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Internal Medicine (Tokyo, Japan) 2015A 21-year-old man twice developed sudden dyspnea during running examinations for promotion. Chest computed tomography (CT) revealed lobular ground-glass opacity and...
A 21-year-old man twice developed sudden dyspnea during running examinations for promotion. Chest computed tomography (CT) revealed lobular ground-glass opacity and centrilobular shadows bilaterally. The bronchoscopy findings were consistent with alveolar hemorrhage, and echocardiography showed severe hypokinesia a few hours later. A mass was subsequently identified along the abdominal aorta on enhanced CT and iodine-131 metaiodobenzylguanidine scintigraphy. Catecholamine oversecretion, which was later confirmed, may have increased the pulmonary vein pressure, thus resulting in the development of an alveolar hemorrhage, and damaged the cardiac muscles as manifested by hypokinesia. Since the patient had not previously developed alveolar hemorrhage during usual exercise, emotional stress superimposed on the physical activity may have triggered the paraganglioma to secrete excessive catecholamines, thus resulting in the observed symptoms.
Topics: Aorta, Abdominal; Bronchoscopy; Catecholamines; Exercise Test; Hemorrhage; Humans; Lung Diseases; Male; Paraganglioma; Tomography, X-Ray Computed; Young Adult
PubMed: 25758075
DOI: 10.2169/internalmedicine.54.2304 -
Journal of Parkinson's Disease 2020In postmortem analysis of late stage Parkinson's disease (PD) neuronal loss in the substantial nigra (SN) correlates with the antemortem severity of bradykinesia and...
BACKGROUND
In postmortem analysis of late stage Parkinson's disease (PD) neuronal loss in the substantial nigra (SN) correlates with the antemortem severity of bradykinesia and rigidity, but not tremor.
OBJECTIVE
To investigate the relationship between midbrain nuclei volume as an in vivo biomarker for surviving neurons in mild-to-moderate patients using 7.0 Tesla MRI.
METHODS
We performed ultra-high resolution quantitative susceptibility mapping (QSM) on the midbrain in 32 PD participants with less than 10 years duration and 8 healthy controls. Following blinded manual segmentation, the individual volumes of the SN, subthalamic nucleus, and red nucleus were measured. We then determined the associations between the midbrain nuclei and clinical metrics (age, disease duration, MDS-UPDRS motor score, and subscores for bradykinesia/rigidity, tremor, and postural instability/gait difficulty).
RESULTS
We found that smaller SN correlated with longer disease duration (r = -0.49, p = 0.004), more severe MDS-UPDRS motor score (r = -0.42, p = 0.016), and more severe bradykinesia-rigidity subscore (r = -0.47, p = 0.007), but not tremor or postural instability/gait difficulty subscores. In a hemi-body analysis, bradykinesia-rigidity severity only correlated with SN contralateral to the less-affected hemi-body, and not contralateral to the more-affected hemi-body, possibly reflecting the greatest change in dopamine neuron loss early in disease. Multivariate generalized estimating equation model confirmed that bradykinesia-rigidity severity, age, and disease duration, but not tremor severity, predicted SN volume.
CONCLUSIONS
In mild-to-moderate PD, SN volume relates to motor manifestations in a motor domain-specific and laterality-dependent manner. Non-invasive in vivo 7.0 Tesla QSM may serve as a biomarker in longitudinal studies of SN atrophy and in studies of people at risk for developing PD.
Topics: Aged; Autopsy; Female; Gait Disorders, Neurologic; Humans; Hypokinesia; Magnetic Resonance Imaging; Male; Middle Aged; Muscle Rigidity; Parkinson Disease; Postural Balance; Red Nucleus; Severity of Illness Index; Substantia Nigra; Subthalamic Nucleus; Time Factors; Tremor
PubMed: 32250317
DOI: 10.3233/JPD-191890 -
Clinical Parkinsonism & Related... 2022Bradykinesia in Parkinson's disease is a marker for clinical levodopa responsiveness, with persistent bradykinesia reflecting suboptimal response. We objectively...
INTRODUCTION
Bradykinesia in Parkinson's disease is a marker for clinical levodopa responsiveness, with persistent bradykinesia reflecting suboptimal response. We objectively measured prevalence and severity of morning bradykinesia using the Personal KinetiGraph® (PKG®).
METHODS
Retrospective evaluation of a large global database of de-identified PKG assessments from individuals (N=12,840) in routine clinical care in the United States (US; n=3288). Median bradykinesia scores (mBKS) and median dyskinesia scores (mDKS) were calculated using a validated algorithm and previously established targets to evaluate percent time in bradykinesia, levodopa responsiveness, and prevalence and severity (0-5; 5=highest severity) of morning bradykinesia.
RESULTS
mBKS was above target (≥26) in 65% of all individuals, and mDKS was above target (≥7) in 3%. Elevated percent time in bradykinesia occurred in 79%. Among individuals where levodopa responsiveness could be evaluated (n=1933), 31% had a significant response (≥1.15 postdose decrease in severity). Morning bradykinesia was identified in 85% of individuals with available morning data (1298/1524), and 64% (954/1501) experienced continued bradykinesia after the first daily levodopa dose. Morning bradykinesia was severe (4.0-4.7) in levodopa-responsive individuals regardless of percent time spent in bradykinesia.
CONCLUSION
Elevated mBKS was very common in the US. Most individuals taking levodopa had morning bradykinesia that persisted even after the first daily dose, and severity was high, indicating a need for additional treatment options.
PubMed: 35620251
DOI: 10.1016/j.prdoa.2022.100145 -
Journal of Neuroengineering and... Jul 2021Fluctuations in motor function in Parkinson's Disease (PD) are frequent and cause significant disability. Frequently device assisted therapies are required to treat...
BACKGROUND
Fluctuations in motor function in Parkinson's Disease (PD) are frequent and cause significant disability. Frequently device assisted therapies are required to treat them. Currently, fluctuations are self-reported through diaries and history yet frequently people with PD do not accurately identify and report fluctuations. As the management of fluctuations and the outcomes of many clinical trials depend on accurately measuring fluctuations a means of objectively measuring time spent with bradykinesia or dyskinesia would be important. The aim of this study was to present a system that uses wearable sensors to measure the percentage of time that bradykinesia or dyskinesia scores are above a target as a means for assessing levels of treatment and fluctuations in PD.
METHODS
Data in a database of 228 people with Parkinson's Disease and 157 control subjects, who had worn the Parkinson's Kinetigraph ((PKG, Global Kinetics Corporation™, Australia) and scores from the Unified Parkinson's Disease Rating Scale (UPDRS) and other clinic scales were used. The PKG's provided score for bradykinesia and dyskinesia every two minutes and these were compared to a previously established target range representing a UPDRS III score of 35. The proportion of these scores above target over the 6 days that the PKG was worn were used to derive the percent time in bradykinesia (PTB) and percent time in dyskinesia (PTD). As well, a previously describe algorithm for estimating the amplitude of the levodopa response was used to determine whether a subject was a fluctuator or non-fluctuator.
RESULTS
Using this approach, a normal range of PTB and PTD based on Control subject was developed. The level of PTB and PTD experienced by people with PD was compared with their levels of fluctuation. There was a correlation (Pearson's ρ = 0.4) between UPDRS II scores and PTB: the correlation between Parkinson Disease Questionnaire scores and UPDRS Total scores and PTB and slightly lower. PTB and PTD fell in response to treatment for bradykinesia or dyskinesia (respectively) with greater sensitivity than clinical scales.
CONCLUSIONS
This approach provides an objective assessment of the severity of fluctuations in Parkinson's Disease that could be used in in clinical trials and routine care.
Topics: Algorithms; Antiparkinson Agents; Dyskinesias; Humans; Hypokinesia; Levodopa; Parkinson Disease
PubMed: 34271971
DOI: 10.1186/s12984-021-00905-4 -
Brain : a Journal of Neurology Mar 2024Dopaminergic dysfunction in the basal ganglia, particularly in the posterior putamen, is often viewed as the primary pathological mechanism behind motor slowing (i.e....
Dopaminergic dysfunction in the basal ganglia, particularly in the posterior putamen, is often viewed as the primary pathological mechanism behind motor slowing (i.e. bradykinesia) in Parkinson's disease. However, striatal dopamine loss fails to account for interindividual differences in motor phenotype and rate of decline, implying that the expression of motor symptoms depends on additional mechanisms, some of which may be compensatory in nature. Building on observations of increased motor-related activity in the parieto-premotor cortex of Parkinson patients, we tested the hypothesis that interindividual differences in clinical severity are determined by compensatory cortical mechanisms and not just by basal ganglia dysfunction. Using functional MRI, we measured variability in motor- and selection-related brain activity during a visuomotor task in 353 patients with Parkinson's disease (≤5 years disease duration) and 60 healthy controls. In this task, we manipulated action selection demand by varying the number of possible actions that individuals could choose from. Clinical variability was characterized in two ways. First, patients were categorized into three previously validated, discrete clinical subtypes that are hypothesized to reflect distinct routes of α-synuclein propagation: diffuse-malignant (n = 42), intermediate (n = 128) or mild motor-predominant (n = 150). Second, we used the scores of bradykinesia severity and cognitive performance across the entire sample as continuous measures. Patients showed motor slowing (longer response times) and reduced motor-related activity in the basal ganglia compared with controls. However, basal ganglia activity did not differ between clinical subtypes and was not associated with clinical scores. This indicates a limited role for striatal dysfunction in shaping interindividual differences in clinical severity. Consistent with our hypothesis, we observed enhanced action selection-related activity in the parieto-premotor cortex of patients with a mild-motor predominant subtype, both compared to patients with a diffuse-malignant subtype and controls. Furthermore, increased parieto-premotor activity was related to lower bradykinesia severity and better cognitive performance, which points to a compensatory role. We conclude that parieto-premotor compensation, rather than basal ganglia dysfunction, shapes interindividual variability in symptom severity in Parkinson's disease. Future interventions may focus on maintaining and enhancing compensatory cortical mechanisms, rather than only attempting to normalize basal ganglia dysfunction.
Topics: Humans; Parkinson Disease; Hypokinesia; Basal Ganglia; Corpus Striatum; Dopamine; Putamen
PubMed: 37757883
DOI: 10.1093/brain/awad325 -
Frontiers in Psychiatry 2022Movement disorders have been suggested to be a cardinal component of schizophrenia. With increased research interests in this area, instrumental measures are needed....
INTRODUCTION
Movement disorders have been suggested to be a cardinal component of schizophrenia. With increased research interests in this area, instrumental measures are needed. This study was to examine if the motion capture system was reliable in measuring hand and facial bradykinesia and dyskinesia and more sensitive to detecting movement differences between schizophrenia patients and healthy people than traditional rating scales.
METHODS
Sixteen schizophrenia patients and 20 control subjects were recruited. Hand and facial bradykinesia and dyskinesia were measured using the motion capture system and rated using the Extrapyramidal Symptom Rating Scale and the Abnormal Involuntary Movement Scale.
RESULTS
The system showed strong test-retest reliability and generated larger effect sizes of group differences than did the rating scales.
CONCLUSIONS
The results may support researchers and clinical practitioners to apply the system to sensitively measuring the hand and facial movement symptoms in schizophrenia patients, which contributes to gaining a deep understanding of movement issues in schizophrenia.
PubMed: 35308887
DOI: 10.3389/fpsyt.2022.803661 -
Frontiers in Aging Neuroscience 2022The aim was to examine the role of sensor measurement in identifying and managing fluctuations in bradykinesia of Parkinson's Disease.
OBJECTIVES
The aim was to examine the role of sensor measurement in identifying and managing fluctuations in bradykinesia of Parkinson's Disease.
METHOD
Clinical scales and data from wearable sensors obtained before and after optimization of treatment from 107 participants who participated in a previous study was used. Fluctuators were identified by a levodopa response or wearing off in their sensor data and were subdivided according to whether the sensor's bradykinesia scores were in target range, representing acceptable bradykinesia for part of the dose (Controlled Fluctuator: = 22) or above target for the whole dose period (Uncontrolled Fluctuator; = 28). Uncontrolled Non-fluctuators ( = 24) were cases without a levodopa response or wearing-off and sensor bradykinesia scores above target throughout the day (un-controlled). Controlled Non-fluctuators ( = 33) were below target throughout the day (controlled) and used as a reference for good control (MDS-UPDRS III = 33 ± 8.6 and PDQ39 = 28 ± 18).
RESULTS
Treating Fluctuators significantly improved motor and quality of life scores. Converting fluctuators into Controlled Non-fluctuators significantly improved motor, non-motor and quality of life scores and a similar but less significant improvement was obtained by conversion to a Controlled Fluctuator. There was a significantly greater likelihood of achieving these changes when objective measurement was used to guide management.
CONCLUSIONS
The sensor's classification of fluctuators bore a relation to severity of clinical scores and treatment of fluctuation improved clinical scores. The sensor measurement aided in recognizing and removing fluctuations with treatment and resulted in better clinical scores, presumably by assisting therapeutic decisions.
PubMed: 35401155
DOI: 10.3389/fnagi.2022.852992 -
Neurology May 2022Relative to the pediatric population, there is limited information about Dravet syndrome (DS) in adults. In addition to some of the gait abnormalities reported in...
BACKGROUND AND OBJECTIVES
Relative to the pediatric population, there is limited information about Dravet syndrome (DS) in adults. In addition to some of the gait abnormalities reported in children with DS (such as crouch gait and ataxia), adults with this condition have other gait and motor disturbances. Our primary objective was to examine gait and motor manifestations in adults with DS.
METHODS
This study includes a prospective arm where 6 patients (mean age, 32 years) were examined through a modified version of the Unified Parkinson's Disease Rating Scale (mUPDRS) in 2014 and again in 2019. mUPDRS scores were assigned to gait, resting tremors, facial expression, arising from a chair, posture, and body bradykinesia. The cross-sectional arm includes mUPDRS testing in patients who were not evaluated in 2014 and an instrumental gait analysis (IGA). These cross-sectional tests were done in the 2019-2020 period. The IGA was performed using ProtoKinetics software with a gait mat built with sensors and 2 cameras capturing the sagittal and coronal planes. The IGA was performed in a group of 17 patients with DS (mean age, 31 years); the control group consisted of 81 healthy individuals, whose mean age was 62 years. Regression analyses were performed for the IGA and mUPDRS data.
RESULTS
Five out of 6 participants evaluated prospectively over 5 years experienced worsening of their parkinsonian manifestations, including gait. Two patients (47 and 51 years of age) who were initially ambulatory could no longer walk 5 years later. The cross-sectional analysis of mUPDRS in a larger group of adults showed that worse scores for arising from a chair ( = 0.04), body bradykinesia ( = 0.01), and gait ( = 0.0003) were positively associated with age. The IGA cross-sectional arm revealed that all 17 adults with DS had abnormal gait measures in all domains tested. This group of patients performed worse than the healthy and older control group.
DISCUSSION
Although seizures may decrease in older adults with DS, this prospective and cross-sectional study showed that their motor symptoms and gait become progressively worse as they age.
Topics: Adult; Aged; Child; Child, Preschool; Cross-Sectional Studies; Epilepsies, Myoclonic; Epileptic Syndromes; Gait; Humans; Hypokinesia; Immunoglobulin A; Middle Aged; Prospective Studies; Spasms, Infantile
PubMed: 35418450
DOI: 10.1212/WNL.0000000000200341 -
Monitoring Motor Symptoms During Activities of Daily Living in Individuals With Parkinson's Disease.Frontiers in Neurology 2018This literature review addressed wearable sensor systems to monitor motor symptoms in individuals with Parkinson's disease (PD) during activities of daily living (ADLs).... (Review)
Review
This literature review addressed wearable sensor systems to monitor motor symptoms in individuals with Parkinson's disease (PD) during activities of daily living (ADLs). Specifically, progress in monitoring tremor, freezing of gait, dyskinesia, bradykinesia, and hypokinesia was reviewed. Twenty-seven studies were found that met the criteria of measuring symptoms in a home or home-like setting, with some studies examining multiple motor disorders. Accelerometers, gyroscopes, and electromyography sensors were included, with some studies using more than one type of sensor. Five studies measured tremor, five studies examined bradykinesia or hypokinesia, thirteen studies included devices to measure dyskinesia or motor fluctuations, and ten studies measured akinesia or freezing of gait. Current sensor technology can detect the presence and severity of each of these symptoms; however, most systems require sensors on multiple body parts, which is challenging for remote or ecologically valid observation. Different symptoms are detected by different sensor placement, suggesting that the goal of detecting all symptoms with a reduced set of sensors may not be achievable. For the goal of monitoring motor symptoms during ADLs in a home setting, the measurement system should be simple to use, unobtrusive to the wearer and easy for an individual with PD to put on and take off. Machine learning algorithms such as neural networks appear to be the most promising way to detect symptoms using a small number of sensors. More work should be done validating the systems during unscripted and unconstrained ADLs rather than in scripted motions.
PubMed: 30619024
DOI: 10.3389/fneur.2018.01036 -
PloS One 2022Dysarthria may present during the natural course of many degenerative neurological conditions. Hypokinetic and ataxic dysarthria are common in movement disorders and...
Dysarthria may present during the natural course of many degenerative neurological conditions. Hypokinetic and ataxic dysarthria are common in movement disorders and represent the underlying neuropathology. We developed an artificial intelligence (AI) model to distinguish ataxic dysarthria and hypokinetic dysarthria from normal speech and differentiate ataxic and hypokinetic speech in parkinsonian diseases and cerebellar ataxia. We screened 804 perceptual speech analyses performed in the Samsung Medical Center Neurology Department between January 2017 and December 2020. The data of patients diagnosed with parkinsonian disorders or cerebellar ataxia were included. Two speech tasks (numbering from 1 to 50 and reading nine sentences) were analyzed. We adopted convolutional neural networks and developed a patch-wise wave splitting and integrating AI system for audio classification (PWSI-AI-AC) to differentiate between ataxic and hypokinetic speech. Of the 395 speech recordings for the reading task, 76, 112, and 207 were from normal, ataxic dysarthria, and hypokinetic dysarthria subjects, respectively. Of the 409 recordings of the numbering task, 82, 111, and 216 were from normal, ataxic dysarthria, and hypokinetic dysarthria subjects, respectively. The reading and numbering task recordings were classified with 5-fold cross-validation using PWSI-AI-AC as follows: hypokinetic dysarthria vs. others (area under the curve: 0.92 ± 0.01 and 0.92 ± 0.02), ataxia vs. others (0.93 ± 0.04 and 0.89 ± 0.02), hypokinetic dysarthria vs. ataxia (0.96 ± 0.02 and 0.95 ± 0.01), hypokinetic dysarthria vs. none (0.86 ± 0.03 and 0.87 ± 0.05), and ataxia vs. none (0.87 ± 0.07 and 0.87 ± 0.09), respectively. PWSI-AI-AC showed reliable performance in differentiating ataxic and hypokinetic dysarthria and effectively augmented data to classify the types even with limited training samples. The proposed fully automatic AI system outperforms neurology residents. Our model can provide effective guidelines for screening related diseases and differential diagnosis of neurodegenerative diseases.
Topics: Artificial Intelligence; Ataxia; Cerebellar Ataxia; Dysarthria; Humans; Hypokinesia; Neural Networks, Computer; Parkinsonian Disorders
PubMed: 35658000
DOI: 10.1371/journal.pone.0268337