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Construction and verification of multimodal model for prognosis in elderly patients with aneurysmal subarachnoid hemorrhage.

Zhang W, Li G, Gao Y … +3 more , Liu Z, Zhang B, Wang J

BMC Neurol · 2026 May · PMID 42104264 · Full text

OBJECTIVE: This study aimed to construct and validate an individualized prediction model for poor prognosis in elderly patients with Aneurysmal subarachnoid hemorrhage (aSAH) based on multimodal indicators such as clinic... OBJECTIVE: This study aimed to construct and validate an individualized prediction model for poor prognosis in elderly patients with Aneurysmal subarachnoid hemorrhage (aSAH) based on multimodal indicators such as clinical, imaging, and laboratory data. METHODS: We retrospectively enrolled 241 consecutive patients with aSAH from January 2017 to December 2020 as the training set. An independent temporal validation set included 104 consecutive patients from January to September 2024, with assignment strictly by chronological order. In the training set, univariate analysis identified candidate predictors of poor prognosis, which were refined via LASSO regression. Significant variables were then entered into multivariate logistic regression to define independent predictors. Using these predictors, we constructed random forest (RF), support vector machine (SVM), and k-nearest neighbor models (KNN). Model performance was assessed using the area under the receiver operating characteristic curve (AUC), calibration curves, and decision curve analysis (DCA). Model interpretability and variable contributions were analyzed with SHapley Additive exPlanations (SHAP). RESULTS: There were no statistically significant differences in the baseline clinical data between the training set and the validation set (all P > 0.05). The results of multivariate Logistic regression analysis showed that World Federation of Neurosurgical Societies (WFNS) score, modified Fisher grade, intracerebral hematoma volume, maximum thickness of subarachnoid blood clots, C-reactive protein, and duration of symptomatic cerebral vasospasm were identified as independent risk factors for poor prognosis (all P < 0.05). The performance evaluation of the machine-learning models showed that the SVM model had the best discrimination, with AUCs of 0.838 (95% CI: 0.764-0.912) and 0.791 (95% CI: 0.688-0.895) in the training set and the validation set, respectively. The calibration curve showed a high consistency between the predicted probability and the actual risk. DCA indicated that the model had clinical net benefits within a wide range of thresholds. SHAP analysis confirmed that C-reactive protein, maximum thickness of subarachnoid clot, and intracerebral hematoma volume were the most important contributing factors to the increased risk of poor prognosis. CONCLUSION: This study successfully constructed and validated a prediction model for poor prognosis in elderly patients with aSAH based on multimodal indicators. The model has robust performance, clinically accessible indicators, and good interpretability, providing a valuable quantitative tool for dynamic risk stratification and implementation of stratified management.

Exploring the phenotypic and genotypic spectrum of spinal muscular atrophy in Bangladeshi children.

Parvin J, Khan HA, Saha NC … +4 more , Hoque SA, Hossain MM, Saha D, Shams N

BMC Neurol · 2026 May · PMID 42104248 · Full text

Spinal muscular atrophy (SMA) is a monogenic neuromuscular disorder caused by SMN1 gene deletion and classified by clinical severity. The objective of this study was to evaluate the genotypic and phenotypic spectrum in S... Spinal muscular atrophy (SMA) is a monogenic neuromuscular disorder caused by SMN1 gene deletion and classified by clinical severity. The objective of this study was to evaluate the genotypic and phenotypic spectrum in SMA in Bangladeshi children.This cross-sectional prospective study was conducted in Pediatric Neurology Department, National Institute of Neurosciences and Hospital of Bangladesh from January 2019 and December 2022. A total of sixty four cases with clinically suspected SMA were enrolled. Genetic confirmation was done using MLPA.Genetic confirmation was achieved in 48 (75%) patients. Homozygous deletion of SMN1 exons 7 and 8 was detected in 44 (68.75%) cases, while isolated exon 7 deletion was found in 4 (6.25%) cases. No deletion was identified in 16 (25%) cases. Among genetically confirmed patients, SMA type I was most common (54%), followed by type II (40%) and type III (6%). The mean age of onset was significantly earlier in SMA type I compared with types II and III (p < 0.05). A significant inverse correlation was observed between SMN2 copy number and disease severity (Spearman's rho = 0.825, p < 0.001). Most type I patients had two SMN2 copies, whereas type II and III patients predominantly had three or more copies. Respiratory complications and mortality were predominantly in type I patients. Two patients received risdiplam and two underwent gene replacement therapy (onasemnogene abeparvovec).In conclusion, A significant genotype-phenotype correlation exists between SMN2 copy number and clinical severity among Bangladeshi children with SMA. Limited access to disease-modifying therapy highlights the need for early diagnosis and improved treatment accessibility in resource-limited settings.

Clinical value of structural cranial magnetic resonance imaging analysis in assessing brain aging in ischemic stroke patients.

Wang T, You X, Yin J … +4 more , Pan H, Liu Z, Dai Q, Yu J

BMC Neurol · 2026 May · PMID 42104247 · Full text

OBJECTIVE: Using structural cranial magnetic resonance imaging (MRI), we constructed a structural Brain Aging Index (sBAI) and evaluated the brain aging status, expressed as Brain Predicted Age Difference (Brain-PAD), in... OBJECTIVE: Using structural cranial magnetic resonance imaging (MRI), we constructed a structural Brain Aging Index (sBAI) and evaluated the brain aging status, expressed as Brain Predicted Age Difference (Brain-PAD), in acute ischemic stroke patients. We aim to investigate its associations with baseline clinical and imaging characteristics, small vessel disease (SVD) burden, and 12-month functional and cognitive outcomes, and explored its potential contribution to prognostic models. METHODS: A retrospective cohort of 150 patients with acute ischemic stroke admitted between November 2021 and November 2023 was analyzed. Diffusion-weighted imaging (DWI) infarct volume, white matter hyperintensity (WMH) volume, lacunes, cerebral microbleeds (CMBs), enlarged perivascular spaces (EPVS), hippocampal volume, and cortical thickness were collected. Clinical and imaging differences were compared between the "age-matched group" (Brain-PAD < + 5 years) and the "accelerated brain aging group" (Brain-PAD ≥ + 5 years). RESULTS: Patients in the accelerated brain aging group had significantly higher admission National Institutes of Health Stroke Scale (NIHSS) scores, larger DWI infarct volumes, greater WMH burden, and higher total SVD scores. During follow-up, the accelerated brain aging group had a lower rate of favorable functional outcome at discharge and a higher incidence of poor 12-month functional outcome, together with significantly lower MoCA scores. Admission NIHSS and WMH volume (aOR = 1.10) were independent predictors of 12-month functional outcome. sBAI was significantly associated with 12-month MoCA scores. CONCLUSION: Brain aging status derived from structural MRI is closely associated with acute lesion burden, cumulative small vessel disease, and poststroke cognitive and functional outcomes.

The effectiveness of virtual reality/motion-sensing games incorporating swing-based elements on upper limb function after stroke: a systematic review and meta-analysis.

Cai W, Zheng Y, Wu J … +4 more , Hu G, Takeshita D, Loh PY, He Y

BMC Neurol · 2026 May · PMID 42104238 · Full text

BACKGROUND: Upper limb impairment is a common consequence of stroke and can markedly limit independence in daily life. Virtual reality (VR) and motion-sensing games incorporating swing-based elements have increasingly be... BACKGROUND: Upper limb impairment is a common consequence of stroke and can markedly limit independence in daily life. Virtual reality (VR) and motion-sensing games incorporating swing-based elements have increasingly been used in rehabilitation, but their effectiveness for post-stroke upper limb recovery remains unclear. This article aims to systematically evaluate the effectiveness of VR/motion-sensing games incorporating swing-based elements on upper limb function after stroke. METHOD: This systematic review and meta-analysis was conducted in accordance with PRISMA guidelines and registered in PROSPERO (CRD420251141175). PubMed, Web of Science, Scopus, and the Cochrane Library were searched from January 1999 to September 2025 for randomized controlled trials. Standardized mean differences (SMDs) with 95% confidence intervals (CIs) were pooled using a random-effects model. Heterogeneity was assessed using the I² statistic and Cochran's Q test. Sensitivity and subgroup analyses were performed, and publication bias was explored using funnel plots and Egger's test. RESULTS: Ten randomized controlled trials involving 426 participants were included. Compared with conventional therapy, virtual reality/motion-based games incorporating swing-like elements demonstrated significant improvement in upper limb functional recovery. The overall meta-analysis showed a merge standardized mean difference of 0.64 (95% CI: 0.44, 0.84) in favour of the intervention group. Substantial between-study heterogeneity was observed (I² = 82.2%, p < 0.001). Exploratory subgroup analyses suggested relatively larger effects in older participants and in those with longer stroke duration, although these findings should be interpreted cautiously. Sensitivity analysis showed that the pooled effect was stable, and Egger's test did not indicate significant publication bias (p = 0.454). CONCLUSION: VR/motion-sensing games incorporating swing-based elements may improve upper limb function after stroke and could serve as a useful adjunct to conventional rehabilitation. Further rigorously designed trials with standardized protocols are required to strengthen the evidence base and determine optimal intervention strategies.

Varicella-zoster virus infection triggering GD3 antibody-positive variant Guillain-Barré syndrome: a case report and literature review.

Wu Y, Gu L, Liu J … +2 more , Yan W, Wei J

BMC Neurol · 2026 May · PMID 42098679 · Full text

BACKGROUND: Bilateral facial palsy variant Guillain-Barré syndrome (GBS) is a distinct clinical subtype. As an acute immune-mediated polyradiculoneuropathy often triggered by infections, varicella-zoster virus (VZV) is a... BACKGROUND: Bilateral facial palsy variant Guillain-Barré syndrome (GBS) is a distinct clinical subtype. As an acute immune-mediated polyradiculoneuropathy often triggered by infections, varicella-zoster virus (VZV) is a rare yet significant precipitating factor for this variant. Anti-ganglioside antibodies, particularly anti-GD3, are involved in the pathogenesis of GBS, but their significance in VZV-associated bilateral facial palsy subtype remains incompletely elucidated. CASE PRESENTATION: We report a 46-year-old female who developed acute bilateral facial palsy and limb sensory deficits following VZV infection. Serum anti-ganglioside testing confirmed GD3 IgG positivity, and cerebrospinal fluid analysis revealed albuminocytological dissociation. Nerve conduction studies supported a demyelinating polyneuropathy, and the patient showed significant improvement after intravenous immunoglobulin therapy. CONCLUSIONS: This case suggests that VZV may act as a potential trigger for GD3 antibody-positive variant GBS. Early immunotherapy may improve outcomes, emphasizing the need for systematic antibody testing in atypical presentations of this variant GBS. Further studies are required to clarify the pathophysiological role of GD3 antibodies in VZV-associated variant GBS.

Long-term cerebral atrophy and cognitive function in patients recovering from progressive multifocal leukoencephalopathy: two case reports.

Takahashi S, Ono D, Yokota T … +1 more , Sanjo N

BMC Neurol · 2026 May · PMID 42098678 · Full text

BACKGROUND: Long-term brain atrophy and cognitive function have rarely been reported in patients without pre-existing brain disorders who survived progressive multifocal leukoencephalopathy (PML). CASE PRESENTATION: Pati... BACKGROUND: Long-term brain atrophy and cognitive function have rarely been reported in patients without pre-existing brain disorders who survived progressive multifocal leukoencephalopathy (PML). CASE PRESENTATION: Patient 1 was a 38-year-old woman with late-onset combined immunodeficiency, who presented with progressive cognitive decline and right hemiparesis. Brain MRI revealed a hyperintense lesion in the left frontal lobe. Patient 2 was a 53-year-old man in remission from follicular lymphoma, who experienced cognitive deterioration and displayed hyperintense signals in the left frontal and bilateral posterior lobes on MRI. Brain biopsy confirmed PML in both patients. After recovering from acute neurological decline, the cognitive and physical function of the patients was followed and we retrospectively analyzed sequential alterations in the MRI over eight years. Hyperintense lesion areas, cerebral hemisphere size, and intracranial volume were measured to calculate the monthly change ratios of the brain (MCRB), along with assessment of the third ventricle width. Both patients experienced no relapse. Wechsler Memory Scale-Revised scores improved in Patient 1 but slightly declined in Patient 2 despite stable Mini-Mental State Examination scores in both patients. MRI results were classified as lesion expansion, lesion reduction, and chronic atrophy. During the chronic atrophic stage, MCRB decreased by 0.06% and 0.09% in Patients 1 and 2, respectively. The third ventricle widened by 3 and 25 μm/month in Patients 1 and 2, respectively. CONCLUSION: Progressive cerebral atrophy occurs during PML's chronic stage, even without relapse. In these two cases, the patient with extensive bilateral lesions in the acute phase showed greater third ventricle enlargement and cognitive decline.

Prognostic significance of early ACTH levels on neurological recovery in patients with aneurysmal subarachnoid hemorrhage: a prospective cohort study.

Aljboor GSR, Tulemat A, Tan GHE … +4 more , De Clercq NC, Rădoi MP, Corneliu T, Papacocea TM

BMC Neurol · 2026 May · PMID 42098676 · Full text

BACKGROUND/OBJECTIVES: Aneurysmal subarachnoid hemorrhage (aSAH) triggers a marked systemic stress response and may be accompanied by hypothalamic-pituitary-adrenal (HPA) axis dysregulation. Adrenocorticotropic hormone (... BACKGROUND/OBJECTIVES: Aneurysmal subarachnoid hemorrhage (aSAH) triggers a marked systemic stress response and may be accompanied by hypothalamic-pituitary-adrenal (HPA) axis dysregulation. Adrenocorticotropic hormone (ACTH) may reflect the acute neuroendocrine response to injury and could be associated with early functional status. This study aimed to describe the frequency of acute-phase ACTH abnormalities and evaluate the association between early ACTH levels and functional outcomes at hospital discharge in patients with aSAH. METHODS: In this prospective cohort study, 20 consecutive aSAH patients admitted within 48 hours of symptom onset were enrolled. Plasma ACTH levels were obtained at random time points within the first 48 hours of admission (predefined cutoff ≤7 days; no patient exceeded 48 hours), prior to definitive aneurysm treatment. Blood samples were obtained within 24 hours of admission and prior to surgical intervention. Sampling was targeted between 08:00 and 10:00 to minimize circadian variation; however, minor deviations were unavoidable in the acute neurocritical care setting. Functional outcomes were assessed at discharge using the Glasgow Outcome Score (GOS) and Modified Rankin Scale (MRS). Associations were evaluated using Spearman correlation, non-parametric group comparisons, and exploratory multivariable regression analyses adjusting for key severity variables. RESULTS: Using institutional laboratory reference ranges, 12/20 patients (60%) had ACTH values below the reference range, 7/20 (35%) were within range, and 1/20 (5%) had elevated ACTH. ACTH levels were significantly associated with discharge outcomes (mRS: ρ = +0.67, p = 0.0014; GOS: ρ = -0.59, p = 0.0063). ACTH distributions differed between favourable (mRS ≤ 2) and unfavourable (mRS > 2) outcome groups (p = 0.014). In exploratory multivariable analysis, ACTH showed a consistent association with outcome but did not remain statistically significant after adjustment. CONCLUSIONS: Early ACTH levels obtained during the acute hospitalization were associated with short-term functional outcomes at discharge after aSAH. Given the random sampling design, the stress-sensitive nature of ACTH, and the absence of paired cortisol measurements, these findings should be interpreted as reflecting ACTH dysregulation rather than definitive endocrine deficiency. Larger studies with standardized sampling and longer-term follow-up are warranted.

Modified Berlin Score for predicting sleep apnea in patients with acute ischemic stroke.

Wong AC, Wong YS, Sung SF … +5 more , Wu CS, Su YH, Lin MH, Su CY, Ong CT

BMC Neurol · 2026 May · PMID 42092836 · Full text

BACKGROUND: Sleep-disorder breathing is common among ischemic stroke patients. It associated with increasing risk of stroke and poor functional outcome after stroke. Polysomnography (PSG) is the diagnostic standard but t... BACKGROUND: Sleep-disorder breathing is common among ischemic stroke patients. It associated with increasing risk of stroke and poor functional outcome after stroke. Polysomnography (PSG) is the diagnostic standard but the application for acute stroke patients is limited. The Berlin Questionnaire (BQ) is a tool used for screen obstructive sleep apnea in general population, however, the diagnostic accuracy in acute ischemic stroke patients is inconsistent. OBJECTS: The study aims to explore the accuracy of Berlin Questionnaire (BQ) for detecting the sleep apnea for acute ischemic stroke patients, using home-based sleep apnea testing as a reference. We compare the accuracy between BQ and a modified BQ. METHODS: We conducted a prospective observational study between March 2023 and February 2025 at a teaching hospital in central Taiwan. Patients aged 20-85 years with MRI-confirmed acute ischemic stroke admitted within 48 h of onset were included. Each participant completed the Berlin Questionnaire and underwent home-based sleep apnea testing (Alice NightOne, Philips Respironics). Sleep apnea severity was defined by the respiratory event index (REI). The accuracy of BQ and mBQ in predicting moderate-to-severe sleep apnea (REI ≥ 15) was assessed using sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV). RESULTS: Forty-six patients completed both assessments. Twenty-one patients (45.7%) had no or mild sleep apnea (REI < 15), and 25 (54.3%) had moderate-to-severe sleep apnea (REI ≥ 15). Alcohol use and higher body mass index (BMI) were significantly associated with moderate-to-severe apnea (p = 0.01 for both). The BQ demonstrated a sensitivity of 45.8%, specificity of 72.7%, PPV of 64.7%, and NPV of 55.2% for predicting moderate-to-severe sleep apnea. The mBQ improved performance to a sensitivity of 54.1%, specificity of 72.7%, PPV of 68.4%, and NPV of 59.3%. Patients with moderate-to-severe apnea exhibited significantly lower minimum oxygen saturation compared with those with no or mild apnea (p = 0.03). CONCLUSIONS: The accuracy of the Berlin Questionnaire for detecting sleep apnea in acute ischemic stroke is moderate but improves with modification of BMI weighting. Moderate-to-severe apnea is associated with lower oxygen saturation, highlighting the importance of early screening and targeted management in stroke units. Larger, multicenter studies are warranted to validate these findings and explore the prognostic implications of nocturnal hypoxemia in stroke recovery.

Dynamic recovery of piriform cortex function and its impact on cognitive processing speed following mild COVID-19.

Jeong H, Shim Y, Joo Y … +6 more , Kim Y, Suh C, Jin Y, Song H, Lyoo IK, Yoon S

BMC Neurol · 2026 May · PMID 42092802 · Full text

BACKGROUND: Although the neurological sequelae of severe COVID-19 are increasingly recognized, the long-term neural impact of uncomplicated mild SARS-CoV-2 infection remains insufficiently characterized. This study inves... BACKGROUND: Although the neurological sequelae of severe COVID-19 are increasingly recognized, the long-term neural impact of uncomplicated mild SARS-CoV-2 infection remains insufficiently characterized. This study investigated post-infection alterations in piriform cortex activity, a region central to olfactory processing and integrative cognitive-emotional networks, and their associations with cognitive outcomes in adults. METHODS: Fifty adults with pre- and post-SARS-CoV-2 magnetic resonance imaging (MRI) and clinical/neuropsychological data were analyzed. A demographically matched control group of 50 individuals without prior COVID-19 or vaccination was included. Resting-state functional MRI assessed amplitude of low-frequency fluctuations (ALFF) within the piriform cortex as an index of regional neural activity. RESULTS: Baseline piriform ALFF did not differ between the two groups (p = 0.421). Following infection, piriform ALFF exhibited a significant quadratic trajectory (p = 0.003), characterized by early elevation relative to baseline, a decline to a nadir at approximately three months, and subsequent recovery toward baseline levels by six months. Greater acute COVID-19 symptom burden correlated with higher piriform ALFF (β = 0.34, p = 0.017). Elevated ALFF was associated with reduced processing speed (β = -0.52, p = 0.001). Mediation analysis demonstrated that piriform ALFF significantly mediated the relationship between symptom burden and processing speed (indirect effect b = -0.081, p = 0.037). CONCLUSIONS: Piriform cortex function demonstrated a dynamic, nonlinear pattern within six months after mild COVID-19, marked by early hyperactivity, later downregulation, and gradual normalization. Altered piriform activity mediated the association between symptom burden and processing speed deficits, suggesting transient post-infection neurophysiological disruption with potential relevance to cognitive symptoms. Longitudinal follow-up is warranted to clarify underlying mechanisms and long-term implications.

Galeal scoring for salvage scalp closure following cranioplasty failure: a case report.

Khan M, Sajjad W, Khan S … +2 more , Haq NU, Sadbar S

BMC Neurol · 2026 May · PMID 42092799 · Full text

INTRODUCTION: Achieving tension-free scalp closure after decompressive craniectomy or cranioplasty can be quite demanding, especially in patients who have developed fibrosis or infection from previous procedures. In such... INTRODUCTION: Achieving tension-free scalp closure after decompressive craniectomy or cranioplasty can be quite demanding, especially in patients who have developed fibrosis or infection from previous procedures. In such situations, galeal scoring (galeotomies) can improve scalp mobility. Although this technique is well known in plastic surgery, it is not commonly reported in neurosurgical work. CASE PRESENTATION: A 30-year-old man sustained a high-velocity head injury that produced a large right fronto-parietal acute subdural hematoma with marked midline shift. He underwent an emergency decompressive craniectomy, leaving the bone flap off. After recovery, a delayed cranioplasty was performed using a custom implant, but the wound later became infected, requiring removal of the prosthesis and repeated debridement. As a result, the scalp contracted, and primary closure became difficult. During the final reconstruction, several small galeal-relaxing cuts were made to relieve tension and allow a comfortable closure. OUTCOME: The wound healed completely without further infection. At six months, the patient remained neurologically stable with a satisfactory cosmetic appearance. CONCLUSION: Galeal scoring offers a simple, inexpensive, and safe way to gain additional scalp mobility when closure is tight. It can often prevent the need for complex flap or graft procedures and is worth considering in similar neurosurgical cases.

Rare coding variation and stroke heterogeneity in Saudi Arabia: an exome‑wide association study across severity, etiology, vascular territory, and early‑onset disease.

Alkhamis F, Alabdali MM, Alamri AS … +9 more , Alali R, Habara AH, Akhtar MS, Alkhamis SF, Rophina M, Alkhudair AM, Vatte C, Keating B, Al-Ali AK

BMC Neurol · 2026 May · PMID 42087103 · Full text

BACKGROUND: Ischemic stroke in Saudi Arabia arises in a highly consanguineous population with a distinctive genetic architecture, likely enriching rare coding variants that influence stroke risk. Yet the contribution of... BACKGROUND: Ischemic stroke in Saudi Arabia arises in a highly consanguineous population with a distinctive genetic architecture, likely enriching rare coding variants that influence stroke risk. Yet the contribution of these variants to stroke susceptibility, age at onset, and subtype patterns in this setting remains incompletely defined. METHODS: We analyzed whole-exome sequencing data from 514 stroke patients in a case only study design. Gene-based rare variant burden analyses was performed using SAIGE-GENE+ burden and SKAT-O tests across predefined phenotype contrasts within the cohort, stroke severity (modified Rankin Scale, mild < 3 vs. severe ≥ 3 ,), age at onset (early-onset < 25 years vs. late-onset > 45 years; mid-life onset < 45 vs. > 45), etiological subtypes (TOAST classification), and vascular imaging patterns (intracranial vs. extracranial).Post-association functional annotations included GTEx expression, gnomAD constraint metrics, and draggability insights. RESULTS: Gene-level associations at a suggestive threshold (p < 0.005) identified several candidates including HSP90AB1, PRR23A, and LRRC42 (severity and age-at-onset); POGZ and SMIM34 (age-at-onset); and COL9A3, DCP1B, and ADGRV1 (imaging and etiology subtypes). The highlighted genes showed varying expression across brain and vascular tissues and intolerance to loss-of-function variation. Notably, HSP90AB1, a molecular chaperone highly expressed in the brain and vasculature, has small-molecule inhibitors, supporting its potential relevance to stroke biology. CONCLUSIONS: Our findings identify exploratory candidate gene-level signals across clinically defined ischemic stroke phenotypes based on case-only within-cohort comparisons, in underrepresented population. These results should be considered hypothesis-generating and require replication and functional validation before biological or clinical inferences can be made.

A case of autoimmune encephalitis with autoimmune gastritis presenting as refractory hyponatremia.

Sun W, Wang C, Wang Y … +3 more , Liu H, Zhang J, Bai Y

BMC Neurol · 2026 May · PMID 42087100 · Full text

BACKGROUND: The coexistence of autoimmune encephalitis (AE) and autoimmune gastritis (AIG) is exceedingly rare, and their simultaneous presentation often manifests as refractory hyponatremia, which can be easily overlook... BACKGROUND: The coexistence of autoimmune encephalitis (AE) and autoimmune gastritis (AIG) is exceedingly rare, and their simultaneous presentation often manifests as refractory hyponatremia, which can be easily overlooked. This report describes a case of anti-leucine-rich glioma-inactivated 1 (LGI1) antibody-associated AE coexisting with AIG, presenting initially as refractory hyponatremia, aiming to enhance awareness of multisystem autoimmune involvement. CASE PRESENTATION: A 77-year-old man presented with a two-month history of poor appetite, fatigue, and drowsiness, which had worsened over the preceding week. He had an 8-year history of psoriasis. Laboratory evaluation revealed profound hyponatremia (serum sodium nadir 109 mmol/L), macrocytic anemia (hemoglobin 88 g/L, mean corpuscular volume 118 fL), and deficiencies of vitamin B12 and iron. Gastroscopy with histopathological examination confirmed the diagnosis of autoimmune gastritis. Due to poor response of hyponatremia to conventional sodium supplementation and the presence of left limb spasms, further diagnostic work-up was pursued. Cerebrospinal fluid analysis tested positive for anti-LGI1 antibodies (titer 1:64), and brain magnetic resonance imaging revealed bilateral hippocampal T2-FLAIR hyperintensity and a T2 hyperintense signal in the pituitary gland, leading to the diagnosis of anti-LGI1 antibody-associated AE complicated by central adrenal insufficiency. Following methylprednisolone pulse therapy, the patient's serum sodium normalized, left limb spasms resolved, and mental status improved. At the 8-month follow-up, the patient remained clinically stable. CONCLUSION: This case highlights that in patients presenting with refractory hyponatremia accompanied by multisystem manifestations, the possibility of coexisting autoimmune diseases such as AE and AIG should be considered. Comprehensive autoimmune antibody screening and neuroimaging are crucial for early diagnosis and timely intervention.

A case of CD36 deficiency with multiple white matter lesions.

Kizuka Y, Yamakawa H, Iwabuchi Y … +8 more , Sakai J, Terayama C, Yatabe Y, Arai T, Matsushita H, Ieda M, Nakahara J, Izawa Y

BMC Neurol · 2026 May · PMID 42087097 · Full text

BACKGROUND: CD36 deficiency is associated with abnormal fatty acid metabolism, which may increase the risk of developing atherosclerosis. However, there are few reports on a possible link between CD36 deficiency and cere... BACKGROUND: CD36 deficiency is associated with abnormal fatty acid metabolism, which may increase the risk of developing atherosclerosis. However, there are few reports on a possible link between CD36 deficiency and cerebral white matter lesions. CASE REPORT: We present the case of a 44-year-old woman with heart failure due to CD36 deficiency and multiple white matter lesions. Her comprehensive examination for heart failure, including a single-photon emission computed tomography (SPECT) with thallium and I-β-methyl-p-iodophenyl pentadecanoic acid, revealed a fatty acid metabolism disorder in the myocardium. Flow cytometry confirmed CD36 deficiency, and a subsequent head magnetic resonance imaging (MRI) demonstrated multiple T2-hyperintense lesions in the cerebral white matter. Although the patient had hypertriglyceridemia and a history of smoking, the contribution of CD36 deficiency to the formation of white matter lesions remains unclear. CONCLUSION: This case suggests a potential association between CD36 deficiency and cerebral small-vessel disease. Further studies in patient cohorts with CD36 deficiency are warranted to clarify the impact of this condition on cerebral microcirculation.

Pure agraphia following a focal lesion in exner's area: a case study supporting the dual-route and network models of writing.

Gutbrod K, Albert D, Müri RM

BMC Neurol · 2026 May · PMID 42087089 · Full text

BACKGROUND: Pure agraphia represents a rare neuropsychological syndrome in which writing is selectively impaired while other language modalities remain intact, providing a unique window into the specialized cognitive and... BACKGROUND: Pure agraphia represents a rare neuropsychological syndrome in which writing is selectively impaired while other language modalities remain intact, providing a unique window into the specialized cognitive and neural mechanisms that support written language production. Drawing on the dual-route cognitive model that posits separable lexical and sublexical spelling mechanisms (Patterson, Acquired disorders of spelling, 1988; Rapp and Caramazza, J Exp Psychol: Human Perception and Performance 23:1130-152, 1997; Rapp et al., Cogn Neuropsychology 19:1-29, 2002), whereas network models emphasize distributed interactions, this case offers a multilevel analysis of the deficits observed in pure agraphia due to a focal lesion in Exner's area in the left frontal lobe (Chen et al., Sci Rep 9:55129, 2019; Hickok and Poeppel, Nat Rev Neurosci 8:393-402, 2007; Stevens et al., J Neurosci 37:5288-297, 2017; Vigneau et al. NeuroImage 30:1414-432,2006). Exner's area-located in the posterior portion of the left middle frontal gyrus, immediately anterior to the hand region of the primary motor cortex and anterior and inferior to the left Frontal Eye Field- has been implicated in the interface between linguistic and motor aspects of writing. CASE PRESENTATION: The patient was a 47-year-old right-handed male with 14 years of formal education and no prior history of language or writing deficits. Four weeks before admission, his wife reported progressive deterioration in writing, characterized by incorrect word selection and spelling errors. Mild drooling from the right corner of the mouth and food retention on the right side of the tongue subsequently appeared. CT and MRI revealed a left frontotemporal mass lesion with partial haemorrhagic transformation confined to Exner's area in the frontal lobe. Comprehensive neuropsychological and neurolinguistic assessment, including the Aachen Aphasia Test and the Lexicon Model Oriented battery, revealed intact oral language, reading, repetition, copying, and general cognition, in striking contrast to a profound writing impairment. The patient exhibited selective difficulties with irregular words, loanwords, and pseudowords, with writing accuracy declining to 40% for pseudowords and 25% for loanwords, despite 100% reading accuracy. Neuropsychological testing showed only mild deficits in verbal working memory. These findings indicate a mixed central agraphia affecting both lexical and sublexical spelling mechanisms, consistent with Exner's area functioning as a supramodal graphemic convergence hub. CONCLUSION: This case provides lesion-based evidence for the theoretical dissociation of central writing mechanisms and supports the crucial role of Exner's area acting as a critical integration hub linking phonological, orthographic, and motor processes.

The relationship between shoulder pain and joint position sense, postural control, tactile sensation, and muscle strength in chronic hemiplegia.

Kütükcü B, Yana M

BMC Neurol · 2026 May · PMID 42082948 · Full text

OBJECTIVE: The aim of this study is to investigate the relationship between shoulder pain and joint position sense, postural control, tactile sensation, and muscle strength in chronic hemiplegic stroke patients. METHODS:... OBJECTIVE: The aim of this study is to investigate the relationship between shoulder pain and joint position sense, postural control, tactile sensation, and muscle strength in chronic hemiplegic stroke patients. METHODS: The study was designed as a cross-sectional descriptive study, with 78 individuals with chronic stroke (48 males, 30 females). The participants were divided into two groups: those with shoulder pain and those without. Assessments were performed on the affected upper limb and included shoulder proprioception (inclinometer), postural control (Postural Assessment Scale for Stroke Patients (PASS-T)), tactile sensation (Semmes-Weinstein Monofilament test), gross grip strength (dynamometer), and pinch grip strength (pinch meter). RESULTS: The findings of the study revealed significant differences between individuals with and without shoulder pain across joint position sense, postural control, tactile sensation, and grip strength (all p < 0.05). Effect size analyses indicated that these differences ranged from small-to-moderate (d = 0.49-0.75) to large-to-very large (d ≥ 0.98), supporting the clinical relevance of the observed group differences. CONCLUSIONS: This study demonstrated that shoulder pain is significantly associated with motor and sensory functions in chronic hemiplegic individuals. It also underscores the necessity for rehabilitation programs to be tailored according to individuals' pain conditions, emphasizing the critical role of this factor in patient recovery processes. TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT06793852. Registered on January 21, 2025.

Association of antibodies against neural antigens with futile reperfusion in patients with anterior circulation large vessel occlusion ischemic stroke after endovascular treatment: a cohort study.

Gao B, Wang S, Han N … +4 more , Duan J, Li S, Zhang G, Chang M

BMC Neurol · 2026 May · PMID 42082929 · Full text

BACKGROUND: The prevalence and titer patterns of anti-neuronal antibodies in patients with large vessel occlusion (LVO) strokes remain unclear. The relationship between anti-neuronal antibodies and ischemic stroke progno... BACKGROUND: The prevalence and titer patterns of anti-neuronal antibodies in patients with large vessel occlusion (LVO) strokes remain unclear. The relationship between anti-neuronal antibodies and ischemic stroke prognosis remains debated. This cohort study aimed to clarify the prevalence and titer characteristics of anti-neuronal antibodies in patients with anterior circulation LVO stroke and to examine their effect on outcomes following endovascular treatment. METHOD: We retrospectively analyzed data from consecutive patients with first-ever acute ischemic stroke due to anterior circulation LVO admitted to Xi'an No. 3 Hospital (China) between November 2019 and September 2023. Logistic regression and propensity score matching were utilized to explore the relationship between anti-neuronal antibodies and outcomes in anterior circulation LVO stroke patients. Sensitivity analysis was conducted by calculating E-values and adjusting the caliper width for propensity score matching. RESULTS: Among 153 patients, 28 tested positive for antibodies against neuronal antigens, with anti-N-methyl-D-aspartate receptor 1 (NMDAR1)-IgM being the most common. Anti-dipeptidyl-peptidase (DPPX)-IgG showed the highest titers, while anti-NMDAR1-IgM was predominantly low-titer. The presence of anti-neuronal antibodies is significantly and negatively correlated with a favorable prognosis following endovascular treatment in stroke patients with anterior circulation LVO. (ORs: 0.12-0.28, all p < 0.05). E-values ranged from 3.19 to 5.22, and adjusting the caliper to 0.1 confirmed the robustness of the study's findings. CONCLUSIONS: Anti-neuronal antibodies may be associated with an increased risk of futile recanalization in patients with anterior-circulation large-vessel occlusion stroke.

The status and influencing factors of health behavior with first-ever ischemic stroke in young adults: a structural equation model.

Fan K, Qiao Y, Yang T … +2 more , Yao H, Chang H

BMC Neurol · 2026 May · PMID 42082926 · Full text

OBJECTIVES: To investigate the status of health behavior and its influencing factors in stroke among young adults. BACKGROUND: Previous research on young stroke patients has focused mainly on clinical risk factors, with... OBJECTIVES: To investigate the status of health behavior and its influencing factors in stroke among young adults. BACKGROUND: Previous research on young stroke patients has focused mainly on clinical risk factors, with limited attention paid to health behavior. Factors associated with health behavior in younger adults is unclear. METHODS: A cross-sectional survey of 220 Chinese younger adults was conducted. A structural equation model(SEM) identified factors associated with health behavior and their effect magnitude. RESULTS: The young adult participants had a mean age of 39.62 ± 4.85 years(18 to 44 years), with male accounting for the majority (74.5%, n = 164). Mean the Health Behavior Scale for Stroke Patients (HBS-SP) score was 65.64 (± 12.30). The results showed that personal mastery, social support, health literacy, self-efficacy, health belief, occupation and comorbidities were independently associated with HBS-SP(p < 0.05). SEM showed HBS-SP had the strongest association with personal mastery, followed by health literacy, Health belief, Self-efficacy, social support. CONCLUSIONS: Future research on tailored health behavior interventions should prioritize the enhancement of personal mastery, health literacy, and health beliefs, alongside the strengthening of self-efficacy and the expansion of social support systems. These interconnected psychosocial and contextual factors serve as critical underpinnings for sustainable health behavior change, and targeted efforts to optimize them will likely yield more effective and equitable intervention outcomes in clinical, community, or public health settings.

Biopsy-proven tumefactive demyelinating lesions: a systematic review and meta-analysis.

Alaskar MA, Alissa M, Alsaber N … +1 more , Aljarallah S

BMC Neurol · 2026 May · PMID 42071194 · Full text

BACKGROUND: A tumefactive demyelinating lesion (TDL) is an area of pathological changes in the central nervous system consistent with demyelination, larger than 2 cm, and can mimic tumors on imaging; hence the term "tume... BACKGROUND: A tumefactive demyelinating lesion (TDL) is an area of pathological changes in the central nervous system consistent with demyelination, larger than 2 cm, and can mimic tumors on imaging; hence the term "tumefactive." TDLs are often challenging, as they have been reported in isolation or in conjunction with demyelinating diseases. OBJECTIVES: To describe the clinical and radiological features of TDLs, explore the treatments used and outcomes, and describe different nomenclatures reported. METHODS: We followed PRISMA guidelines, and our protocol was registered on PROSPERO [CRD42024593078]. We searched PubMed, Web of Science, EMBASE, and Google Scholar. We included published cases with TDL (> 2 cm) on magnetic resonance imaging (MRI) in patients aged ≥ 18 years. We excluded a priori any established diagnoses of the lesions other than TDLs, cases without biopsy confirmation of demyelination, and cases lacking relevant data. The study framework is exploratory and descriptive in nature, and analyses are taken as hypothesis-generating. Comprehensive Meta-Analysis V3.0 was used for meta-analyses. RESULTS: We examined 112 cases from 51 publications. The average age was 41.5 years, with women representing 54% of patients. MRI revealed solitary lesions in 65% of cases, mainly in the frontal (35%) and parietal (28%) lobes, with a mean lesion size of 4.25 ± 1.32 cm. CSF analysis showed oligoclonal bands in 20% of cases. The meta-analysis demonstrated a 72% pooled response rate to intravenous methylprednisolone (IVMP) (95% CI: 57.9% - 83.2%, p < 0.01), with moderate heterogeneity (I = 62.4%), which should be interpreted cautiously given the small number of contributing studies. Subgroup analysis revealed no meaningful differences between nomenclatures. Publication bias assessments were exploratory and suggested potential bias, and that the observed variability may reflect study-level and reporting differences rather than disease-related effects. CONCLUSION: Acute IVMP treatment had a high response rate and similar use rate. Despite variations in nomenclature and presentation, the broader pathological and clinical features, as well as the response to IVMP in TDLs, appear broadly similar across studies, acknowledging the methodological limitations of the available literature. Our study was also limited by the small sample due to the rarity of the condition.

The influence of adolescent diet on the occurrence of late-onset multiple sclerosis: a population-based case-control study.

Abbasi Kasbi N, Rezaeimanesh N, Khodadadi S … +8 more , Nikkhah Bahrami S, Kohandel K, Ghane Ezabadi S, Khodaie F, Sahraian AH, Arab Bafrani M, Eskandarieh S, Sahraian MA

BMC Neurol · 2026 May · PMID 42070054 · Full text

OBJECTIVE: The role of diet in multiple sclerosis (MS) development is still a matter of debate and its impact on MS course is not well understood. We tried to investigate the possible role of adolescent diet in late-onse... OBJECTIVE: The role of diet in multiple sclerosis (MS) development is still a matter of debate and its impact on MS course is not well understood. We tried to investigate the possible role of adolescent diet in late-onset multiple sclerosis (LOMS) odds. METHODS: LOMS patients were obtained from the National MS Registry of Iran. Controls were sex and age matched with no history of neurological disorders. We assessed dietary factors using a questionnaire based on multinational studies. Food consumption levels were classified into low, medium, and high for each item. Logistic regression models were used to evaluate diet's impact on LOMS odds. RESULTS: We included 83 LOMS cases and 207 matched healthy controls. The mean age for LOMS patients was 61.14, compared to 61.51 years for controls. The results showed that higher consumption of dairy as in the third tertile associated with 79% decline in LOMS odds (AOR: 0.21; 95%CI: 0.09-0.47). Higher seafood consumption, in the third tertile was associated with a decrease in the odds of LOMS (AOR: 0.32; 95%CI: 0.14-0.72). Furthermore, increased nut consumption, in the third tertile (AOR: 0.37; 95%CI: 0.18-0.77), decreased the odds of LOMS. Additionally, higher consumption of fruits (AOR: 0.22; 95%CI: 0.07-0.63) and vegetables (AOR: 0.26; 95%CI: 0.12-0.55) was linked to a reduced odds of LOMS. CONCLUSION: This research highlights the advantageous impact of dairy products, seafood, nuts, fruits, and vegetables in lowering the LOMS odds. Hence, advocating for nutrition role in development of LOMS could represent a preventive measure for people susceptible to MS.

Diagnostic and therapeutic challenges in anti-NMDA receptor encephalitis associated with an ovarian immature teratoma.

Doudein M, Doudin SM, Lahlouh RA … +2 more , Omarya O, Ismail A

BMC Neurol · 2026 May · PMID 42067849 · Full text

BACKGROUND: Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is an autoimmune disorder characterized by antibodies against the NR1 subunit of the NMDA receptor. It predominantly affects young women and is fre... BACKGROUND: Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is an autoimmune disorder characterized by antibodies against the NR1 subunit of the NMDA receptor. It predominantly affects young women and is frequently associated with ovarian teratomas. Early recognition, tumor removal, and immunotherapy are critical for favorable outcomes. CASE PRESENTATION: We report a 33-year-old woman with subacute headache, low-grade fever, generalized weakness, and mood disturbances, who developed generalized tonic-clonic seizures and cognitive impairment. Brain MRI and CSF were initially normal, and EEG showed diffuse slowing without epileptiform discharges. Pelvic imaging revealed a large right ovarian mass. She was treated with corticosteroids, plasmapheresis, and ultimately rituximab, alongside right salpingo-oophorectomy. Histopathology confirmed an immature teratoma. Anti-NMDAR antibodies were later detected in CSF, confirming the diagnosis. Following tumor removal and immunotherapy, she achieved complete neurological recovery and remained seizure-free at six months. CONCLUSION: This case highlights the diagnostic challenges of anti-NMDAR encephalitis, particularly when MRI and CSF are normal. Early tumor detection and removal, combined with prompt escalation to second-line immunotherapy when needed, are essential for favorable outcomes. Immature ovarian teratomas, although rare, can trigger severe disease and warrant careful evaluation in adult women presenting with acute neuropsychiatric symptoms.
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