Background: Chronic daily headache (CDH) in youth is a functionally disabling and diagnostically heterogeneous condition that often requires multifaceted intervention. Real-world care is frequently fragmented-either limited to pharmacologic management or constrained by limited access to behavioral services. We evaluated the effectiveness of a generalist-deliverable bio-psycho-social (BPS) management model for children and adolescents with CDH, addressing both pharmacologic and contextual factors in the absence of subspecialty mental health services.
Methods: This retrospective, single-center observational study included 37 pediatric patients diagnosed with CDH according to ICHD-3 criteria. All participants received a structured BPS model delivered by general pediatricians. The model comprised (1) feature-guided pharmacologic treatment, (2) narrative-based psychosocial dialog, and (3) gradual school reintegration. Headache frequency, school attendance, and Pediatric Migraine Disability Assessment-assessed disability were evaluated at baseline, 6 months, and 24 months.
Results: By 6 months, only 13.5% of patients met CDH criteria; by 24 months, this decreased to 5.4%. Headache frequency, school attendance, and disability scores improved significantly (P < 0.001 for all). Improvements occurred across diagnostic subtypes and were not solely dependent on pharmacologic treatment, underscoring the contribution of nonpharmacologic components. Functional recovery-particularly school attendance-often lagged behind symptom resolution.
Conclusions: A generalist-deliverable BPS model was associated with sustained improvement in CDH outcomes, even in the absence of subspecialty resources. This pragmatic framework may support individualized, function-centered care for pediatric CDH in real-world settings.
{"title":"A Generalist-Deliverable Bio-Psycho-Social Model for Pediatric Chronic Daily Headache: A 24-Month Retrospective Study.","authors":"Soken Go, Natsumi Morishita, Mika Takeshita, Misako Murakami, Saori Minami, Wakako Matsumoto, Kanako Hayashi, Ryo Takahashi, Yusuke Watanabe, Naoko Saito, Koko Ohno, Akiko Kasuga, Shinichiro Morichi, Yu Ishida, Chiako Ishii, Naoko Kinjo, Gaku Yamanaka","doi":"10.1016/j.pediatrneurol.2025.12.025","DOIUrl":"https://doi.org/10.1016/j.pediatrneurol.2025.12.025","url":null,"abstract":"<p><strong>Background: </strong>Chronic daily headache (CDH) in youth is a functionally disabling and diagnostically heterogeneous condition that often requires multifaceted intervention. Real-world care is frequently fragmented-either limited to pharmacologic management or constrained by limited access to behavioral services. We evaluated the effectiveness of a generalist-deliverable bio-psycho-social (BPS) management model for children and adolescents with CDH, addressing both pharmacologic and contextual factors in the absence of subspecialty mental health services.</p><p><strong>Methods: </strong>This retrospective, single-center observational study included 37 pediatric patients diagnosed with CDH according to ICHD-3 criteria. All participants received a structured BPS model delivered by general pediatricians. The model comprised (1) feature-guided pharmacologic treatment, (2) narrative-based psychosocial dialog, and (3) gradual school reintegration. Headache frequency, school attendance, and Pediatric Migraine Disability Assessment-assessed disability were evaluated at baseline, 6 months, and 24 months.</p><p><strong>Results: </strong>By 6 months, only 13.5% of patients met CDH criteria; by 24 months, this decreased to 5.4%. Headache frequency, school attendance, and disability scores improved significantly (P < 0.001 for all). Improvements occurred across diagnostic subtypes and were not solely dependent on pharmacologic treatment, underscoring the contribution of nonpharmacologic components. Functional recovery-particularly school attendance-often lagged behind symptom resolution.</p><p><strong>Conclusions: </strong>A generalist-deliverable BPS model was associated with sustained improvement in CDH outcomes, even in the absence of subspecialty resources. This pragmatic framework may support individualized, function-centered care for pediatric CDH in real-world settings.</p>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"177 ","pages":"77-84"},"PeriodicalIF":2.1,"publicationDate":"2026-01-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146114006","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}
Transport protein particle complex 11 (TRAPPC11)-related disease, with autosomal recessive inheritance, exhibits a multisystemic involvement that goes widely beyond muscle weakness. Poor growth, psychomotor development delay, intellectual disability, microcephaly, ophthalmic involvement, and movement disorders are some of the typical features. Elevated serum creatine kinase levels are present in all previously reported TRAPPC11 c.1278+5G > A variant cases.
Methods
Clinical characterization of three siblings from a Roma family with TRAPPC11-related disease, all harboring a homozygous c.1287+5G > A variant.
Results
The older siblings presented typical features of the disease, including significant microcephaly, intellectual delay, and psychomotor regression precipitated by infections. Ataxia was consistently observed across all cases, albeit with varying severity. None of the cases had clinical signs compatible with muscular dystrophy. Notably, despite sharing the same mutation, the siblings exhibited remarkable phenotypic variability, with the youngest sibling displaying a milder phenotype.
Conclusions
This case series elucidates the intricate presentation of TRAPPopathies and underscores its phenotypic diversity, emphasizing the influence of the implicated deleterious variant. This study contributes to our understanding of TRAPPC11-related disease and highlights the importance of recognizing and characterizing phenotypic variability in this genetic disorder.
{"title":"Recurrent Encephalopathy as a Form of Presentation of Transport Protein Particle Complex 11–Related Disease: A Family Matter","authors":"Inês Noites MD , Catarina Borges MD , Sandra Catarina Ferraz MD , Cláudia Falcão-Reis MD , Cristina Garrido MD , Inês Carrilho MD","doi":"10.1016/j.pediatrneurol.2025.12.021","DOIUrl":"10.1016/j.pediatrneurol.2025.12.021","url":null,"abstract":"<div><h3>Background</h3><div>Transport protein particle complex 11 (TRAPPC11)-related disease, with autosomal recessive inheritance, exhibits a multisystemic involvement that goes widely beyond muscle weakness. Poor growth, psychomotor development delay, intellectual disability, microcephaly, ophthalmic involvement, and movement disorders are some of the typical features. Elevated serum creatine kinase levels are present in all previously reported <em>TRAPPC11</em> c.1278+5G > A variant cases.</div></div><div><h3>Methods</h3><div>Clinical characterization of three siblings from a Roma family with TRAPPC11-related disease, all harboring a homozygous c.1287+5G > A variant.</div></div><div><h3>Results</h3><div>The older siblings presented typical features of the disease, including significant microcephaly, intellectual delay, and psychomotor regression precipitated by infections. Ataxia was consistently observed across all cases, albeit with varying severity. None of the cases had clinical signs compatible with muscular dystrophy. Notably, despite sharing the same mutation, the siblings exhibited remarkable phenotypic variability, with the youngest sibling displaying a milder phenotype.</div></div><div><h3>Conclusions</h3><div>This case series elucidates the intricate presentation of TRAPPopathies and underscores its phenotypic diversity, emphasizing the influence of the implicated deleterious variant. This study contributes to our understanding of TRAPPC11-related disease and highlights the importance of recognizing and characterizing phenotypic variability in this genetic disorder.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 86-88"},"PeriodicalIF":2.1,"publicationDate":"2025-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146011617","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}
Pub Date : 2025-12-31DOI: 10.1016/j.pediatrneurol.2025.12.023
Ghazaleh Homaghostar, Ehsan Alimohammadi
{"title":"Prognostic Value of Brain Magnetic Resonance Imaging in Children After Out-of-Hospital Cardiac Arrest: Predictive Value of Normal Magnetic Resonance Imaging for a Favorable Two-Year Outcome.","authors":"Ghazaleh Homaghostar, Ehsan Alimohammadi","doi":"10.1016/j.pediatrneurol.2025.12.023","DOIUrl":"https://doi.org/10.1016/j.pediatrneurol.2025.12.023","url":null,"abstract":"","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"177 ","pages":"95-96"},"PeriodicalIF":2.1,"publicationDate":"2025-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"146157935","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}
Pub Date : 2025-12-31DOI: 10.1016/j.pediatrneurol.2025.12.019
Michael S. Salman MBBS, BSc, MSc, PhD , Chelsea A. Ruth MD, MSc, FRCPC , Randy Walld BSc, BComm (Hons) , Marina S. Yogendran MSc , Lisa M. Lix PhD
Background
To describe educational and social outcomes in optic nerve hypoplasia/septo-optic-pituitary dysplasia (ONH/SOD) in Manitoba, Canada.
Methods
A population-based case-control study used administrative health, education, and social data from the Manitoba Population Research Data Repository. A total of 124 ONH/SOD patients diagnosed during 1990-2019 were matched to 620 unrelated population-based controls on area of residence, birth year, and sex. Multivariable logistic regression tested for differences between cases and controls on selected educational and social outcomes. Odds ratios (ORs) with 95% confidence intervals (CIs) were estimated.
Results
Cases had higher odds than controls for requiring specialized educational funding (OR: 13.12, 95% CI: 7.06-24.40). Vulnerability in any five domains of Early Development Instrument, a measure of school readiness, showed higher odds in cases (OR: 2.58, 95% CI: 1.09-6.10). In addition, cases had higher odds of contact with Child and Family Services (OR: 1.81, 95% CI: 1.22-2.69), and being taken into care at any time after birth (OR: 2.10, 95% CI: 1.32-3.35).
Conclusions
Cases with ONH/SOD had a need for greater resources and more adverse educational and social outcomes. It is recommended for social and educational service providers to work together with health care providers to ensure appropriate supports are in place for cases with ONH/SOD.
{"title":"Educational and Social Outcomes in Optic Nerve Hypoplasia and Septo-Optic-Pituitary Dysplasia","authors":"Michael S. Salman MBBS, BSc, MSc, PhD , Chelsea A. Ruth MD, MSc, FRCPC , Randy Walld BSc, BComm (Hons) , Marina S. Yogendran MSc , Lisa M. Lix PhD","doi":"10.1016/j.pediatrneurol.2025.12.019","DOIUrl":"10.1016/j.pediatrneurol.2025.12.019","url":null,"abstract":"<div><h3>Background</h3><div>To describe educational and social outcomes in optic nerve hypoplasia/septo-optic-pituitary dysplasia (ONH/SOD) in Manitoba, Canada.</div></div><div><h3>Methods</h3><div>A population-based case-control study used administrative health, education, and social data from the Manitoba Population Research Data Repository. A total of 124 ONH/SOD patients diagnosed during 1990-2019 were matched to 620 unrelated population-based controls on area of residence, birth year, and sex. Multivariable logistic regression tested for differences between cases and controls on selected educational and social outcomes. Odds ratios (ORs) with 95% confidence intervals (CIs) were estimated.</div></div><div><h3>Results</h3><div>Cases had higher odds than controls for requiring specialized educational funding (OR: 13.12, 95% CI: 7.06-24.40). Vulnerability in any five domains of Early Development Instrument, a measure of school readiness, showed higher odds in cases (OR: 2.58, 95% CI: 1.09-6.10). In addition, cases had higher odds of contact with Child and Family Services (OR: 1.81, 95% CI: 1.22-2.69), and being taken into care at any time after birth (OR: 2.10, 95% CI: 1.32-3.35).</div></div><div><h3>Conclusions</h3><div>Cases with ONH/SOD had a need for greater resources and more adverse educational and social outcomes. It is recommended for social and educational service providers to work together with health care providers to ensure appropriate supports are in place for cases with ONH/SOD.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 77-85"},"PeriodicalIF":2.1,"publicationDate":"2025-12-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145978544","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}
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.
Methods
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.
Results
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, P = 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, P = 0.012).
Conclusions
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.
{"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":"2025-12-31","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}
Pub Date : 2025-12-29DOI: 10.1016/j.pediatrneurol.2025.12.011
Canan Üstün MD , Gevher Rabia Genç Perdecioğlu MD , Ömer Taylan Akkaya MD , Deniz Yüksel MD
Background
This study evaluated the efficacy and safety of noninvasive pulsed radiofrequency (NiPRF) therapy for childhood migraine and compared its outcomes with calcium channel blockers (CCBs).
Methods
A randomized controlled trial included 60 pediatric migraine patients (7-18 years). Patients were randomized into two groups: the CCB group (n = 30), receiving 5 mg flunarizine daily for 3 months, and the NiPRF group (n = 30), undergoing three weekly sessions of transcutaneous pulsed radiofrequency to the greater occipital nerve. Headache severity and frequency were recorded using a headache diary, and migraine-related disability was assessed with the Pediatric Migraine Disability Assessment score at baseline, one month, and 3 months. Two patients in the CCB group were excluded due to elevated transaminase levels and one in the NiPRF group for incomplete sessions, leaving 57 patients for analysis.
Results
Both treatments significantly reduced headache frequency and headache severity from baseline at 1 and 3 months. At one month, there was no significant difference between groups. However, at 3 months, the CCB group showed greater headache frequency reduction. Pediatric Migraine Disability Assessment scores improved in both groups, with a greater reduction in the CCB group at 3 months. Two CCB patients experienced transient liver enzyme elevation, while no significant side effects were observed in the NiPRF group.
Conclusions
NiPRF is a safe and effective treatment for childhood migraine, with comparable short-term efficacy to CCBs. Its noninvasive nature and minimal side effects make it a promising alternative. Further studies should assess long-term efficacy and optimize protocols.
{"title":"A Novel Neuromodulation Method for Childhood Migraine: Comparing Noninvasive Pulsed Radiofrequency Therapy With Calcium Channel Blockers, a Randomized Controlled Trial","authors":"Canan Üstün MD , Gevher Rabia Genç Perdecioğlu MD , Ömer Taylan Akkaya MD , Deniz Yüksel MD","doi":"10.1016/j.pediatrneurol.2025.12.011","DOIUrl":"10.1016/j.pediatrneurol.2025.12.011","url":null,"abstract":"<div><h3>Background</h3><div>This study evaluated the efficacy and safety of noninvasive pulsed radiofrequency (NiPRF) therapy for childhood migraine and compared its outcomes with calcium channel blockers (CCBs).</div></div><div><h3>Methods</h3><div>A randomized controlled trial included 60 pediatric migraine patients (7-18 years). Patients were randomized into two groups: the CCB group (n = 30), receiving 5 mg flunarizine daily for 3 months, and the NiPRF group (n = 30), undergoing three weekly sessions of transcutaneous pulsed radiofrequency to the greater occipital nerve. Headache severity and frequency were recorded using a headache diary, and migraine-related disability was assessed with the Pediatric Migraine Disability Assessment score at baseline, one month, and 3 months. Two patients in the CCB group were excluded due to elevated transaminase levels and one in the NiPRF group for incomplete sessions, leaving 57 patients for analysis.</div></div><div><h3>Results</h3><div>Both treatments significantly reduced headache frequency and headache severity from baseline at 1 and 3 months. At one month, there was no significant difference between groups. However, at 3 months, the CCB group showed greater headache frequency reduction. Pediatric Migraine Disability Assessment scores improved in both groups, with a greater reduction in the CCB group at 3 months. Two CCB patients experienced transient liver enzyme elevation, while no significant side effects were observed in the NiPRF group.</div></div><div><h3>Conclusions</h3><div>NiPRF is a safe and effective treatment for childhood migraine, with comparable short-term efficacy to CCBs. Its noninvasive nature and minimal side effects make it a promising alternative. Further studies should assess long-term efficacy and optimize protocols.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 41-47"},"PeriodicalIF":2.1,"publicationDate":"2025-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145927905","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}
Few studies have examined the participation of children with Duchenne muscular dystrophy (DMD), and further investigation is needed to understand the factors influencing it. This study aimed to compare the participation of children with DMD to typically developing (TD) male peers, explore the relationship between participation and environmental factors, and assess the role of the environment in participation levels.
Methods
In this cross-sectional study, 30 children with DMD and 30 TD male children aged 5–13 years were included. Participation levels were measured using the Assessment of Life Habits, and environmental conditions were assessed with the European Child Environment Questionnaire.
Results
Children with DMD exhibited significantly lower total participation scores compared to TD children (6.45 ± 1.88 vs. 8.67 ± 1.22; P < 0.001). Significant correlations were found between the total participation level and environmental factors (r = -0.526, P = 0.003). Regression analysis showed that environmental factors explained 34.1% of the variance in participation, with the physical environment identified as the sole significant predictor (beta = -0.517, P = 0.041).
Conclusions
These findings highlight the need for occupational therapists to systematically evaluate participation and environmental factors to plan effective interventions.
背景：对杜氏肌营养不良（DMD）患儿的参与情况研究较少，需要进一步调查了解其影响因素。本研究旨在比较DMD儿童与正常发育（TD）男性同伴的参与情况，探讨参与与环境因素的关系，并评估环境因素在参与水平中的作用。方法：采用横断面研究方法，选取5 ~ 13岁的30例DMD儿童和30例TD男性儿童。参与水平用生活习惯评估来衡量，环境条件用欧洲儿童环境问卷来评估。结果：DMD患儿的总参与得分明显低于TD患儿（6.45±1.88比8.67±1.22;P < 0.001）。总参与水平与环境因素呈显著相关（r = -0.526, P = 0.003）。回归分析显示，环境因素解释了34.1%的参与方差，其中物理环境是唯一的显著预测因子（beta = -0.517, P = 0.041）。结论：这些发现强调了职业治疗师需要系统地评估参与和环境因素，以计划有效的干预措施。
{"title":"Environmental Determinants of Participation in Children With Duchenne Muscular Dystrophy","authors":"Duygu Mine Alataş OT, MSc , Mustafa Cemali PT, PhD (Asst. Prof.) , Çiğdem Öksüz PT, PhD (Prof.) , Aynur Ayşe Karaduman PT, PhD (Prof.)","doi":"10.1016/j.pediatrneurol.2025.12.013","DOIUrl":"10.1016/j.pediatrneurol.2025.12.013","url":null,"abstract":"<div><h3>Background</h3><div>Few studies have examined the participation of children with Duchenne muscular dystrophy (DMD), and further investigation is needed to understand the factors influencing it. This study aimed to compare the participation of children with DMD to typically developing (TD) male peers, explore the relationship between participation and environmental factors, and assess the role of the environment in participation levels.</div></div><div><h3>Methods</h3><div>In this cross-sectional study, 30 children with DMD and 30 TD male children aged 5–13 years were included. Participation levels were measured using the Assessment of Life Habits, and environmental conditions were assessed with the European Child Environment Questionnaire.</div></div><div><h3>Results</h3><div>Children with DMD exhibited significantly lower total participation scores compared to TD children (6.45 ± 1.88 vs. 8.67 ± 1.22; <em>P</em> < 0.001). Significant correlations were found between the total participation level and environmental factors (r = -0.526, <em>P</em> = 0.003). Regression analysis showed that environmental factors explained 34.1% of the variance in participation, with the physical environment identified as the sole significant predictor (beta = -0.517, <em>P</em> = 0.041).</div></div><div><h3>Conclusions</h3><div>These findings highlight the need for occupational therapists to systematically evaluate participation and environmental factors to plan effective interventions.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 48-53"},"PeriodicalIF":2.1,"publicationDate":"2025-12-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145952942","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}
Pub 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
Background
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.
Methods
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.
Results
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.
Conclusions
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.
{"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":"2025-12-26","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}
Pub Date : 2025-12-19DOI: 10.1016/j.pediatrneurol.2025.12.009
James W. Varni PhD , Kathy Zebracki PhD , Miriam Hwang MD, PhD , M.J. Mulcahey PhD , Lawrence C. Vogel MD
Background
The study tests a conceptual model in which daily activities and mobility serve as mediators or intervening variables in the association between pain intensity and psychosocial health in young people with spinal cord injury (SCI). A moderated mediation conceptual model is also tested with biological sex as the moderator variable.
Methods
The Pain Intensity Item, Daily Activities Scale and Mobility Scale from the Pediatric Quality of Life Inventory SCI Module and the Pediatric Quality of Life Inventory Generic Core Scales Psychosocial Health Summary Score were completed by 125 young people with SCI ages 8-24 years with an average age of 17.84 years.
Results
The findings demonstrate that daily activities and mobility mediate the predictive effects of pain intensity on psychosocial health in young people with SCI. For the full mediator models consisting of age, sex, race/ethnicity demographic covariates, and pain intensity, the daily activities and mobility mediation models separately accounted for 39 percent and 28 percent, respectively, of the variance in psychosocial health, representing large effect sizes. Biological sex was not found to be a significant moderator of the mediation effects, and hence the mediator effects were not conditional on biological sex.
Conclusions
Daily activities and mobility explain in part the mechanism of pain predictive effects on psychosocial health in young people with SCI. Targeting mediators of pain intensity on psychosocial health from the perspective of children, adolescents, and young adults with SCI may inform clinical interventions and future clinical research to improve daily functioning and psychosocial health for these young people.
{"title":"Pain, Daily Activities, Mobility, and Psychosocial Health in Young People With Spinal Cord Injury: A Test of Biological Sex in a Moderated Mediation Analysis","authors":"James W. Varni PhD , Kathy Zebracki PhD , Miriam Hwang MD, PhD , M.J. Mulcahey PhD , Lawrence C. Vogel MD","doi":"10.1016/j.pediatrneurol.2025.12.009","DOIUrl":"10.1016/j.pediatrneurol.2025.12.009","url":null,"abstract":"<div><h3>Background</h3><div>The study tests a conceptual model in which daily activities and mobility serve as mediators or intervening variables in the association between pain intensity and psychosocial health in young people with spinal cord injury (SCI). A moderated mediation conceptual model is also tested with biological sex as the moderator variable.</div></div><div><h3>Methods</h3><div>The Pain Intensity Item, Daily Activities Scale and Mobility Scale from the Pediatric Quality of Life Inventory SCI Module and the Pediatric Quality of Life Inventory Generic Core Scales Psychosocial Health Summary Score were completed by 125 young people with SCI ages 8-24 years with an average age of 17.84 years.</div></div><div><h3>Results</h3><div>The findings demonstrate that daily activities and mobility mediate the predictive effects of pain intensity on psychosocial health in young people with SCI. For the full mediator models consisting of age, sex, race/ethnicity demographic covariates, and pain intensity, the daily activities and mobility mediation models separately accounted for 39 percent and 28 percent, respectively, of the variance in psychosocial health, representing large effect sizes. Biological sex was not found to be a significant moderator of the mediation effects, and hence the mediator effects were not conditional on biological sex.</div></div><div><h3>Conclusions</h3><div>Daily activities and mobility explain in part the mechanism of pain predictive effects on psychosocial health in young people with SCI. Targeting mediators of pain intensity on psychosocial health from the perspective of children, adolescents, and young adults with SCI may inform clinical interventions and future clinical research to improve daily functioning and psychosocial health for these young people.</div></div>","PeriodicalId":19956,"journal":{"name":"Pediatric neurology","volume":"176 ","pages":"Pages 22-28"},"PeriodicalIF":2.1,"publicationDate":"2025-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145918188","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}
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