AIMS/BACKGROUND:It remains unclear whether vascular characteristics or hemodynamics of the vertebrobasilar artery (VBA) among patients with left- and right-sided hemifacial spasm (HFS) lesions correlate with the risk of HFS occurrence and disease severity. This study aims to investigate the correlation of VBA characteristics with the incidence risk and severity of HFS. METHODS:A total of 60 patients with HFS who were admitted to Sir Run Run Shaw Hospital, Zhejiang University School of Medicine, between January and October 2024 were retrospectively enrolled in the HFS group. Another 59 healthy individuals who underwent physical examinations were assigned to the control group. Data on the characteristics of the VBA were collected for both groups, and the correlation between these characteristics and the occurrence risk of HFS was analyzed. In addition, patients in the HFS group were further stratified according to the lesion side (left vs right) and HFS severity grade (mild vs moderate vs severe) for subgroup analysis and comparison of VBA imaging features. RESULTS:The diameters of the left vertebral artery (VA), right VA, and basilar artery in the HFS group were all found to be larger than those in the control group, and the difference in the diameter of the left VA between the two groups was more significant (p < 0.05). Multivariate logistic regression analysis demonstrated that the diameter of the left VA (odds ratio [OR] = 4.014, 95% confidence interval [CI]: 1.997-8.070), the diameter of the right VA (OR = 3.890, 95% CI: 2.217-6.825), and the diameter of the basilar artery (OR = 2.022, 95% CI: 1.008-4.058) were independent influencing factors for the occurrence of HFS (p < 0.05). There were no statistically significant differences in the diameters of the VA and basilar artery among patients with severe, moderate, and mild HFS (p > 0.05). CONCLUSION:The diameters of the left VA, right VA, and basilar artery are significantly correlated with the occurrence of HFS, while the severity of the disease shows no correlation with the diameter of the ipsilateral VA. This suggests that the morphological changes of the VBA system may play an important role in the pathogenesis of HFS.
Background: Obsessive‒compulsive disorder is a chronic, disabling mental disorder. While repetitive transcranial magnetic stimulation has emerged as a promising neuromodulation intervention for psychiatric disorders, its efficacy in treatment-naïve obsessive‒compulsive disorder patients remains understudied. Objective: This study aimed to test the preliminary efficacy of low-frequency repetitive transcranial magnetic stimulation in treatment-naïve obsessive‒compulsive disorder patients. Methods: Treatment-naïve obsessive‒compulsive disorder patients (n = 41) were randomized to receive either standardized fluvoxamine therapy (150-200 mg/day) or daily low-frequency (1 Hz) repetitive transcranial magnetic stimulation targeting the supplementary motor area for 2 weeks. Clinical outcomes were longitudinally assessed via validated instruments, with a Yale-Brown Obsessive-Compulsive Scale score reduction rate ≥ 25% as the primary endpoint, supplemented by the Beck Depression Inventory and Beck Anxiety Inventory for comorbid symptom evaluation. Safety profiles were monitored throughout the trial. Results: The experimental results revealed that the difference in the response rate at the end of the intervention between the two groups was not statistically significant ( χ2 = 0.183, p = 0.669), with 41.7% (5/12) in the repetitive transcranial magnetic stimulation group and 60% (6/10) in the fluvoxamine cohort. No severe adverse events were reported in either group. Conclusion: This trial revealed that low-frequency repetitive transcranial magnetic stimulation over the supplementary motor area might have preliminary positive outcomes for treatment-naïve patients with obsessive‒compulsive disorder. Our findings can be considered a good signal to promote further research in the form of randomized, double-blind, sham-controlled multicenter trials with extended follow-up periods.
BackgroundRetroclival subdural hematoma (rcSDH) secondary to spontaneous intracranial hypotension (SIH) is an exceedingly rare clinical entity, characterized by complex and incompletely understood pathophysiological mechanisms.CaseA 24-year-old female presented with acute and persistent orthostatic headache, with no history of trauma or anticoagulant therapy. Neuroimaging revealed subdural hematomas (SDH) located in the retroclival, infratentorial, and right frontal regions. It was hypothesized that veinous rupture, resulting from venous traction due to decreased cerebrospinal fluid (CSF) pressure, was the underlying mechanism. Following epidural blood patch (EBP) therapy, the patient exhibited marked symptomatic improvement and radiological resolution of hematomas on follow-up imaging.ConclusionRcSDH is considered an uncommon complication of SIH, potentially resulting from venous rupture in the retroclival subdural space due to reduced CSF pressure. SIH should be considered in cases of rcSDH. The treatment is typically focused on addressing the underlying etiology, with early diagnosis and timely intervention being essential for achieving favorable outcomes. In cases of severe brainstem compression, hematoma evacuation should be performed in conjunction with EBP.
Recently, mutations affecting a microRNA-29 (miR-29)-binding site in the 3’-untranslated region of COL4A1 have been identified as a cause of pontine autosomal dominant microangiopathy with leukoencephalopathy (PADMAL) and hereditary multi-infarct dementia (hMID) of the Swedish type. PADMAL and Swedish hMID are extremely rare disorders with no de novo mutations previously reported. A patient with cerebral small vessel disease underwent comprehensive neurological examinations, neuroimaging analysis, and whole-exome sequencing. Co-segregation analysis was performed on his family, and haplotype analysis was conducted to confirm the biological relationship. The patient experienced recurrent ischemic strokes since age 32. Brain MRI showed multiple acute and chronic lacunar infarcts in the pons and bilateral cerebral hemispheres. A previously reported pathogenic mutation of PADMAL, COL4A1 c.*32G > T, was identified and found to be absent in both parents. Identity testing confirmed the biological parentage, classifying the c.*32G > T variant as a de novo mutation. The discovery of PADMAL in a patient with a de novo mutation indicates that COL4A1 gene miR-29-binding site variant sequencing should be considered in patients exhibiting typical clinical and MRI features, even if there is no family history.
BACKGROUND:Large duplications or triplications involving the 13q33-34 chromosomal region, which encompass the COL4A1 and COL4A2 genes, have been reported in association with cerebral small vessel disease (CSVD) in a few patients. Herein, we report an additional case of CSVD linked to a duplication of COL4A1 and COL4A2 and provide a detailed summary of the associated clinical and MRI findings. METHODS:A patient with CSVD underwent detailed clinical and neuroimaging evaluations. Targeted next-generation sequencing (NGS) and copy number variation sequencing (CNV-seq) based on whole genome sequencing were used to identify the genetic basis of the disease. RESULTS:The patient experienced his first ischemic stroke at age 51. Cranial MRI revealed extensive acute and chronic lacunar infarcts and white matter hyperintensities across both cerebral hemispheres, with involvement of the anterior temporal lobe and the external capsule. Bilateral thalamic microbleeds were also noted. The clinical features and MRI findings are similar to those observed in cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL). Targeted NGS and CNV-seq analysis identified a duplication in the region of chromosome 13, which included the COL4A1 and COL4A2 genes. CONCLUSIONS:This case provides further evidence supporting the association of CNVs in COL4A1 and COL4A2 with CSVD. When hereditary CSVD is suspected and no micro-mutations in CSVD-associated genes are identified, CNV analysis of the 13q region should be considered.
BACKGROUND:Functional movement disorders (FMDs) are common within functional neurological disorders, yet understudied in Asian populations, particularly in China. Understanding FMDs across diverse cultural and ethnic contexts is crucial for elucidating disease mechanisms and optimizing treatment strategies. This study aimed to characterize FMDs among Chinese Han individuals and identify key prognostic factors. METHODS:We enrolled 119 FMD patients from 22 centers across China. Data collected included demographics, clinical manifestations, neuropsychological assessments, and treatment details. Statistical analyses including ANOVA, chi-square tests, logistic regression, and so on were used to analyze the clinical characteristics and potential prognostic predictors of FMD patients in China. RESULTS:Patients showed a mean onset age of 45.3 years, female (58.8%), and a possible bimodal age distribution (peaks at 20-30 and ≥ 60 years). Mixed phenotypes (32.8%) and tremor (29.4%) were most common, with high rates of anxiety (61.3%) and depression (53.8%), and 38.7% clinical symptom remission. Physical therapy may be a potential protective factor (OR = 0.077, p < 0.001), while trauma history (OR = 7.863, p = 0.002) and higher baseline CGI scores (OR = 1.933, p = 0.002) predicted poorer outcomes. CONCLUSION:This first multi-center study of FMDs in China highlights a potential tendency toward a bimodal distribution, female predominance, and abnormal scores on psychiatric scales. Notably, physical therapy represents a potential protective factor, while trauma history may be a risk factor. Our findings identify the clinical profile and prognostic factors of FMDs in the Chinese population, offering valuable insights for clinical practice and future research.
BACKGROUND:The safety and effectiveness of deep brain stimulation of the subthalamic nucleus (STN-DBS) for the treatment of dystonia lack high-level evidence-based medical support. This study aimed to clarify the efficacy and safety of STN-DBS and perform a post hoc analysis comparing it with DBS of the internal globus pallidus (GPi-DBS). METHODS:This multicentre, randomised, double-blind, controlled trial included 67 patients aged 6-60 years old diagnosed with genetic or idiopathic isolated generalised or segmental dystonia. They were enrolled from seven hospitals in China and randomly assigned to undergo GPi-DBS or STN-DBS. After surgery, they were randomised to receive either neurostimulation or sham stimulation for 3 months. At the 3-month follow-up, neurostimulation was also initiated in the sham stimulation group, and all patients were followed up for more than 3 years after treatment. The primary outcome was the Burke-Fahn-Marsden Dystonia Rating Scale movement (BFMDRS-M) score. RESULTS:In the STN group, the neurostimulation subgroup exhibited significant improvement (p<0.001), which is also superior to the sham stimulation subgroup (p=0.028) at 3-month follow-up. At the 6-month and >3-year follow-ups, all patients receiving STN-DBS showed a significant improvement in BFMDRS-M scores (p<0.001). Further post hoc analysis revealed that both STN-DBS and GPi-DBS could produce similar therapeutic effects on motor symptoms (P6 months=0.865, P>3 years=0.905). There were no ongoing serious adverse events throughout the study. CONCLUSIONS:For isolated generalised and segmental dystonia patients, the STN is a selectable DBS target with ensured safety and efficacy. STN-DBS and GPi-DBS may achieve comparable therapeutic effects on motor symptoms. TRIAL REGISTRATION NUMBER:NCT03017586.
BACKGROUND:DaxibotulinumtoxinA for injection (DAXI), the first long-acting botulinum toxin (BoNT) type A, is FDA approved for cervical dystonia (CD). DAXI's novel formulation, which includes a custom-engineered peptide, is designed to provide an extended duration of clinical benefit. OBJECTIVE:To evaluate the pooled efficacy and safety of DAXI for CD across two phase 3, multicenter, randomized, double-blind, placebo-controlled trials: ASPEN-1, conducted in North America and Europe, and ASPEN-1-CN, a similarly designed, smaller pivotal clinical trial, conducted in China. METHODS:Adults with moderate-to-severe CD were randomized (3:3:1) to receive DAXI 125U, DAXI 250U, or placebo. The primary endpoint was change from baseline in Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) total score averaged across weeks 4 and 6. A key secondary endpoint was duration, defined as time until loss of >80 % of peak effect. RESULTS:In all, 357 subjects were randomized and received DAXI 125U (n = 149), DAXI 250U (n = 154), or placebo (n = 54). DAXI 125U (-12.0) and DAXI 250U (-11.9) significantly improved the mean TWSTRS total score versus placebo (-4.6; P < 0.0001). Median (95 % CI) duration of effect was 24.1 (20.6-28.9) weeks for DAXI 125U and 22.0 (20.1-24.3) weeks for DAXI 250U. Rates of treatment-related dysphagia (125U: 4.7 %, 250U: 4.5 %) and muscle weakness (125U: 5.4 %, 250U: 4.5 %) were low for both active doses. CONCLUSIONS:This pooled analysis of two phase 3 trials demonstrates that DAXI is an effective, safe, and long-acting treatment for CD. Key adverse events occurred at rates lower than prior pivotal trials of BoNTs for CD.
Neutrophils are among the earliest and most abundant immune cells infiltrating the brain following ischemic stroke, aggravating neuroinflammation through the formation of neutrophil extracellular traps (NETs). Pyroptosis, an inflammasome-mediated form of programmed cell death, occurs in post-stroke brain tissue and amplifies inflammation by releasing proinflammatory mediators, propagating the inflammatory cascade. However, the mechanistic link between NETs and pyroptosis remains unclear. This study demonstrated significantly elevated NET levels in arterial blood at the infarct site compared with venous or femoral arterial blood in stroke patients. A positive correlation was observed between the 24-h change in NIHSS score (NIHSSbaseline - NIHSS24h) and the difference in arterial citrullinated histone 3 (CitH3)-DNA (NETs) levels between the infarct site and femoral artery (NETsinfarct site - NETsfemoral artery). In a murine stroke model, NETs were detected in the brain parenchyma. Pharmacological inhibition of NET formation with GSK484, a selective protein-arginine deiminase type 4 antagonist, suppressed NET production, reduced absent in melanoma 2 (AIM2) inflammasome expression, and improved neurological outcomes in mice following stroke. AIM2 knockdown in brain tissue achieved similar neuroprotective effects. In both BV2 cells and stroke-induced mice, NETs triggered AIM2-dependent pyroptosis. These findings suggest that neutrophils in peripheral blood infiltrate the brain parenchyma to generate NETs, activating the AIM2 inflammasome in microglia and exacerbating stroke-induced brain injury through pyroptosis. Targeting NET formation or AIM2 inflammasome activation represents a potential therapeutic strategy for attenuating post-stroke neuroinflammation and secondary neuronal damage.
BACKGROUND:Hereditary hemorrhagic telangiectasia (HHT) is a rare autosomal dominant inherited vascular disorder that can involve multiple organs, thus can be associated with so many clinical departments that proper screening and diagnosis of HHT are needed for providing better management of both patients and their family members. CASE PRESENTATION:We present a 58-year-old female patient with recurrent paradoxical brain embolism due to HHT. She received aspirin therapy and underwent pulmonary arteriovenous malformation embolization, recovering well and discharged 3 days postoperatively. Though ischemic stroke caused by HHT-induced vascular disorders has been reported, our patient presented with both recurrent paradoxical brain embolisms and radiologic findings of bilateral globus pallidus manganese deposition at the same time, a combination rarely reported. We also review the literature on the clinical features and management of HHT for prompt diagnosis of this genetic disease behind paradoxical embolism. CONCLUSIONS:When patients with ischemic stroke, especially recurrent ischemic stroke, have combined arteriovenous malformations (AVMs) in single or multiple organs, or clues for AVMs like manganese deposition in globus pallidus, genetic diseases such as HHT may be the reason for ischemic stroke and shouldn't be missed in the evaluation of embolic sources.
BackgroundAcquired prolapse of the cerebellar tonsils in spontaneous intracranial hypotension (SIH) patients is rare. This study aims to evaluate neuroimaging changes of acquired prolapse of the cerebellar tonsils below the foramen magnum in SIH patients due to spontaneous spinal cerebrospinal fluid leakage, which was treated by targeted epidural blood patches (EBP).MethodsWe retrospectively reviewed clinical and neuroimaging characteristics of 5 cases of SIH with acquired prolapse of the cerebellar tonsils that received targeted EBP in our institution from January 2013 to December 2016.ResultsOf these SIH patients, all of them suffered from an orthostatic headache. Initial cranial MRI demonstrated descent of the cerebellar tonsils ≥5 mm. Intrathecal gadolinium-enhanced spinal MR myelography and/or spinal MR hydrography were performed to evaluate the level of spinal cerebrospinal fluid leakage. Symptoms were alleviated in all 5 patients after two (n = 4), or three (n = 1) targeted EBP during hospitalization. Follow-up cranial MRI revealed that the descent of cerebellar tonsils was reversed after EBP treatment.ConclusionAcquired tonsillar herniation can occur in patients with SIH and spinal cerebrospinal fluid leakage. Symptoms of these patients may be resolved and radiologic findings may be reversed after EBP treatment.
Abstract Objective The Bern score is based on brain magnetic resonance imaging (MRI) to predict the probability of cerebrospinal fluid (CSF) leak in spontaneous intracranial hypotension (SIH). The aim of this study is to investigate the association between lumbar puncture opening pressure (OP) and the Bern score. Methods We retrospectively reviewed OP measurement records and neuroimaging of patients with SIH in our center. The Bern score and its components were measured based on contrast‐enhanced brain MRI. The associations between OP and the Bern score, as well as its components, were analyzed. Patients were divided as low‐pressure (LP) group (OP < 60 mmH2O) and not‐low‐pressure (NLP) group (OP ≥ 60 mmH2O). Differences in terms of the Bern score and its components were compared between the two groups. Results Seventy‐one (mean age 40.4 ± 10.6 years) patients with myelography confirmed CSF leak were included in this study. The mean disease duration when performed brain MRI was 32 ± 29 days, with a mean Bern score of 5.1 ± 2.7 and a mean OP of 68.6 ± 60.3 mmH2O. There are statistically negative correlations between OP and the Bern score (p < .001), as well as suprasellar cistern (p < .01) and prepontine cistern (p < .01). The presence of venous engorgement (p < .01) and pachymeningeal enhancement (p < .001) were significantly associated with OP. The LP groups have higher Bern scores than the NLP group (5.9 ± 2.5 vs. 4.2 ± 2.6, p = .004). Conclusions A higher Bern score is indicative of not only an increased likelihood of a CSF leak but also a greater probability of low OP in patients with SIH. For patients with a Bern score ≥5 and positive heavily T2‐weighted MR myelography findings, epidural blood patch is reasonable before invasive myelography.
INTRODUCTION:Rasagiline is indicated for treating idiopathic Parkinson's disease (PD) as monotherapy and adjunct therapy to levodopa in patients.OBJECTIVES:To assess the post-marketing safety and tolerability of rasagiline in Chinese PD patients, as well as its effectiveness in improving motor symptoms.METHODS:This prospective, non-interventional, multicenter, cohort study included PD patients administered rasagiline monotherapy or adjunct therapy to levodopa. The primary outcome was the incidence of adverse drug reactions (ADRs) according to MedDRA® (version 22.0), and the secondary outcomes were the Parkinson's Disease Unified Rating Scale (UPDRS) part III, Clinical Global Impression-Severity (CGI-S), and Clinical Global Impression-Global-Improvement (CGI-I), assessed at Weeks 4, 12, and 24.RESULTS:In total, 734 patients, 95 in the monotherapy subgroup and 639 in the adjunct therapy subgroup, were included in the safety population. The incidence rates of all ADRs were comparable between the monotherapy (15.8%) and adjunct therapy (13.6%) subgroups. The most common ADRs by system organ class were nervous system disorders (5.6%), gastrointestinal disorders (3.3%), psychiatric disorders (1.8%), vascular disorders (1.2%), and general disorders and administration site conditions (1.1%). Five (0.7%) participants experienced 5 serious ADRs. Improvements in UPDRS part III, CGI-S and CGI-I at Weeks 4, 12 and 24 from baseline were observed.CONCLUSIONS:Safety data in this study indicated no extra safety concerns. Rasagiline is generally safe and well tolerated in Chinese PD patients. The safety profile and tolerability were in line with the established safety profile. Moreover, rasagiline reduced the severity of PD motor symptoms, confirming findings by previous clinical trials.
Background Hereditary hemorrhagic telangiectasia (HHT) is a rare autosomal dominant inherited vascular disorder that can involve multiple organs, thus can be associated with so many clinical departments that proper screening and diagnosis of HHT are needed for providing better management of both patients and their family members. Case presentation: we present a 58-year-old female patient with recurrent paradoxical brain embolism due to HHT. Though ischemic stroke caused by HHT-induced vascular disorders has been reported, our patient presented with two neurological complications at the same time: recurrent paradoxical brain embolisms and bilateral globus pallidus manganese deposition, which is rarely reported. We also review the literature on the clinical features and management of HHT for prompt diagnosis of this genetic disease behind paradoxical embolism. Conclusions When patients with ischemic stroke, especially recurrent ischemic stroke, have combined AVMs in single or multiple organs, or clues for AVMs like manganese deposition in globus pallidus, genetic diseases such as HHT may be the reason for ischemic stroke and shouldn't be missed in the evaluation of embolic sources.
—Although Cervical Dystonia (CD) is regarded as the most common type of focal dystonia, the therapeutic options available for CD are remarkably limited and are unsatisfactory for many patients. Recent functional and clinical studies have made great progress in unraveling the mechanisms underlying CD and other types of dystonia. Much research is still needed; however, the deeper understanding of the etiology of CD is expected to lead to better management of dystonia symptoms and the development of novel therapeutic options. The objective of this review was to summarize the most recent studies examining the pathophysiological features of CD, including genetic mutations, studies about neuronal signaling, and metabolomic studies.
Objective To preliminarily summarize the epidemiological and clinical characteristics of functional movement disorder(FMD)based on movement disorders clinic in China.Methods and Results A total of 593 patients with movement disorders newly diagnosed in movement disorders clinic of 22 clinical medical centers from August to September in 2023 were included,including 37 patients(6.24%)with FMD and 556 patients(93.76%)with organic movement disorder.Comparison of the epidemiological characteristics of the 2 groups,the proportions of corona virus disease(COVID-19)infection(x2=4.217,P=0.040)and psychopsychological symptoms(x2=18.694,P=0.000)in the FMD group were higher than those in the organic movement disorder group,while age(t=3.757,P=0.000),age of onset(t=3.720,P=0.000)and proportion of hypertension(x2=4.736,P=0.030)were lower than those in the organic movement disorder group.Comparison of the clinical characteristics of the 2 groups,the FMD group had higher proportions of tremor(x2=3.955,P=0.047)and myoclonus(Fisher's exact probability:P=0.011)than the organic movement disorder group,and the proportions of parkinsonism(x2=8.491,P=0.004)and gait disorder(x2=5.028,P=0.025)were lower than those in the organic movement disorder group.Conclusions The age and age of onset,as well as the proportions of COVID-19 infection,hypertension,psychopsychological symptoms,tremor,myoclonus,parkinsonism and gait disorder with FMD were different from those with organic movement disorder in China.
Botulinum toxin type A (BoNT/A) is extensively applied in spasticity and dystonia as it cleaves synaptosome-associated protein 25 (SNAP25) in the presynaptic terminals, thereby inhibiting neurotransmission. An increasing number of randomized clinical trials have suggested that glabellar BoNT/A injection improves depressive symptoms in patients with major depressive disorder (MDD). However, the underlying neuronal circuitry of BoNT/A-regulated depression remains largely uncharacterized. Here, we modeled MDD using mice subjected to chronic restraint stress (CRS). By pre-injecting BoNT/A into the unilateral whisker intrinsic musculature (WIM), and performing behavioral testing, we showed that pre-injection of BoNT/A attenuated despair- and anhedonia-like phenotypes in CRS mice. By applying immunostaining of BoNT/A-cleaved SNAP25 (cl.SNAP25197), subcellular spatial localization of SNAP25 with markers of cholinergic neurons (ChAT) and post-synaptic membrane (PSD95), and injection of monosynaptic retrograde tracer CTB-488-mixed BoNT/A to label the primary nucleus of the WIM, we demonstrated that BoNT/A axonal retrograde transported to the soma of whisker-innervating facial motoneurons (wFMNs) and subsequent transcytosis to synaptic terminals of second-order neurons induced central effects. Furthermore, using transsynaptic retrograde and monosynaptic antegrade viral neural circuit tracing with c-Fos brain mapping and co-staining of neural markers, we observed that the CRS-induced expression of c-Fos and CaMKII double-positive neurons in the ventrolateral periaqueductal grey (vlPAG), which sent afferents to wFMNs, was down-regulated 3 weeks after BoNT/A facial pre-administration. Strikingly, the repeated and targeted silencing of the wFMNs-projecting CaMKII-positive neurons in vlPAG with a chemogenetic approach via stereotactic injection of recombinant adeno-associated virus into specific brain regions of CRS mice mimicked the antidepressant-like action of BoNT/A pre-treatment. Conversely, repeated chemogenetic activation of this potential subpopulation counteracted the BoNT/A-improved significant antidepressant behavior. We reported for the first time that BoNT/A inhibited the wFMNs-projecting vlPAG excitatory neurons through axonal retrograde transport and cell-to-cell transcytosis from the injected location of the WIM to regulate depressive-like phenotypes of CRS mice. For the limited and the reversibility of side effects, BoNT/A has substantial advantages and potential application in MDD.
BACKGROUND Subdural hematoma (SDH) is a potentially life-threatening complication in patients with spontaneous intracranial hypotension (SIH). Though bed rest is the basis of conservative treatment, no clear evidence exists regarding the association between bed rest and the later complication of SDH in these patients. OBJECTIVES This study aimed to evaluate the association between bed rest and SDH development in patients with SIH. STUDY DESIGN A retrospective study was conducted from March 2013 through December 2019. Four hundred twenty adult patients diagnosed with SIH were enrolled. Clinical presentations and radiographic findings were recorded. The cumulative duration of bed rest in hours was used to measure the bed rest length. The clinical outcomes during follow-up were assessed. METHODS Categorical data were compared using chi-square tests; continuous data were compared using the Mann-Whitney U test or Kruskal-Wallis test. A backwards stepwise Cox proportional hazard regression model adjusted with confounders which differed between SDH and non-SDH in univariate analysis was used to estimate the risk of cumulative duration of bed rest for SDH. A stratified Cox regression was performed to exclude the effect of the treatment algorithm. RESULTS Of the 420 patients with SIH, 88 (21%) were in the SDH Group and 332 (79%) were in the non-SDH (NSDH) Group. The cumulative duration of bed rest in hours was a protective factor for SDH in SIH (Hazard Ratio [HR] = 0.997; P < 0.001). A stratified Cox regression analysis showed that the cumulative duration of bed rest remained a protective factor for SDH both in patients who received conservative treatment before admission (HR = 0.997; P < 0.001) and in those who did not (HR = 0.996; P = 0.061). Age (HR = 1.029, 95% CI, 1.009-1.050; P = 0.004) and orthostatic headache (HR = 4.770, 95% 95% CI, 2.177-10.450; P < 0.001) were risk factors for SDH in SIH. The clinical outcomes, including length of hospital stay, epidural blood patch (EBP) therapy, and repeated EBP therapy, were higher in the SDH Group. The revisit rate was similar between the 2 groups. LIMITATIONS Retrospective studies are susceptible to different radiological procedures and therapeutic strategies. A bed rest score based on a patient's memory is susceptible to recognition and reporting bias. This is a single-center study and the sample size is not large. The validity of the bed rest scale has not been previously evaluated in any other study. CONCLUSIONS Bed rest was a protective factor for SDH in patients with SIH. With more time and proper treatment, patients with SIH who have an SDH can achieve good prognosis in the long term.
Background: Isolated cortical vein thrombosis (ICVT), a rare type of cerebral venous thrombosis (CVT), is diagnostically challenging in some cases, and intracranial hypotension (IH) is known to cause CVT. Methods: In this study, we reviewed the clinical and imaging characteristics of ICVT in patients with IH caused by spinal cerebrospinal fluid leakage, based on a literature review and investigation of cases from our hospital. Results: Between January 1, 2007, and November 1, 2019, 735 patients were diagnosed with IH at our hospital; three patients developed ICVT (incidence similar to 0.4%, 3/735), and the literature review yielded an additional 23 cases. Therefore, 26 patients (mean age 35.9 +/- 11.4 years old) were included in this study. The most common symptoms were headache (100.0%, 26/26), focal neurological deficits (53.8%, 14/26), and seizure (34.6%, 9/26). The initial headache was orthostatic in 96.2% (25/26) of patients, and 38.5% (10/26) of patients reported a change in the headache pattern following diagnosis of ICVT. Neuroimaging findings associated with ICVT included the cord sign (61.5%, 16/26) and parenchymal brain lesions (46.2%, 12/26), such as intracerebral hemorrhage (30.8%, 8/26), hemorrhagic infarcts (11.5%, 3/26), and localized edema (11.5%, 3/26). The percentage of patients who received anticoagulation and epidural blood patch therapy was similar (69.2% [18/26] vs. 65.4% [17/26]), and most patients recovered completely (92.3%, 24/26). Conclusion: IH should be considered in the differential diagnosis in patients with ICVT. Knowledge of the relevant clinical and neuroimaging features is important to facilitate early diagnosis for favorable prognosis.