medications.&lt;/p&gt;&lt;p&gt;Antiseizure medications were halted after continuous electroencephalography showed no epileptiform discharges during his abnormal movements. Blood cultures and cerebrospinal fluid analysis were unremarkable, aside from an elevated cerebrospinal fluid protein. A respiratory culture for &lt;i&gt;Ureaplasma&lt;/i&gt; was negative, but next-generation DNA sequencing of serum confirmed evidence of &lt;i&gt;Ureaplasma&lt;/i&gt; urealyticum, for which he received azithromycin. His initial serum ammonia level was dramatically elevated (1284 μg/dL). His ammonia level increased to 1374 μg/dL despite the infusion of sodium benzoate and sodium phenylacetate, and he began continuous kidney replacement therapy. Urine organic acids, plasma amino acids, serum pyruvate, and carnitine were normal. Genetic testing was not completed due to his improving clinical condition, his resolving hyperammonemia, and the &lt;i&gt;Ureaplasma&lt;/i&gt; sepsis diagnosis.&lt;/p&gt;&lt;p&gt;At 1 week of age, multifocal cerebellar hemorrhages were documented on ultrasound and computed tomography (Figure 1). The hemorrhages were also evident with magnetic resonance imaging (MRI). No hemorrhages were identified in other areas of the brain, nor did the MRI reveal abnormalities suggestive of inborn errors of metabolism.&lt;/p&gt;&lt;p&gt;By 3 weeks of age, his condition had improved and his ammonia level had fallen to 63 μg/dL. At 13 months of age, he was starting to walk, playfully interacting, and saying several words. He has experienced no seizures or periods of lethargy.&lt;/p&gt;&lt;p&gt;This child was transferred due to suspected seizures, but continuous electroencephalography showed no epileptiform discharges, even during the movements. His serum ammonia level was dramatically elevated, leading to the initial suspicion of an inborn error of metabolism. However, neither metabolic testing nor MRI showed evidence of hereditary metabolic disorders, and the subsequent resolution of his hyperammonemia and his normal outcome furthe","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"57-58"},"PeriodicalIF":0.0,"publicationDate":"2025-02-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20107","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143689150","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Explosive-Onset Epilepsia Partialis Continua and Chorea","authors":"Peter F. Sarnacki,&nbsp;Gary Hsich,&nbsp;Aaron Abrams,&nbsp;Sumit Parikh","doi":"10.1002/cns3.20106","DOIUrl":"https://doi.org/10.1002/cns3.20106","url":null,"abstract":"<p>This 2-year-old neurodevelopmentally normal boy presented with abrupt onset of multiple leg clonic seizures with retained awareness (Video 1). Video-electroencephalography confirmed epileptic spikes and ictal onset arising from the vertex region, with an otherwise normal background. Initially controlled with levetiracetam, the seizures evolved to right leg epilepsia partialis continua (EPC). Two weeks later, he developed a hyperkinetic movement disorder reminiscent of chorea-ballismus. One week later, he exhibited developmental regression with encephalopathy, mutism, and insomnia. Anti-<i>N</i>-methyl-<span>d</span>-aspartate receptor (anti-NMDAR) antibody was positive in serum and cerebrospinal fluid, and oligoclonal bands were present. He received intravenous high-dose steroids and intravenous immune globulin and had incremental improvement. By 9 weeks from presentation he had made a remarkable recovery with almost complete symptom resolution.</p><p>The vast majority (95%) of children with anti-NMDAR encephalitis will develop a movement disorder, most commonly orofacial-lingual dyskinesias, but these may manifest broadly as chorea, athetosis, ballismus, dystonia, stereotypies, opisthotonus, oculogyric crisis, or bradykinesia [<span>1</span>]. Compared with adults, EPC and movement disorders occur more often as the initial presenting manifestation in children [<span>2</span>]. A diagnosis of anti-NMDAR encephalitis should be considered in young children with new-onset movement disorders and seizures (in particular, EPC), even in the absence of classically associated psychiatric or cognitive symptoms.</p><p><b>Peter F. Sarnacki:</b> conceptualization, investigation, writing–original draft, methodology, visualization, writing–review and editing, formal analysis, project administration, data curation. <b>Gary Hsich:</b> investigation, visualization, supervision, writing–review and editing. <b>Aaron Abrams:</b> conceptualization, writing–review and editing, visualization, supervision. <b>Sumit Parikh:</b> conceptualization, writing–review and editing, visualization, supervision.</p><p>The authors declare no conflicts of interest.</p>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"62-63"},"PeriodicalIF":0.0,"publicationDate":"2025-02-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20106","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143689147","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Robert S. Rust, Jr. (1948–2024)","authors":"Howard P. Goodkin,&nbsp;David E. Mandelbaum,&nbsp;John R. Mytinger,&nbsp;Phillip L. Pearl","doi":"10.1002/cns3.70002","DOIUrl":"https://doi.org/10.1002/cns3.70002","url":null,"abstract":"&lt;p&gt;Robert S. Rust, born August 11, 1948 in Van Nuys, California, was a true polymath—physician extraordinaire, investigator, teacher, scholar, historian, and musician. He exemplified and imparted the importance of Osler's &lt;i&gt;Aequanimitas&lt;/i&gt; as well as the value of a firm handshake. A bibliophile and prolific reviewer for the &lt;i&gt;Virginia Quarterly&lt;/i&gt;, his interests, expertise, and impact leave an extraordinary legacy (Figures 1-3).&lt;/p&gt;&lt;p&gt;Dr. Rust received a Bachelor of Arts in History and English &lt;i&gt;magna cum laude&lt;/i&gt; from Kent State University in 1970, followed by a Master of Arts in History from his beloved University of Virginia (UVA) and certificate in Greek language, history, and culture at the University of Thessaloniki/Institute for Balkan Studies. His education career began as a biology teacher at Albemarle High School (Albemarle County, VA), then as Associate Director of Studies at the International College Salzburg, Austria, where he lectured on the history of philosophy and science.&lt;/p&gt;&lt;p&gt;Rob then spent several years as a research associate in the UVA Department of Surgery, studying wound infections and the lymphatic aspects of immune function—an area that proved to be especially relevant in hindsight. During this time, he also developed a deep interest in the writings of Osler and Penfield. He would matriculate into the medical school with the plan of entering surgery until he encountered luminaries such as the renowned neuroanatomist Lennart Heimer, epileptologist Fritz Dreifuss, and neurologist/neuropathologist James Q. Miller, leading to a historic change of direction. He completed pediatric residency at Yale followed by pediatric neurology at Washington University, influenced particularly by Laura Ment, George Lister, Philip Dodge, Arthur Prensky, Ed Dodson, and Joseph Volpe. The early interest in immunology was especially fostered by Dodson and adult neurologist John Trotter, with Dr. Rust's first publication establishing reference values for cerebrospinal fluid immunoglobulins in children published in the &lt;i&gt;Annals of Neurology&lt;/i&gt; [&lt;span&gt;1&lt;/span&gt;]. Rob then worked in Dr. Volpe's laboratory, studying the regulation of the dolichol synthase pathway and protein glycosylation [&lt;span&gt;2&lt;/span&gt;]. He also worked in the famed metabolism laboratory of Oliver Lowry, meticulously quantifying enzymatic activity from cell culture analysis in cerebral cortex and superior cervical ganglia [&lt;span&gt;3&lt;/span&gt;].&lt;/p&gt;&lt;p&gt;Dr. Rust then moved to the University of Wisconsin, succeeding Ray Chun as Director of Child Neurology and Medical Director, Cerebral Palsy Clinic, and established a National Institutes of Health (NIH)–funded Developmental Brain Chemistry Laboratory. Dr. Chun became a lifelong mentor and friend; Rob kept a photograph of Dr. Chun, grinning widely in the presence of a child, on his desk throughout his career. Dr. Rust was especially touched to deliver the Raymond Chun Memorial Address in Madison in 2014.&lt;/p&gt;&lt;p&gt;In 1997, Rob joined Bosto","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"4-6"},"PeriodicalIF":0.0,"publicationDate":"2025-02-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.70002","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143689149","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Correction to Annals of the Child Neurology Society articles","authors":"","doi":"10.1002/cns3.20097","DOIUrl":"https://doi.org/10.1002/cns3.20097","url":null,"abstract":"<p>The below Conflicts of Interest related to membership on the ACNS editorial board were missing in the following articles.</p><p>van Haren KP, et al. Vitamin D status and latitude predict brain lesions in adrenoleukodystrophy. <i>Ann Child Neurol Soc</i>. 2023;1(2):155-161. doi:10.1002/cns3.4</p><p>In the above article, the following sentence should have been included in the Conflicts of Interest section: “J. B. L. is a member of the ACNS editorial board.”</p><p>Wanigasinghe J, et al. Demographic characteristics and clinical presentation of infants with infantile epileptic spasms syndrome and their response to therapy: data from Sri Lanka Infantile Spasms Registry. <i>Ann Child Neurol Soc</i>. 2023;1(2):137-143. doi:10.1002/cns3.20014</p><p>In the above article, the Conflicts of Interest section should have read, “Jithangi Wanigasinghe is a member of the ACNS editorial board. The remaining authors declare no conflicts of interest.”</p><p>Mohammadpour Touserkani F, Andriotis T, Zhang Y, Pavlakis S. Cerebral venous thrombosis and B12 deficiency. <i>Ann Child Neurol Soc</i>. 2023;1(2):152-154. doi:10.1002/cns3.9</p><p>In the above article, the Conflicts of Interest section should have read, “Steven Pavlakis is a member of the ACNS editorial board. The remaining authors declare no conflicts of interest.”</p><p>Bansal S, et al. A question prompt list for sudden unexpected death in epilepsy. <i>Ann Child Neurol Soc</i>. 2023;1(2):144-148. doi:10.1002/cns3.20027</p><p>In the above article, these two declarations should have been included in the Conflicts of Interest section: “Dr. Shellhaas is a member of the ACNS editorial board.” and “Dr. Lemmon is also a member of the ACNS editorial board.”</p><p>We apologize for this error.</p>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"64"},"PeriodicalIF":0.0,"publicationDate":"2025-02-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20097","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143689148","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Neuromyelitis optica spectrum disorder and autoimmune glial fibrillary acidic protein astrocytopathy overlap syndrome mimicking a pontine mass: The utility of brainstem biopsy","authors":"Vivien X. Xie,&nbsp;Gilbert Vezina,&nbsp;John S. Myseros,&nbsp;Lakshmi Ramachandran Nair,&nbsp;Elizabeth M. Wells,&nbsp;Benjamin I. Siegel","doi":"10.1002/cns3.20103","DOIUrl":"https://doi.org/10.1002/cns3.20103","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>Diffuse intrinsic pontine gliomas (DIPG) are high-grade tumors with a dismal prognosis and are classically diagnosed by radiologic features. DIPG is a critical differential consideration for a pediatric patient presenting with an infiltrative brainstem mass. However, inflammatory and infectious etiologies must also be considered, especially in individuals with atypical radiographic features. In a carefully selected clinical scenario, biopsy can be employed to quickly diagnose and direct treatment for patients with brainstem masses.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>This 10-year-old girl presented with acute onset of dysarthria, ataxia, left-sided weakness, hypertonicity, and dysmetria. Magnetic resonance imaging revealed an infiltrative pontine lesion with atypical features for that of DIPG or specific inflammatory disease. Due to rapid clinical deterioration, stereotactic brainstem biopsy was performed for diagnostic clarity and showed inflammation but no malignant cells. She was then treated for a presumed antibody-mediated autoimmune etiology with evaluation later revealing neuromyelitis optica spectrum disorder (NMOSD) and autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy overlap syndrome.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>We present a novel example of pediatric NMOSD and autoimmune GFAP astrocytopathy overlap syndrome originally presenting as an infiltrative pontine mass. Our report highlights the safety and utility of brainstem biopsy for brainstem masses atypical for DIPG.</p>\u0000 </section>\u0000 </div>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"46-51"},"PeriodicalIF":0.0,"publicationDate":"2025-02-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20103","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688850","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"In children with attention-deficit/hyperactivity disorder, less task-related up-modulation of motor cortex during response inhibition","authors":"Donald L. Gilbert,&nbsp;Deana Crocetti,&nbsp;Paul S. Horn,&nbsp;Steve W. Wu,&nbsp;David A. Huddleston,&nbsp;Jacqueline M. Ehrman,&nbsp;Karlee Y. Migneault,&nbsp;Stewart H. Mostofsky","doi":"10.1002/cns3.20101","DOIUrl":"https://doi.org/10.1002/cns3.20101","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objective</h3>\u0000 \u0000 <p>The aim of this study was to identify a quantitative, brain-based measure reflecting impaired response inhibition in children with attention-deficit/hyperactivity disorder (ADHD).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>In this cross-sectional study, we used transcranial magnetic stimulation (TMS) to evoke potentials in hand muscle during both a simple reaction time and a response inhibition task in 8-to-12-year-old children, 41 with ADHD (42% girls, 76% white, mean age 10.3 years) and 38 typically developing controls (53% girls, 74% white, mean age 9.8 years). We used mixed-model linear regressions of evoked potential amplitudes to compare motor cortex excitability at (1) task-onset (“START”: 550 ms prior to action); (2) preparing-to-go (“GO”: 150 ms prior to action); and (3) selecting-to-stop (“STOP”: 150 ms after stop cue). We hypothesized that task-related up-modulation of motor cortex excitability (motor evoked potential amplitudes) would depend both on task (STOP &gt; GO &gt; START) and on diagnosis (controls &gt; patients).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Motor cortex up-modulation was significantly greater for STOP trials than during GO or START. Children with ADHD had both worse response inhibition performance (longer stop-signal reaction times) and significantly less task effect on motor cortex up-modulation. The largest diagnostic difference in motor cortex activation occurred during STOP trials. Reduced up-modulation during stopping was also associated with higher parent-rated symptom severity.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>Our findings suggest that motor cortex up-modulation of excitability, assessed indirectly by TMS motor evoked potentials, reflects the cognitive load during response inhibition tasks and may be a quantitative, brain-based indicator of impaired response inhibition in children with ADHD.</p>\u0000 </section>\u0000 </div>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"26-36"},"PeriodicalIF":0.0,"publicationDate":"2025-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20101","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688880","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Early neurodevelopmental follow-up results from the NEOLEV2 cohort","authors":"Cynthia Sharpe,&nbsp;Gail E. Reiner,&nbsp;Peter W. Reed,&nbsp;Priscilla Joe,&nbsp;Francessa Wilson,&nbsp;Suzanne L. Davis,&nbsp;Lilly Lee,&nbsp;Sonya Wang,&nbsp;Jeff Gold,&nbsp;Richard H. Haas","doi":"10.1002/cns3.20096","DOIUrl":"https://doi.org/10.1002/cns3.20096","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Aim</h3>\u0000 \u0000 <p>The aim of this study was to evaluate predictors of neurodevelopmental outcome following hypoxic-ischemic encephalopathy (HIE) and neonatal seizures in the randomized controlled trial cohort from the NEOLEV2 study.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Method</h3>\u0000 \u0000 <p>Seizure burden, randomized levetiracetam versus phenobarbital antiseizure medication, and duration of breastfeeding were studied as predictors of neurodevelopmental outcome.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Outcome could be assessed in 115 patients with HIE and/or seizures; 89 patients with HIE (50% of 178 patients with HIE in the original cohort), and 60 patients with seizures (63% of 96 patients with seizures in the original cohort), and including 34 patients who had both HIE and seizures. A strong association between seizure burden and outcome was shown. However, the association was not statistically significant after correction for known predictors of outcome: magnetic resonance imaging severity of injury score, Sarnat score, and hypothermia treatment. This study was underpowered to assess for effect of randomized antiseizure medication or duration of breastfeeding on neurodevelopmental outcome; however, no large trends were seen.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Interpretation</h3>\u0000 \u0000 <p>This study adds to the debate as to whether neonatal seizures have an independent detrimental effect on neurodevelopmental outcome. Larger studies with longer neurodevelopmental follow-up are needed to investigate these questions.</p>\u0000 </section>\u0000 </div>","PeriodicalId":72232,"journal":{"name":"Annals of the Child Neurology Society","volume":"3 1","pages":"7-15"},"PeriodicalIF":0.0,"publicationDate":"2025-01-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cns3.20096","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143688878","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}