Effect of ultrasound-guided injection of botulinum toxin type A into shoulder joint cavity on shoulder pain in poststroke patients: study protocol for a randomized controlled trial.

BACKGROUND: Hemiplegic shoulder pain (HSP) is a common complication after stroke. It severely affects the recovery of upper limb motor function. Early shoulder pain in hemiplegic patients is mainly neuropathic caused by central nerve injury or neuroplasticity. Commonly used corticosteroid injections in the shoulder joint can reduce shoulder pain; however, the side effects also include soft tissue degeneration or increased tendon fragility, and the long-term effects remain controversial. Botulinum toxin injections are relatively new and are thought to block the transmission of pain receptors in the shoulder joint cavity and inhibit the production of neuropathogenic substances to reduce neurogenic inflammation. Some studies suggest that the shoulder pain of hemiplegia after stroke is caused by changes in the central system related to shoulder joint pain, and persistent pain may induce the reorganization of the cortical sensory center or motor center. However, there is no conclusive evidence as to whether or not the amelioration of pain by botulinum toxin affects brain function. In previous studies of botulinum toxin versus glucocorticoids (triamcinolone acetonide injection) in the treatment of shoulder pain, there is a lack of observation of differences in changes in brain function. As the content of previous assessments of pain improvement was predominantly subjective, objective quantitative assessment indicators were lacking. Functional near-infrared imaging (fNIRS) can remedy this problem. METHODS: This study protocol is designed for a double-blind, randomized controlled clinical trial of patients with post-stroke HSP without biceps longus tenosynovitis or acromion bursitis. Seventy-eight patients will be randomly assigned to either the botulinum toxin type A or glucocorticoid group. At baseline, patients in each group will receive shoulder cavity injections of either botulinum toxin or glucocorticoids and will be followed for 1 and 4 weeks. The primary outcome is change in shoulder pain on the visual analog scale (VAS). The secondary outcome is the assessment of changes in oxyhemoglobin levels in the corresponding brain regions by fNIRS imaging, shoulder flexion, external rotation range of motion, upper extremity Fugl-Meyer, and modified Ashworth score. DISCUSSION: Ultrasound-guided botulinum toxin type A shoulder joint cavity injections may provide evidence of pain improvement in patients with HSP. The results of this trial are also help to analyze the correlation between changes in shoulder pain and changes in cerebral hemodynamics and shoulder joint motor function. TRIAL REGISTRATION: Chinese clinical Trial Registry, ChiCTR2300070132. Registered 03 April 2023, https://www.chictr.org.cn/showproj.html?proj=193722 .

Real life retrospective study of cannabidiol therapy in alternating hemiplegia of childhood.

BACKGROUND: Many alternating hemiplegia of childhood (AHC) patients have received Cannabidiol (CBD) but, to our knowledge, there are no published data available. GOALS: Test the hypothesis that CBD has favorable effects on AHC spells. METHODS: Retrospective review of available data of AHC patients who received CBD. Primary analysis: Clinical Global Impression Scale of Improvement (CGI-I) score for response of AHC spells to CBD with calculation of 95% confidence interval (CI) for rejection of the null hypothesis. Secondary analyses, performed to achieve an understanding of the effect of CBD as compared to flunarizine, were CGI-I scores of 1) epileptic seizures to CBD, 2) AHC spells to flunarizine, 3) epileptic seizures to flunarizine. Also, Mann-Whitney test was done for comparison of CGI-I scores of CBD and flunarizine to both AHC spells and seizures. RESULTS: We studied 16 AHC patients seen at Duke University and University of Lyon. CI of CGI-I scores for AHC spells in response to CBD and to flunarizine, each separately, indicated a positive response to each of these two medications: neither overlapped with the null hypothesis score, 4, indicating significant positive responses with p < 0.05 for both. These two scores also did not differ (p = 0.84) suggesting similar efficacy of both: CBD score was 2 ± 1.1 with a 95% CI of 1.5-2.6 and flunarizine score was 2.3 ± 1.3 with a 95% CI of 1.7-3.1. In patients who had seizures, CI calculations indicated a positive effect of CBD on seizure CGI scores but not of flunarizine on seizure scores. CBD was well tolerated with no patients discontinuing it due to side effects and with some reporting positive behavioral changes. CONCLUSION: Our study indicates a real-life positive effect of CBD on AHC type spells.

Curative effect of medicine cake sticking ultrasound drug penetration combined with body training on hemiplegia after stroke: An in vitro ultrasound targeted drug controlled release technology.

To explore the efficacy of ultrasound drugs in the treatment of hemiplegia after stroke. The evaluation included clinical symptoms and signs, the Stroke Scale, activities of daily living, sensory disorder Fugl-Meyer and Lindmark, electromyography sensory nerve amplitude, and conduction velocity indexes in both groups. There was no significant difference in the improved Fugl-Meyer and Lindmark score between treatment (26.97 ± 2.78) and the control group (27.45 ± 3.1) (t = 14.528, P = 0.593). After treatment, the observation group (37.10 ± 4.2) was significantly different from the control group (34.76 ± 4.36) (t = 11.259, P = 0.005) and (t = 10.15 ± 1.69), (40.87 ± 6.58) (t = 7.943,9.538, P = 0.564,0.826). After treatment, the observation group the Stroke Scale (4.27 ± 0.57), activities of daily living score (76.15 ± 12.38) and the control group (5.36 ± 0.89), (58.41 ± 9.69) (t = 16.274,5.379, P = 0.035,0.000) after treatment and F wave and M wave. The cure rate of the observation group was 77.50% (31/40), which was significantly better than that of the control group, 47.50% (19/40), with a significant difference (χ2 = 11.724,P = 0.000). After comparison, the total response rate of the observed group reached 92.500% (37 / 40), which was significantly higher than the 80.00% (32 / 40) of the control group. This difference was statistically significant (χ 2 = 9.458, P = 0.015). This therapy closely links the theoretical knowledge of modern medicine with the theoretical knowledge of traditional Chinese medicine, and uses the meridian theory to give full play to the unique advantages of traditional Chinese medicine.

Ultrasonography-guided botulinum toxin injection for lumbrical muscle spasticity in a hemiplegic patient.

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Efficiency of botulinum toxin injection into the arm on postural balance and gait after stroke.

The purpose of this study was to clarify the association between improvement of spasticity in hemiplegic patient's upper extremity with Botulinum toxin injection and improvement in postural balance and gait function. For this prospective cohort study, sixteen hemiplegic stroke patients with upper extremity spasticity were recruited. The plantar pressure with gait parameters, postural balance parameters, Modified Ashworth Scale, and Modified Tardieu Scale were evaluated before, 3 weeks and 3 months after Botulinum toxin A (BTxA) injection. Spasticity of hemiplegic upper extremity before, and after BTxA injection were significantly changed. Plantar pressure overload in affected side was reduced after BTxA injection. The mean X-speed and the horizontal distance decreased in postural balance analysis with eyes-opened test. Improvement in hemiplegic upper extremity spasticity showed positive correlation with gait parameters. In addition, improvement in hemiplegic upper extremity spasticity was positively correlated with change in balance parameters in postural balance analysis with eyes-closed and dynamic tests. This study focused on the effect of stroke patient's hemiplegic upper extremity spasticity on their gait and balance parameters and identified that the BTxA injection on hemiplegic patient's spastic upper extremity improve postural balance and gait function.

Use of Botulinum Toxin as a Treatment of Hemiplegic Shoulder Pain Syndrome: A Randomized Trial.

OBJECTIVE: The primary objective of this paper is to assess whether the use of 200 units of abobotulinum in the pectoralis major and subscapularis muscles modifies the pain complaint assessed using the visual analog scale in subjects with shoulder pain after the onset of spastic hemiplegia due to cerebrovascular disease when compared to the application of a placebo to the same muscles. DESIGN: A prospective, double-blind, randomized, and placebo-controlled clinical trial study in two different rehabilitation centers. SETTING: Two distinct outpatient neurological rehabilitation services. PARTICIPANTS: Patients older than 18 years who were included presented upper limb spasticity resulting from ischemic or hemorrhagic stroke and a diagnosis of Painful Hemiplegic Shoulder Syndrome (PHSS) that was independent of motor dominance. INTERVENTIONS: Patients were divided into two groups, one of them underwent the application of botulinum toxin (TXB-A) in the pectoralis major and subscapularis muscles, at a total dose of 400 U. MAIN OUTCOME MEASURE: Patients were assessed for a change in pain using the Visual Analog Scale (VAS) for at least 13 mm. RESULTS: An improvement in pain and spasticity levels in both groups, more intense in the toxin group, but without statistical significance. The comparison between the groups showed a reduction in pain by VAS (p = 0.52). CONCLUSIONS: The use of botulinum toxin in the subscapularis and pectoralis major muscles resulted in a reduction in shoulder pain in spastic hemiplegic patients without statistical significance.

[Alternating Hemiplegia of Childhood associated with a pathogenic variant of the ATP1A3 gene]. / Hemiplejía Alternante de la Infancia asociada a variante patogénica del gen ATP1A3.

INTRODUCTION: Alternating hemiplegia of childhood (AHC), is a rare disease characterized by episodes of hemi/quadriplegia, dystonic postures, abnormal eye movements, and movement disorders. ATP1A3 gene mu tations are the most frequently associated with AHC. OBJECTIVE: To present a clinical case of AHC, where genetic study and the observation of home videos were of great diagnostic utility. CLINICAL CASE: Female patient who at 3 months of age presented with several episodes of dystonic postures, clonic movements of extremities, cephalic version, and lateral gaze deviation lasting several minutes. Epilepsy was diagnosed and levetiracetam was administrated, without improvement. EEG and brain MRI were performed, with normal results. Therefore, epilepsy was ruled out and transient dystonia of infancy was suspected, however, the events became more frequent, longer in duration, and charac teristically subsided during sleep. Family members provided home videos that clarified the events. At 6 months of age, the patient presented with alternating hemiparesis. Dystonia genetic panel showed a pathogenic variant of the ATP1A3 gene, confirming the diagnosis. Flunarizine treatment was ini tiated with a good clinical response at 12 months of follow-up. CONCLUSIONS: The diagnosis of AHC is complex and is frequently confused with epilepsy, so it is important to correctly perform the diffe rential diagnosis, including anamnesis, tests such as EEG, and careful observation of clinical events that, with the current access to audiovisual technology, becomes more accurate. The genetic analysis is a great diagnostic tool that, when performed in time, avoids other unnecessary tests and therapies.

Comprehensive Assessment of the Time Course of Biomechanical, Electrophysiological and Neuro-Motor Effects after Botulinum Toxin Injections in Elbow Flexors of Chronic Stroke Survivors with Spastic Hemiplegia: A Cross Sectional Observation Study.

Botulinum neurotoxin (BoNT) is commonly used to manage focal spasticity in stroke survivors. This study aimed to a perform comprehensive assessment of the effects of BoNT injection. Twelve stroke subjects with spastic hemiplegia (age: 52.0 ± 10.1 year; 5 females) received 100 units of BoNT to the spastic biceps brachii muscles. Clinical, biomechanical, electrophysiological, and neuro-motor assessments were performed one week (wk) before (pre-injection), 3 weeks (wks) after, and 3 months (mons) after BoNT injection. BoNT injection significantly reduced spasticity, muscle strength, reflex torque, and compound muscle action potential (CMAP) amplitude of spastic elbow flexors (all p < 0.05) during the 3-wks visit, and these values return to the pre-injection level during the 3-mons visit. Furthermore, the degree of reflex torque change was negatively correlated to the amount of non-reflex component of elbow flexor resistance torque. However, voluntary force control and non-reflex resistance torque remained unchanged throughout. Our results revealed parallel changes in clinical, neurophysiological and biomechanical assessment after BoNT injection; BoNT injection would be more effective if hypertonia was mainly mediated by underlying neural mechanisms. BoNT did not affect voluntary force control of spastic muscles.

Capacity for Hand-as-a-Holder Function of the Spastic Combination Hand: A Proof-of-Concept Study.

ABSTRACT: We call a hemiparetic hand with paralyzed finger extensors and volitional but spastic flexors a "spastic combination hand." Anecdotally, patients report hand-as-a-holder function with objects like pill bottles, motivating us to formally study spastic combination hand holding capacity using cylinders with different diameters. Nine participants with spastic hemiparesis and spastic combination hand more than 24 mos performed a standardized motor task with 10 cylinder diameters ranging between 1.3 and 9.5 cm and weighing 0.8 and 8.4 oz. Using the unaffected hand, participants attempted to insert a given cylinder into their spastic combination hand, holding it independently for 5 secs. Successful holds were counted during two sessions before and one session after botulinum toxin intervention of Ashworth 3 hand muscles. Findings revealed that a median capacity of six different cylinder diameters was successfully inserted into spastic combination hand at least once before block and a median capacity of 10 cylinders was inserted after block. A mixed-effect statistical model using fixed effects of cylinder diameter and session revealed that total number of successful holds was 43% higher after botulinum. We conclude that this proof-of-concept study does support the idea that spastic combination hand has holding capacity for cylindrical objects of specified diameter and weight and that botulinum neurotoxin offers potential for enlarging spastic combination hand capacity.

Effects of acupoint injection for stroke patients with hemiplegia: A protocol for systematic review and meta-analysis of randomized controlled trials.

BACKGROUND: Acupoint injection has currently received increasing attention as a treatment for hemiplegia. A number of studies have reported that acupoint injection have some advantages in treatment of hemiplegia. However, currently no article has summarized the existing evidence. Our study will evaluate the efficacy and safety of acupoint injection as a clinical treatment for hemiplegia, so that it can provide an important reference for clinical decision-making. METHODS: Randomized controlled trials and case control studies of acupoint injection for hemiplegia according to the included and excluded standard were identified in searches of 6 databases from their inception to February 2021. All data were assessed and extracted by 2 authors independently. The risk of bias assessment recommended by the Cochrane Collaboration was used to assess the quality of the selected studies. Review Manager 5.4 (Cochrane Collaboration) was used to conduct meta-analysis for the efficacy and safety of acupoint injection. RESULT: The results of this systemic review and meta-analysis will be submitted to a recognized journal for publication. CONCLUSION: This systemic review and meta-analysis will evaluate the efficacy and safety of acupoint injection as a clinical treatment for hemiplegia. We hope this study can make a definitive conclusion for acupoint injection in the treatment of hemiplegia. REGISTRATION: PROSPERO (registration number CRD42021234453).

Primary pyomyositis in an infant following one-day afebrile upper limb monoplegia.

Background : Pyomyositis is a subacute bacterial infection of the skeletal muscles. Its most common features are fever, muscle swelling, and focal pain. There have been insufficient data regarding pyomyositis in healthy infants. Case report : A one-month-old boy presented with an impairment of his left arm movement. He was well-nourished and not under any apparent distress. His vital signs were within the normal limits but neurological examination revealed left forearm paralysis. Physical examination showed no abnormal findings in the region from the left shoulder joint to the fingertips. Considering these factors, an intracranial pathology was initially suspected. However, he developed a fever, regular tachycardia, and swelling in the left forearm. Magnetic resonance imaging revealed inflammation in the left forearm muscles. He was diagnosed with bacterial myositis and started on intravenous antibiotics. On the 17th day, he was discharged with oral antibiotic treatment, which was completed over 25 days without any sequelae nor relapse. Conclusion : Here we report the case of Japanese primary pyomyositis following one-day afebrile upper limb monoplegia in an infant. Even when infants exhibit afebrile symptoms, a bacterial infection should be suspected. J. Med. Invest. 68 : 372-375, August, 2021.

The effectiveness of botulinum toxin on spasticity and gait of hemiplegic patients after stroke: A systematic review and meta-analysis.

The aim of the study is to evaluate the evidence supporting the efficacy of botulinum toxin type A (BTA) injections in lower limb of hemiplegic patients, after stroke or cerebrovascular accident, and their gait analysis. This study included: randomized controlled trials (RCTs), non-randomized or controlled clinical trials (CCTs) or cluster trials, clinical trials of various phases (I-III), interrupted time series (ITS) studies with at least three data points before and after the intervention, controlled before and after (CBA) studies, prospective and retrospective comparative cohort studies, case-control and multicentred studies. The patients included in these studies had similar characteristics: age over 18 years, history of stroke and following hemiplegia, minimum modified Ashworth scale (MAS) score of 2 and duration since stroke over 6 months. The number of studies included in this review was 21. A meta-analysis was performed on a fraction of them depending on the reported index and the methodology as reported in detail in the results section. MAS score, following BTA injections, was significantly improved (Hedges' g: -1.17; 95% CI: -1.66, 0.67; p < 0.001). The same applied for the 10 Meter Walk Test (MWT) (-0.35; 95% CI: -0.68, -0.02; p = 0.016). The gait velocity showed improvement, yet without statistical significance (0.27; 95% CI: -0.09, 0.63; p = 0.285). We concluded that botulinum toxin injections showed effectiveness on lower limb hypertonia reduction of hemiplegic patients after stroke. Apart from significantly reducing the MAS scores, 10 MWT was also improved. However, more research is required in order to determine the advancement in specific gait and posture parameters.

Comparison of the effectiveness of pulsed radiofrequency of the suprascapular nerve and intra-articular corticosteroid injection for hemiplegic shoulder pain management.

Many patients complain of hemiplegic shoulder pain following stroke. Here, the effectiveness of pulsed radiofrequency stimulation of the suprascapular nerve is compared with intra-articular corticosteroid injection for chronic hemiplegic shoulder pain following stroke. This single-center, prospective, randomized controlled study included 20 patients with hemiplegic shoulder pain after stroke, randomly assigned to the pulsed radiofrequency and intra-articular corticosteroid injection treatment groups (n = 10 in each). Hemiplegic shoulder pain severity was measured by numeric rating scale and passive shoulder range motion was assessed at baseline and one and two months after each procedure. Compared to the baseline numeric rating scale scores, post-treatment scores decreased significantly in both groups (p < 0.001). However, score reduction through time was significantly greater for intra-articular corticosteroid injection for pulsed radiofrequency (p < 0.001). Similarly, a significant post-treatment increase was observed in almost all range of motion measurements in both groups (pulsed radiofrequency group: flexion, p = 0.015; abduction, p = 0.014; external rotation, p = 0.038; internal rotation, p = 0.063; intra-articular corticosteroid injection group: all range of motion, p < 0.001). Moreover, the measurements for all ranges of motion in the intra-articular corticosteroid injection group were significantly higher than those in the pulsed radiofrequency group (p < 0.001). Thus, intra-articular corticosteroid injection appears more effective than pulsed radiofrequency for control of hemiplegic shoulder pain, whereas, pulsed radiofrequency of the suprascapular nerve has minimal effect. However, in patients at risk for developing complications following corticosteroid injections, pulsed radiofrequency of the suprascapular nerve may be an option in management of hemiplegic shoulder pain.

Ultrasound-Guided BoNT-A (Botulinum Toxin A) Injection Into the Subscapularis for Hemiplegic Shoulder Pain: A Randomized, Double-Blind, Placebo-Controlled Trial.

BACKGROUND AND PURPOSE: This study aimed to assess the efficacy of an ultrasound-guided lateral approach for BoNT-A (botulinum toxin A) injections into the subscapularis in patients with hemiplegic shoulder pain. METHODS: This single-center trial used a randomized, double-blind, placebo-controlled design. The key inclusion criteria were a visual analogue scale score of ≥4 cm and a modified Ashworth scale score of ≥1+. The patients were randomized to receive either BoNT-A injections or a placebo. The outcomes included the visual analogue scale score, modified Ashworth scale score, pain-free passive range of motion of the hemiplegic shoulder, Fugl-Meyer assessment score for the upper extremities, and Stroke-Specific Quality-of-Life score. RESULTS: A total of 49 hemiplegic shoulder pain patients were screened, and 36 were included. The participants receiving the BoNT-A injection reported a significant decrease in pain (visual analogue scale, -1.39 [95% CI, -2.41 to -0.36]; P=0.002) and spasticity (modified Ashworth scale score for shoulder internal rotation, -0.72 [95% CI, -1.10 to -0.35]; P=0.001; modified Ashworth scale score for shoulder abduction, -0.44 [95% CI, -0.90 to -0.01]; P=0.026) and improved pain-free passive shoulder internal rotation range of motion (14.56 [95% CI, 6.70-21.41]; P<0.001) and quality of life (Stroke-Specific Quality-of-Life upper extremity subscale, P=0.025) compared with those receiving the placebo at the end point. The shoulder abduction range of motion did not significantly improve after the BoNT-A injection at the end point (P=0.127). In addition, the patients in the BoNT-A group showed significant improvements in the visual analogue scale score and shoulder external rotation range of motion at the 12-week follow-up. No injection-related adverse events were observed during or after the interventions in either group. CONCLUSIONS: The ultrasound-guided lateral approach for BoNT-A injections into the subscapularis is a precise and reliable method for reducing pain and spasticity and improving quality of life in stroke survivors with hemiplegic shoulder pain. Registration: URL: https://www.chictr.org.cn; Unique identifier: ChiCTR1900023513.

Infant with right hemiplegia due to acute encephalopathy with biphasic seizures and late reduced diffusion (AESD): A case report.

RATIONALE: Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a condition characterized by biphasic convulsions and disturbance of consciousness. In Japan, the most common pediatric cases of acute encephalopathy are associated with infection. AESD usually occurs in early childhood, with the characteristic magnetic resonance imaging (MRI) appearance called "bright tree appearance." The disease often has neurological sequelae and interferes with the schooling of children and their activities of daily living; however, there are few clinical case reports of hemiplegia caused by AESD. PATIENT CONCERNS: A case with right-sided hemiplegia due to AESD in an 11-month-old girl who was followed up to 30 mo of age. DIAGNOSES: The patient was diagnosed with overlap AESD and hemiconvulsion-hemiplegia-epilepsy syndrome (HHE syndrome), based on the clinical course and imaging findings. DNA tests of her blood and cerebrospinal fluid revealed the presence of human herpesvirus 6. INTERVENTIONS: Pharmacotherapy and rehabilitation therapy. OUTCOME: Gross motor function has recovered considerably, but she had a mild developmental delay at 30 mo old. LESSONS: Hemiplegia due to AESD was extremely rare, and appropriate rehabilitation treatment resulted in recovery of physical function. However, as mild developmental delay was observed, the patient was referred to a specialized facility before entering school.

Changes in Muscle Mass after Botulinum Toxin Injection in Children with Spastic Hemiplegic Cerebral Palsy.

We aimed to evaluate muscle mass changes after injection of botulinum toxin (BoNT) in children with spastic hemiplegic cerebral palsy (CP). Children aged between 2 and 12 years who were diagnosed with hemiplegic CP with spastic equinus foot were prospectively recruited and administered BoNT in the affected leg. Lean body mass (LBM) of both legs and total limbs was measured by dual-energy X-ray absorptiometry (DXA) preinjection and 4 and 12 weeks after injection. A total of 15 children were enrolled into the study. LBM of both legs and total limbs increased significantly over 12 weeks of growth. The ratio of LBM of the affected leg to total limbs and to the unaffected leg significantly reduced at 4 weeks after injection compared with preinjection but significantly increased at 12 weeks after injection compared with 4 weeks after injection. In conclusion, the muscle mass of the affected leg after BoNT injection in children with hemiplegic spastic CP decreased at 4 weeks after BoNT injection but significantly recovered after 12 weeks after injection.

Pituitary tumor apoplexy associated with extrapontine myelinolysis during pregnancy: A case report.

RATIONALE: Pituitary tumor apoplexy (PTA) is a rare clinical syndrome which requires urgent diagnosis and treatment due to its life-threatening consequences. Management of undiagnosed pituitary tumor before pregnancy is a problem during pregnancy. PATIENT CONCERNS: We reported a case with PTA which was not diagnosed before pregnancy presenting with vomiting associated with hyponatremia during the third trimester. After supplying the sodium the patient presented with dysarthria and hemiplegia. DIAGNOSES: MRI examination showed PTA accompanied with extrapontine myelinolysis (EPM). INTERVENTIONS: The patient was given hydrocortisone according to the symptoms gradually to taper off dose, at the same times oral levothyroxine therapy (25µg/day) was given. OUTCOMES: The patient delivered a healthy baby via cesarean section at hospital at 38 + 1 week of gestation. We performed MRI examination regularly and the tumor regressed significantly 8 months postpartum. LESSONS: We reported a case as PTA associated with EPM. Headache during pregnancy is often nonspecific, so careful medical history inquiry is very important.

Effectiveness of Botulinum Toxin A in Treatment of Hemiplegic Shoulder Pain: A Systematic Review and Meta-analysis.

OBJECTIVE: To evaluate the effectiveness of botulinum toxin A (BTX-A) in the treatment of hemiplegic shoulder pain. DATA SOURCES: PubMed, EMBASE, Elsevier, Springer, Cochrane Library, Physiotherapy Evidence Database, CNKI, and VIP were researched from the earliest records to September 1, 2020. STUDY SELECTION: Randomized controlled trials that compared shoulder BTX-A injections vs a control intervention in patients with a history of hemiplegic shoulder pain after stroke were selected. Among the 620 records screened, 9 trials with 301 eligible patients were included. DATA EXTRACTION: Outcome data were pooled according to follow-up intervals (1, 2, 4, and 12 wk). The primary evaluation indices were pain reduction (visual analog scale [VAS] score) and range of motion (ROM) improvement. The second evaluation indices were upper limb functional improvement, spasticity improvement, and incidence of adverse events. Cochrane risk-of-bias was used to assess the methodological quality of studies independently by 2 evaluators. DATA SYNTHESIS: Meta-analysis revealed a statistically significant decrease in the VAS score in the BTX group vs the control group at 1, 4, and 12 weeks postinjection (wk 1: standardized mean difference [SMD], 0.91; 95% confidence interval [CI], 0.27 to 1.54; wk 4: SMD, 1.63; 95% CI, 0.76 to 2.51; wk 12: SMD, 1.96; 95% CI, 1.44 to 2.47). Furthermore, the meta-analysis demonstrated a statistically significant increase in abduction at 1, 4, and 12 weeks postinjection (wk 1: SMD, 3.71; 95% CI, 0 to 7.41; wk 4: SMD, 8.8; 95% CI, 2.22 to 15.37; wk 12: SMD, 19.59; 95% CI, 9.05 to 30.13) and external rotation at 1, 2, 4 weeks postinjection (wk 1: SMD, 5.67; 95% CI, 0.88 to 10.47; wk 2: SMD, 9.62; 95% CI, 5.57 to 13; wk 4: SMD, 6.89; 95% CI, 2.45 to 11.33) in the BTX group. CONCLUSIONS: BTX-A injection provided greater analgesic effects and increased shoulder abduction and external rotation ROM compared with steroid or placebo injection for the treatment of HSP.

Intravenous Immunoglobulin in the Treatment of Alternating Hemiplegia of Childhood.

ABSTRACT: Alternating hemiplegia of childhood (AHC) is a neurological disorder with early-onset alternating hemiplegia and other paroxysmal events such as epilepsy and dystonia due to de novo pathogenic mutations in the ATP1A3. Physicians and scientists investigated several agents in the treatment without strong evidence of definitive long-term benefit. Knowledge regarding utility of anti-inflammatory agents in the treatment is scarce except the anecdotal report of corticosteroid use. We described 2 patients with AHC who were exposed to intravenous immunoglobulin (IVIG) treatment because of an alternative diagnosis. An 8-year-old girl received 4 years of periodic IVIG infusion and was free of paroxysmal events during the first 16 months of therapy. A 2-year-old boy received IVIG infusion for 10 months and remained seizure-free for 2 years since the beginning of the treatment, but without a definite change in hemiplegic episodes. Our report is the first description of IVIG use in patients with AHC. Although these patients did not achieve complete remission, partial responsiveness was noted. Although the definite role of IVIG in the treatment of AHC cannot be extrapolated from our study, further prospective controlled studies should be done because of the recent discovery of the connection between an anti-inflammatory hormone (Oubain) and Na+/K+ ATPase.