Pediatric neurologyPub Date : 2026-03-01Epub Date: 2025-12-16DOI: 10.1016/j.pediatrneurol.2025.12.004
Adam Le MSc , Kelly-Ann Thibault DEC , Pouneh Amir Yazdani MD , Alexandra Chapleau HBSc , Romy J. van Voorst MSc , Enrico Bertini MD , Francesco Nicita MD, PhD , Daniela Pohl MD, PhD , Sunita Venkateswaran MD , Stephanie Keller MD , Deborah Renaud MD , Dolores Gonzalez Moron MD, PhD , Marcelo Kauffman MD, MSc, PhD , Danilo De Assis Pereira MD , Adeline Vanderver MD , Marjo S. van der Knaap MD, PhD , Maxime Morsa PhD , Geneviève Bernard MD, MSc, FRCPc
{"title":"The Impact of RNA Polymerase III–Related Leukodystrophy on Nonaffected Family Members: A Qualitative Study","authors":"Adam Le MSc , Kelly-Ann Thibault DEC , Pouneh Amir Yazdani MD , Alexandra Chapleau HBSc , Romy J. van Voorst MSc , Enrico Bertini MD , Francesco Nicita MD, PhD , Daniela Pohl MD, PhD , Sunita Venkateswaran MD , Stephanie Keller MD , Deborah Renaud MD , Dolores Gonzalez Moron MD, PhD , Marcelo Kauffman MD, MSc, PhD , Danilo De Assis Pereira MD , Adeline Vanderver MD , Marjo S. van der Knaap MD, PhD , Maxime Morsa PhD , Geneviève Bernard MD, MSc, FRCPc","doi":"10.1016/j.pediatrneurol.2025.12.004","DOIUrl":"10.1016/j.pediatrneurol.2025.12.004","url":null,"abstract":"<div><h3>Background</h3><div>RNA polymerase III–related hypomyelinating leukodystrophy (POLR3-HLD) is a rare, neurodegenerative, brain white matter disorder characterized by hypomyelination, hypodontia, and hypogonadotropic hypogonadism. Due to the complex and progressive nature of this disorder, parents and siblings of patients face many potential challenges and stressors. We, therefore, sought to explore parents' and siblings’ experiences to understand their specific needs and identify modifiable factors to limit familial burden and improve their quality of life.</div></div><div><h3>Methods</h3><div>We conducted semistructured interviews with parents and siblings of patients with POLR3-HLD. Interview questions focused on the financial, emotional, and psychosocial impacts on parents, as well as siblings’ relationship with their affected sibling and the psychosocial impacts they may have experienced. All interviews were recorded, transcribed, and analyzed using reflexive thematic analysis. Through the coding process, themes surrounding the impact on and experiences of parents and siblings were developed.</div></div><div><h3>Results</h3><div>Nineteen semistructured interviews with 24 parents and nine interviews with 9 siblings were completed between March and October 2023 and February and May 2024, respectively. Four themes from parent interviews included extensive caregiver burden, emotional and psychosocial challenges, the importance of parental self-health, and comfort in the leukodystrophy community. Three themes from sibling interviews included the spectrum of emotional impacts, limited knowledge about POLR3-HLD, and adapting to their sibling's needs.</div></div><div><h3>Conclusions</h3><div>This study provides a comprehensive understanding of the family experience, identifying the common challenges and specific needs of parents and siblings, highlighting areas of improvement in the global care offered to this vulnerable patient population.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 31-40"},"PeriodicalIF":2.1,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145927906","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-03-01Epub Date: 2025-12-26DOI: 10.1016/j.pediatrneurol.2025.12.010
Nuria Lamagrande-Casanova MD, Francisca Romero-Adújar MD, Azucena Lloris MD, Karina Sifontes MD, Elena González-Alguacil MD, Verónica Cantarín MD, PhD, Marta Bascuas MD, Cristina Benítez MD, María Ballará MD, Juan José García-Peñas MD, PhD, Anna Duat-Rodríguez MD, PhD, Víctor Soto-Insuga MD, PhD
{"title":"Obstructive Sleep Apnea Associated With Vagus Nerve Stimulation in Children With Drug Resistant Epilepsy: A Retrospective Case Series","authors":"Nuria Lamagrande-Casanova MD, Francisca Romero-Adújar MD, Azucena Lloris MD, Karina Sifontes MD, Elena González-Alguacil MD, Verónica Cantarín MD, PhD, Marta Bascuas MD, Cristina Benítez MD, María Ballará MD, Juan José García-Peñas MD, PhD, Anna Duat-Rodríguez MD, PhD, Víctor Soto-Insuga MD, PhD","doi":"10.1016/j.pediatrneurol.2025.12.010","DOIUrl":"10.1016/j.pediatrneurol.2025.12.010","url":null,"abstract":"<div><h3>Background</h3><div>Obstructive sleep apnea (OSA) is a common condition in children with drug resistant epilepsy. Its association with vagus nerve stimulation (VNS) is well-documented in adults, but pediatric data remain scarce. VNS-induced OSA may negatively impact seizure control and quality of life. This study aims to define the incidence, characteristic features, mechanistic underpinnings, and management strategies of VNS-associated OSA in children.</div></div><div><h3>Methods</h3><div>We conducted a retrospective case series of 14 children with drug resistant epilepsy who developed new-onset snoring following VNS implantation. All underwent polygraphy or polysomnography. We evaluated the temporal relationship between VNS stimulation and respiratory events, explored anatomical and functional mechanisms through drug-induced sleep endoscopy (DISE), and assessed therapeutic interventions including VNS parameter adjustments, Continuous Positive Airway Pressure, and surgical treatments.</div></div><div><h3>Results</h3><div>OSA was identified in 11 of 14 patients (79%) ranging from mild to severe. A highly characteristic respiratory pattern emerged, consisting of rhythmic obstructive events tightly synchronized with VNS active stimulation cycles. DISE identified vocal cord adduction during VNS activation in 3 patients, indicating an active obstructive mechanism. Nighttime VNS parameter adjustments effectively resolved OSA in 67% of patients without worsening seizure control. Continuous Positive Airway Pressure showed limited efficacy, likely due to active vocal cord adduction.</div></div><div><h3>Conclusions</h3><div>VNS-associated OSA is a frequent and under-recognized complication in children with epilepsy. Its distinctive polysomnographic signature and demonstrable laryngeal mechanism highlight the need for systematic sleep evaluation in VNS-treated children. DISE provides high diagnostic yield, but when unavailable, targeted VNS adjustment—particularly overnight modulation—offers an effective and practical management strategy. Early identification and treatment of OSA may contribute to improved seizure outcomes and overall quality of life.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 117-123"},"PeriodicalIF":2.1,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146023386","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Etiology of Infantile Epileptic Spasms Syndrome and Clinical Response With Vigabatrin as the First Treatment","authors":"Kullasate Sakpichaisakul MD , Pornnipa Sakjirapapong MD , Rachata Boonkrongsak MD , Sirorat Suwannachote MD","doi":"10.1016/j.pediatrneurol.2025.12.014","DOIUrl":"10.1016/j.pediatrneurol.2025.12.014","url":null,"abstract":"<div><h3>Background</h3><div>Etiology is an important predictor for treatment outcomes of infantile epileptic spasms syndrome (IESS). In Thailand, vigabatrin (VGB) is the first-line treatment for all patients due to the unavailability of adrenocorticotropic hormone. We aimed to determine the etiology of IESS using the 2017 International League Against Epilepsy classification and evaluate the clinical response of VGB as a first-line treatment.</div></div><div><h3>Methods</h3><div>A retrospective cohort study was conducted on IESS-diagnosed patients between January 2013 and December 2022. Etiology was categorized per the 2017 International League Against Epilepsy classification. Clinical outcomes were assessed at days 14-42 and one year after treatment.</div></div><div><h3>Results</h3><div>We included 191 IESS patients (57.6% males). Etiology was identified in 75.4% (structural 61.2%: 48.1% with nontuberous sclerosis complex [non-TSC] and 13.1% with TSC; genetic, 6.3%; infectious, 6.3%; and metabolic, 1.6%). Among the 163 patients who received VGB as first-line treatment, 50 (30.7%) achieved clinical cessation of epileptic spasms at days 14-42, and 44 patients (27.0%) had sustained freedom of epileptic spasms at one year. Patients with TSC etiology were more likely to achieve cessation of epileptic spasms at day 14-42 after treatment (adjusted OR 3.548, 95% confidence interval 1.193, 10.550, <em>P</em> = 0.023). Definite developmental delay at spasm diagnosis decreased the odds of sustained clinical freedom from epileptic spasms at one year (adjusted OR 0.26, 95% confidence interval 0.09, 0.74, <em>P</em> = 0.012).</div></div><div><h3>Conclusions</h3><div>The etiology of IESS was identified in 75%. VGB was most effective as first-line, short-term treatment in TSC patients, and one-year treatment in children with normal development at diagnosis of IESS.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 54-61"},"PeriodicalIF":2.1,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145965651","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Efficacy of Zoledronic Acid on Bone Mineral Density in Boys With Duchenne Muscular Dystrophy: A Prospective Observational Study","authors":"Santashree Chatterjee MD , Renu Suthar MD, DM , Tulika Singh MD , Mini Tageja MSc , Jaivinder Yadav MD , Harvinder Kaur PhD , Arushi G. Saini MD, DM , Savita Verma Attri PhD","doi":"10.1016/j.pediatrneurol.2025.11.007","DOIUrl":"10.1016/j.pediatrneurol.2025.11.007","url":null,"abstract":"<div><h3>Background</h3><div>To evaluate the efficacy and safety of intravenous zoledronic acid (ZA) in improving bone mineral density (BMD) and reducing bone turnover in boys with Duchenne muscular dystrophy (DMD) from North India.</div></div><div><h3>Methods</h3><div>In this prospective study, boys aged 5-18 years with genetically confirmed diagnosis of DMD, and either a history of low-impact long-bone or vertebral compression fractures (VCFs), or a lumbar spine/right femur neck dual-energy x-ray absorptiometry (DXA) BMD ≤ −2 z-score for age were enrolled. ZA was administered in three doses (total 0.125 mg/kg/year) at 3-month intervals. Outcomes assessed at 12 ± 2 months included changes in lumbar spine and femur neck BMD z-scores, VCFs number and grade, serum C-terminal telopeptide of type 1 collagen (CTX) levels, back pain, functional status, and quality of life.</div></div><div><h3>Results</h3><div>Fifty-five boys (mean age 10.7 ± 2.4 years) were enrolled. Seven boys (13%) had 8 long-bone fractures, and 33 (60%) had 86 VCFs on spine radiographs. Lumbar spine BMD z-score improved by 0.67 (95% confidence interval: 0.42-0.91; <em>P</em> = 0.001), and femur neck z-score by 0.49 (95% confidence interval: 0.06-0.93; <em>P</em> < 0.001). VCFs prevalence remained unchanged, though 26.7% developed new grade 1 VCFs. Mean serum CTX levels decreased from 0.488 to 0.179 ng/mL post ZA therapy (<em>P</em> < 0.001). ZA was well tolerated with no serious adverse events. Functional status and quality-of-life scores remained stable.</div></div><div><h3>Conclusions</h3><div>Intravenous ZA significantly improved BMD and reduced bone turnover in boys with DMD without major adverse effects.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 142-150"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145681727","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-02-01Epub Date: 2025-11-05DOI: 10.1016/j.pediatrneurol.2025.10.027
Anna Sosa BS , Kimberly A. O'Neill MD , Thibo Billiet PhD , Jingyun Chen PhD , Laura Couvreur MD , Stijn Denissen PhD , Matthew Lustberg MA , Shayna Pehel BS , Annemie Ribbens PhD , Lauren Krupp MD
{"title":"Incorporating Measures of Cognitive Processing Speed and Brain Volume in Clinical Management of Pediatric Onset Multiple Sclerosis","authors":"Anna Sosa BS , Kimberly A. O'Neill MD , Thibo Billiet PhD , Jingyun Chen PhD , Laura Couvreur MD , Stijn Denissen PhD , Matthew Lustberg MA , Shayna Pehel BS , Annemie Ribbens PhD , Lauren Krupp MD","doi":"10.1016/j.pediatrneurol.2025.10.027","DOIUrl":"10.1016/j.pediatrneurol.2025.10.027","url":null,"abstract":"<div><h3>Background</h3><div>While cognitive processing and brain volume assessments are commonly used in multiple sclerosis (MS) research, they are not routinely incorporated into clinical care. Advances in neuroimaging software now allow for brain volume measurement to be incorporated into standard practice and guide clinical decision-making.</div></div><div><h3>Methods</h3><div>In this single-center, outpatient, cohort study, pediatric-onset multiple sclerosis (POMS) patients underwent at least two volumetric brain magnetic resonance imagings (MRIs) and two Symbol Digit Modalities Tests (SDMT) at least 6 months apart. Associations were analyzed using Pearson's correlation coefficient and linear regressions.</div></div><div><h3>Results</h3><div>Forty patients with POMS were included. The first volumetric brain MRI occurred at a median of 2.7 years after symptom onset. At the first volumetric MRI, 12% of patients had white matter brain volumes within 1% of normative values (n = 2000 healthy controls). Whole brain volume percentile (r = 0.4, <em>P</em> = 0.01), white matter percentile (r = 0.4, <em>P</em> = 0.01), and white matter lesion volume (r = −0.4, <em>P</em> = 0.005) were associated with SDMT score. Annualized percent brain volume change of the hippocampus (measured a median of 1.6 years apart) significantly correlated with follow-up SDMT (r = 0.4, <em>P</em> = 0.02), and annualized percent brain volume change of the thalamus correlated with annual change in SDMT z-score (r = 0.4, <em>P</em> = 0.009).</div></div><div><h3>Conclusions</h3><div>Application of new software allows for volumetric assessment to be incorporated into clinical scans and provides clinicians with added data for patient management. This is critical for patients with POMS as the neurological examination often shows few to no abnormalities. Furthermore, a subset of POMS patients have smaller than expected brain volume which links to subsequent poorer cognitive performance.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 50-57"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145571169","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Investigating Cerebral Anomalies in Preterm Infants and Associated Risk Factors With Magnetic Resonance Imaging at Term-Equivalent Age","authors":"Nicolas Elbaz MD , Valérie Biran MD, PhD , Chloé Ghozland MSc , Laurie Devisscher MSc , Aline Gonzalez Carpinteiro MSc , Aurélie Bourmaud MD, PhD , Monique Elmaleh-Bergès MD , Lucie Hertz-Pannier MD, PhD , Yann Leprince PhD , Alice Frérot MD , Alice Héneau MD , Jessica Dubois PhD , Marianne Alison MD, PhD","doi":"10.1016/j.pediatrneurol.2025.11.012","DOIUrl":"10.1016/j.pediatrneurol.2025.11.012","url":null,"abstract":"<div><h3>Background</h3><div>Being born very or extreme preterm is a major source of cerebral anomalies and neurodevelopmental disorders, whose occurrence depends on many perinatal factors. A better understanding of these factors could be provided by cerebral magnetic resonance imaging (MRI) at term-equivalent age (TEA).</div></div><div><h3>Objective</h3><div>To investigate, through cerebral TEA-MRIs, the relationship between the main perinatal factors, the occurrence of cerebral anomalies, and cerebral volumetry.</div></div><div><h3>Methods</h3><div>We assembled a cohort of preterm babies born before 32 weeks of gestation who underwent a cerebral TEA-MRI. We assessed cerebral anomalies using a radiological scoring system — the Kidokoro scoring — and performed cerebral volumetry. We investigated the relationships between 9 clinical factors (birth characteristics, resuscitation treatments…), Kidokoro scores, and brain volumes.</div></div><div><h3>Results</h3><div>Among 110 preterms who underwent a cerebral MRI at TEA, only 6% suffered moderate-to-severe brain anomalies. We identified mechanical ventilation as a risk factor for cerebral anomalies (adjusted odds ratio = 4.6, 95% confidence interval [1.7-12.8]) and prolonged parenteral nutrition as a protective factor for white matter anomalies (adjusted odds ratio = 0.2, 95% confidence interval [0.1-0.8]). Mechanical ventilation (<em>P</em> = 0.01) and being born small for gestational age (<em>P</em> < 0.001) were risk factors for the reduction of cerebral volumes. An increase in brain lesion severity was associated with decreased cerebral volumes (<em>P</em> = 0.016).</div></div><div><h3>Conclusions</h3><div>Our study highlights the importance of treatment-related perinatal factors on the occurrence of cerebral anomalies in very and extreme preterms, and the interest in using both qualitative (Kidokoro scoring) and quantitative (volumetry) MRI-tools.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 156-164"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145715438","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-02-01Epub Date: 2025-11-28DOI: 10.1016/j.pediatrneurol.2025.11.018
Margaret Kasbarian, Harrison Cheng, Jada Phan, Troy Zeier MS, Jessica Bee PharmD, BCPPS, Jacklyn L. Wright PharmD, BCPPS, Jenna A. Chiang PsyD, ABPP-CN, Asri Yuliati MD
{"title":"Safety and Effectiveness of Cenobamate in Pediatric Patients With Treatment-Resistant Epilepsy","authors":"Margaret Kasbarian, Harrison Cheng, Jada Phan, Troy Zeier MS, Jessica Bee PharmD, BCPPS, Jacklyn L. Wright PharmD, BCPPS, Jenna A. Chiang PsyD, ABPP-CN, Asri Yuliati MD","doi":"10.1016/j.pediatrneurol.2025.11.018","DOIUrl":"10.1016/j.pediatrneurol.2025.11.018","url":null,"abstract":"<div><h3>Background</h3><div>Cenobamate is an antiseizure medication (ASM) for the treatment of focal-onset seizures in adults aged 18 years and older—while currently used off-label in children and adolescents, its pediatric potential is significant. Studies demonstrated a promising median seizure reduction of up to 65% in patients with treatment-resistant focal epilepsy.</div></div><div><h3>Methods</h3><div>This study aimed to investigate the effectiveness and safety of cenobamate as an adjunctive ASM in epilepsy patients under 18 years. The primary objective was to assess the effectiveness of cenobamate by examining seizure reduction. Secondary end points included adverse drug reactions (ADRs) and the median dose. Data were summarized using descriptive statistics, including mean, standard deviation, median, interquartile range, and percentages.</div></div><div><h3>Results</h3><div>Among 31 patients, 52% experienced a 75-100% reduction in seizure frequency, 16% had a 50-75% reduction, 13% had a 25-50% reduction, and 19% had no reduction or an increase in seizure frequency upon initiating cenobamate. These outcomes correspond to a median seizure reduction rate of 80%. The most common ADR was sedation (32.3%), followed by aggression and other mood changes. The median maintenance dose was 150 mg; patients weighing <50 kg received a median dose of 137.5 mg/day (5.2 mg/kg/day). Notably, patients were on a median of five ASMs before starting cenobamate, which decreased to two with cenobamate, suggesting a potential role in simplifying treatment regimens.</div></div><div><h3>Conclusions</h3><div>Our study showed that initiating cenobamate as adjunctive therapy was effective and safe for pediatric patients with treatment-resistant epilepsy, reducing seizure frequency and establishing improved seizure control.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 218-222"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145782441","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-02-01Epub Date: 2025-11-12DOI: 10.1016/j.pediatrneurol.2025.11.004
Robin Varughese MD , Josie Whelan , Taylor Koehler , Christine Palladino PNP , Ivan Pavkovic MD , Sanjeev Kothare MD
{"title":"Doxepin and Mirtazapine Use in Children and Adolescents With Symptoms of Insomnia — A Single-Center Retrospective Review","authors":"Robin Varughese MD , Josie Whelan , Taylor Koehler , Christine Palladino PNP , Ivan Pavkovic MD , Sanjeev Kothare MD","doi":"10.1016/j.pediatrneurol.2025.11.004","DOIUrl":"10.1016/j.pediatrneurol.2025.11.004","url":null,"abstract":"<div><h3>Background</h3><div>Sleep disorders are a common issue in children with pediatric insomnia, an under-recognized and undertreated problem affecting children. Beyond melatonin and behavioral interventions, there are few options known to clinicians that have been studied for use in pediatric insomnia. The objective of this study is to assess the efficacy, tolerability, and safety of doxepin and mirtazapine in pediatric insomnia.</div></div><div><h3>Methods</h3><div>This is a retrospective single-center chart review of children less than 18 years of age at the time of treatment initiation who have previously failed behavioral intervention and melatonin, who were then treated with doxepin and/or mirtazapine. Patients were started on doxepin maintenance treatments ranging from 2 to 10 mg and/or mirtazapine, which ranged from 1.875 to 15 mg. Improvement in sleep was assessed on follow-up visits and recorded using a 4-point Likert scale.</div></div><div><h3>Results</h3><div>There were 318 pediatric patients included for analysis. We identified 183 patients on doxepin, 24 patients on mirtazapine, and 19 patients who were on dual doxepin-mirtazapine. In the doxepin group, 167 patients (91.26%) experienced improvement in insomnia symptoms. In the mirtazapine group, 16 patients (66.67%) experienced improvement in insomnia symptoms. In the doxepin-mirtazapine group, 13 patients (68.42%) experienced improvement in insomnia symptoms. Thirty-two patients (10%) and three patients (7%) who took doxepin and mirtazapine, respectively, experienced adverse effects, most commonly aggression/irritability.</div></div><div><h3>Conclusions</h3><div>We found both doxepin and mirtazapine as effective and tolerable treatments options in improving subjective sleep onset and maintenance issues in children with insomnia.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 111-115"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145669192","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-02-01Epub Date: 2025-11-13DOI: 10.1016/j.pediatrneurol.2025.11.005
Corey D. Forrest MBBS, MPHTM , Tommaso Biagioni MBBS , Helen G. Liley MBChB , Melissa M. Lai PhD, MBBS , Paul B. Colditz PhD, MBBS , Robert S. Ware PhD , Roslyn N. Boyd PhD , James A. Roberts PhD
{"title":"Combined Use of Electroencephalography and Magnetic Resonance Imaging in the Prognostication of Neurodevelopmental Outcomes in Preterm Infants — A Systematic Review and Meta-Analysis","authors":"Corey D. Forrest MBBS, MPHTM , Tommaso Biagioni MBBS , Helen G. Liley MBChB , Melissa M. Lai PhD, MBBS , Paul B. Colditz PhD, MBBS , Robert S. Ware PhD , Roslyn N. Boyd PhD , James A. Roberts PhD","doi":"10.1016/j.pediatrneurol.2025.11.005","DOIUrl":"10.1016/j.pediatrneurol.2025.11.005","url":null,"abstract":"<div><h3>Background</h3><div>Neurological injury remains a major cause of neurodevelopmental morbidity in preterm-born infants. Accurate prognostication is essential for support of families and to target interventions. Electroencephalography (EEG) and magnetic resonance imaging (MRI) can be used to precisely assess brain function and structure in preterm infants. This systematic review assessed the prognostic capacity of EEG and MRI alone, and in combination, for the prediction of neurodevelopmental outcomes in preterm-born infants.</div></div><div><h3>Methods</h3><div>PubMed, Embase, Web of Science, and Cochrane databases were searched from inception until April 22, 2025. Studies were included if they examined the use of both MRI and EEG in preterm-born infants to predict neurodevelopmental outcomes.</div></div><div><h3>Results</h3><div>From 1669 records screened, 12 studies reporting 1630 infants were included in the systematic review. Of these 12, seven were sufficiently homogenous for quantitative meta-analysis, yielding pooled sensitivity and specificity values across 952 infants: EEG prognosticated abnormal neurodevelopmental outcomes with sensitivity 64% (95% confidence interval [CI] 48-80%) and specificity 70% (95% CI 59-81%); MRI prognosticated abnormal neurodevelopmental outcomes with sensitivity 64% (95% CI 48-80%) and specificity 89% (95% CI 81-97%). Three of these seven studies, reporting 372 infants, were sufficiently comparable to meta-analyze the combination of EEG and MRI, which predicted neurodevelopmental outcome with sensitivity 70% (95% CI 49-90%) and specificity 96% (95% CI 93-100%).</div></div><div><h3>Conclusions</h3><div>The combined use of EEG and MRI can predict abnormal neurodevelopment in preterm-born infants with high sensitivity and specificity and was superior to either test in isolation; however, data are limited as multimodal neurological-assessment is still an emerging practice in neonates.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 116-129"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145669146","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pediatric neurologyPub Date : 2026-02-01Epub Date: 2025-11-14DOI: 10.1016/j.pediatrneurol.2025.11.008
Darina Dinov DO, Denise Maddox BSN, Benjamin M. Greenberg MD, MHS
{"title":"Inebilizumab: Pediatric Case Series in Anti-Aquaporin 4 Antibody Positive Neuromyelitis Optica Spectrum Disorder","authors":"Darina Dinov DO, Denise Maddox BSN, Benjamin M. Greenberg MD, MHS","doi":"10.1016/j.pediatrneurol.2025.11.008","DOIUrl":"10.1016/j.pediatrneurol.2025.11.008","url":null,"abstract":"<div><h3>Background</h3><div>Neuromyelitis Optica Spectrum Disorder (NMOSD) is an autoimmune condition which can be diagnosed either based on a constellation of neurological symptoms and/or a positive anti-aquaporin 4 antibody. About 3-5% of all cases occur in pediatric patients, and currently there are no FDA-approved therapies for patients under the age of 18 years for NMOSD. There are currently 4 FDA-approved medications for adults with seropositive NMOSD-eculizumab, inebilizumab, satralizumab, and ravulizumab.</div></div><div><h3>Methods/Results</h3><div>Herein, we present a case series of four patients who have seropositive NMOSD and are on inebilizumab. All four patients have had two courses of this medication and have tolerated it well. There was not a significant burden of side effects, and none of the patients have presented with relapses.</div></div><div><h3>Conclusions</h3><div>This study adds to the literature because it shows that in our cohort inebilizumab was a safe medication for pediatric patients with NMOSD and prevented further relapses.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"175 ","pages":"Pages 108-110"},"PeriodicalIF":2.1,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145649046","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}