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1.
Arch Argent Pediatr ; 119(6): e639-e642, 2021 12.
Artigo em Espanhol | MEDLINE | ID: mdl-34813247

RESUMO

Acrocyanosis triggered by standing position as the main sign of postural orthostatic tachycardia (POTS) is little known but well described in scientific literature. In pediatric age, POTS is defined as orthostatic intolerance that is accompanied by an excessive increase in heart rate without arterial hypotension. We present two clinical reports of teenagers who were admitted in the Emergency Department with acrocyanosis and orthostatic intolerance. The first patient was 13-year-old and had an increase in heart rate of 40 bpm when moving from a reclining to a standing position. The second patient was 14-year-old and showed an increasing in heart rate up to 125 bpm after upright position. In both patients' blood pressure was normal and all investigations were negative. They were finally diagnosed with Acrocianosis como primera manifestación de síndrome de taquicardia postural ortostática en dos adolescentes Acrocyanosis as the first manifestation of orthostatic postural tachycardia syndrome in two adolescents postural orthostatic tachycardia. Recognizing acrocyanosis as first sign of this disease is useful for diagnosis and can help to avoid unnecessary testing.


Assuntos
Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Adolescente , Pressão Sanguínea , Criança , Frequência Cardíaca , Humanos , Síndrome da Taquicardia Postural Ortostática/diagnóstico , Posição Ortostática
2.
J Clin Neurosci ; 92: 39-44, 2021 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-34509259

RESUMO

In patients with severe traumatic brain injury, there is limited evidence of the clinical effect of early orthostatic exercise, although such exercise may strengthen systemic or cerebral hemodynamic responses to head-up tilt, thereby minimizing orthostatic intolerance. We measured dynamic cerebral autoregulation (dCA) and the occurrence of orthostatic intolerance after four weeks of regular orthostatic exercise by head-up tilt using a tilt table with integrated stepping using the ERIGO® tilt-table and comparing it to standard care. Thirty-four patients with severe traumatic brain injury admitted to a neurocritical care unit were included in this randomized clinical trial. Middle cerebral artery blood flow velocity (MCAv), non-invasive mean arterial pressure, heart rate and PaCO2 were recorded; dCA was measured by the non-invasive mean flow index (nMxa). Transition from the supine position to head-up tilt triggered a 10-16% decrease in MCAv and increased nMxa in both groups at all time points (P < 0.05), with no differences between groups. There was no difference in the number of episodes with orthostatic intolerance (5 vs 3; 1 vs 2; 1 vs 0) at baseline, two weeks and four weeks, respectively, and no association between changes in PaCO2-adjusted nMxa and the occurrence of orthostatic reactions (P = 0.35). Early orthostatic exercise does not affect dynamic cerebral autoregulation and does not protect against orthostatic intolerance in patients with severe traumatic brain injury. Trial registration: ClinicalTrials.gov identifier: NCT02924649. Registered on 3rd October 2016.


Assuntos
Lesões Encefálicas Traumáticas , Hipotensão Ortostática , Intolerância Ortostática , Velocidade do Fluxo Sanguíneo , Pressão Sanguínea , Lesões Encefálicas Traumáticas/terapia , Circulação Cerebrovascular , Estudos de Viabilidade , Homeostase , Humanos
3.
Clin Obes ; 11(6): e12483, 2021 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-34409762

RESUMO

There have been increased reports of orthostatic intolerance post-bariatric surgery. However, the prevalence, pathophysiology and long-term outcomes have not been well described. Therefore, we sought to summarize evidence of orthostatic intolerance after bariatric surgery. We conducted a systematic review using PubMed, Scopus, CINAHL, Cochrane Database of Systematic Reviews and the Cochrane Central Register of Controlled Trials (CENTRAL) to identify relevant articles from the date of inception until 1st April 2020. Study selection, data extraction and quality assessment of the included studies were performed independently by two reviewers. The findings of the included studies were narratively reported. When feasible, a meta-analysis was done to summarize the relevant results. We included 20 studies (n = 19 843 participants) reporting findings of 12 prospective cohort studies, 5 retrospective cohort studies, 2 cross-sectional studies and one randomized controlled trial. The 5-year cumulative incidence of orthostatic intolerance was 4.2% (one study). Common clinical presentations of orthostatic intolerance were lightheadedness, dizziness, syncope and palpitation. The pooled data suggested improvement in overall cardiac autonomic function (sympathetic and parasympathetic) post-bariatric surgery. In addition, a significant systolic blood pressure drop may reflect a reset of the balance between the sympathetic and parasympathetic nervous systems after weight loss in the pooled analysis. Existing literature on orthostatic intolerance post-bariatric surgeries was limited or of low quality, and larger studies are needed to know the true incidence of orthostatic intolerance post-bariatric surgeries and the pathophysiology. We found one study reporting the 5-years cumulative incidence of orthostatic intolerance post-bariatric surgeries as only 4.2%. This could challenge the idea of increased orthostatic intolerance prevalence post-bariatric surgeries. Registration The review protocol was registered at the International Prospective Register of Systemic Reviews PROSPERO (CRD42020170877).


Assuntos
Cirurgia Bariátrica , Intolerância Ortostática , Cirurgia Bariátrica/efeitos adversos , Estudos Transversais , Humanos , Intolerância Ortostática/epidemiologia , Intolerância Ortostática/etiologia , Estudos Prospectivos , Estudos Retrospectivos
4.
Herzschrittmacherther Elektrophysiol ; 32(3): 335-340, 2021 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-34251502

RESUMO

Patients with primary neurogenic disorders such as pure autonomic failure or multiple system atrophy may initially present with cardiocirculatory symptoms such as orthostatic intolerance or fluctuations in heart rate with symptomatic tachycardia. It is therefore clinically important to identify such patients since circulatory manifestations are only one of a series of symptoms resulting from autonomic dysfunction in various organ systems. These patients require a multimodal diagnostic and therapeutic approach and should undergo extensive evaluation in a specialized autonomic nervous system (ANS) outpatient unit. Based on a clinical case presentation, the current review summarizes the diagnostic and therapeutic approach to key cardiovascular symptoms of primary autonomic disorders and their neurological work-up in a specialized autonomic function laboratory.


Assuntos
Doenças do Sistema Nervoso Autônomo , Cardiologia , Hipotensão Ortostática , Neurologia , Intolerância Ortostática , Doenças do Sistema Nervoso Autônomo/diagnóstico , Doenças do Sistema Nervoso Autônomo/terapia , Humanos
5.
Auton Neurosci ; 235: 102836, 2021 11.
Artigo em Inglês | MEDLINE | ID: mdl-34246578

RESUMO

The National Institutes of Health hosted a workshop in 2019 to build consensus around the current state of understanding of the pathophysiology of postural orthostatic tachycardia syndrome (POTS) and to identify knowledge gaps that must be addressed to enhance clinical care of POTS patients through research. This second (of two) articles summarizes current knowledge gaps, and outlines the clinical and research priorities for POTS. POTS is a complex, multi-system, chronic disorder of the autonomic nervous system characterized by orthostatic intolerance and orthostatic tachycardia without hypotension. Patients often experience a host of other related disabling symptoms. The functional and economic impacts of this disorder are significant. The pathophysiology remains incompletely understood. Beyond the significant gaps in understanding the disorder itself, there is a paucity of evidence to guide treatment which can contribute to suboptimal care for this patient population. The vast majority of physicians have minimal to no familiarity or training in the assessment and management of POTS. Funding for POTS research remains very low relative to the size of the patient population and impact of the syndrome. In addition to efforts to improve awareness and physician education, an investment in research infrastructure including the development of standardized disease-specific evaluation tools and outcome measures is needed to facilitate effective collaborative research. A national POTS research consortium could facilitate well-controlled multidisciplinary clinical research studies and therapeutic trials. These priorities will require a substantial increase in the number of research investigators and the amount of research funding in this area.


Assuntos
Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Sistema Nervoso Autônomo , Consenso , Humanos , National Institutes of Health (U.S.) , Síndrome da Taquicardia Postural Ortostática/diagnóstico , Síndrome da Taquicardia Postural Ortostática/terapia , Estados Unidos
6.
Auton Neurosci ; 235: 102828, 2021 11.
Artigo em Inglês | MEDLINE | ID: mdl-34144933

RESUMO

Postural orthostatic tachycardia syndrome (POTS) is a chronic and often disabling disorder characterized by orthostatic intolerance with excessive heart rate increase without hypotension during upright posture. Patients often experience a constellation of other typical symptoms including fatigue, exercise intolerance and gastrointestinal distress. A typical patient with POTS is a female of child-bearing age, who often first displays symptoms in adolescence. The onset of POTS may be precipitated by immunological stressors such as a viral infection. A variety of pathophysiologies are involved in the abnormal postural tachycardia response; however, the pathophysiology of the syndrome is incompletely understood and undoubtedly multifaceted. Clinicians and researchers focused on POTS convened at the National Institutes of Health in July 2019 to discuss the current state of understanding of the pathophysiology of POTS and to identify priorities for POTS research. This article, the first of two articles summarizing the information discussed at this meeting, summarizes the current understanding of this disorder and best practices for clinical care. The evaluation of a patient with suspected POTS should seek to establish the diagnosis, identify co-morbid conditions, and exclude conditions that could cause or mimic the syndrome. Once diagnosed, management typically begins with patient education and non-pharmacologic treatment options. Various medications are often used to address specific symptoms, but there are currently no FDA-approved medications for the treatment of POTS, and evidence for many of the medications used to treat POTS is not robust.


Assuntos
Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Adolescente , Consenso , Feminino , Frequência Cardíaca , Humanos , National Institutes of Health (U.S.) , Síndrome da Taquicardia Postural Ortostática/diagnóstico , Síndrome da Taquicardia Postural Ortostática/terapia , Estados Unidos
7.
J Transl Med ; 19(1): 193, 2021 05 04.
Artigo em Inglês | MEDLINE | ID: mdl-33947430

RESUMO

BACKGROUND: Orthostatic intolerance (OI) is a frequent finding in individuals with myalgic encephalomyelitis /chronic fatigue syndrome (ME/CFS). Published studies have proposed that deconditioning is an important pathophysiological mechanism in various forms of OI, including postural orthostatic tachycardia syndrome (POTS), however conflicting opinions exist. Deconditioning can be classified objectively using the predicted peak oxygen consumption (VO2) values from cardiopulmonary exercise testing (CPET). Therefore, if deconditioning is an important contributor to OI symptomatology, one would expect a relation between the degree of reduction in peak VO2during CPET and the degree of reduction in CBF during head-up tilt testing (HUT). METHODS AND RESULTS: In 22 healthy controls and 199 ME/CFS patients were included. Deconditioning was classified by the CPET response as follows: %peak VO2 ≥ 85% = no deconditioning, %peak VO2 65-85% = mild deconditioning, and %peak VO2 < 65% = severe deconditioning. HC had higher oxygen consumption at the ventilatory threshold and at peak exercise as compared to ME/CFS patients (p ranging between 0.001 and < 0.0001). Although ME/CFS patients had significantly greater CBF reduction than HC (p < 0.0001), there were no differences in CBF reduction among ME/CFS patients with no, mild, or severe deconditioning. We classified the hemodynamic response to HUT into three categories: those with a normal heart rate and blood pressure response, postural orthostatic tachycardia syndrome, or orthostatic hypotension. No difference in the degree of CBF reduction was shown in those three groups. CONCLUSION: This study shows that in ME/CFS patients orthostatic intolerance is not caused by deconditioning as defined on cardiopulmonary exercise testing. An abnormal high decline in cerebral blood flow during orthostatic stress was present in all ME/CFS patients regardless of their %peak VO2 results on cardiopulmonary exercise testing.


Assuntos
Síndrome de Fadiga Crônica , Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Exercício Físico , Frequência Cardíaca , Humanos
8.
Intern Med ; 60(16): 2577-2584, 2021 Aug 15.
Artigo em Inglês | MEDLINE | ID: mdl-33896862

RESUMO

Objective Central nervous system dysfunction associated with myalgic encephalomyelitis (ME) has been suggested to be the main cause of chronic fatigue syndrome. In animal models of chronic fatigue, minocycline was reported to act as a suppressor of neural inflammation. Minocycline may thus exert favorable therapeutic effects in patients with ME. Methods Oral minocycline (100 mg×2 on the first day, followed by 100 mg/day for 41 days) was administered to 100 patients with ME. The performance status score (0-9), orthostatic intolerance during the 10-min standing test, neurologic disequilibrium, and neuropathic pain were compared before and after treatment. Results After therapy completion, favorable effects were observed with a decrease in the performance status score of ≥2 points in 27 patients (27%). Before treatment, 6 of the 27 patients had orthostatic intolerance with an inability to complete the 10-min standing test; after treatment, this symptom resolved in 4 and improved in 2 patients. In addition, after treatment, postural orthostatic tachycardia resolved in five of eight patients, disequilibrium resolved in five of eight patients, and fibromyalgia or neuropathic pain was attenuated in four of five patients. The favorable effects appeared dependent on a shorter disease duration, primarily for a duration of less than three years and most frequently within six months of the disease onset. However, acute adverse effects with nausea and/or dizziness caused 38 patients (38%) to discontinue treatment in the first few days. Conclusion Oral minocycline therapy may be an effective treatment option for patients with ME, especially in the initial stage of the disease.


Assuntos
Síndrome de Fadiga Crônica , Intolerância Ortostática , Tontura , Síndrome de Fadiga Crônica/tratamento farmacológico , Humanos , Minociclina , Transtornos das Sensações
9.
Clin Auton Res ; 31(3): 385-394, 2021 06.
Artigo em Inglês | MEDLINE | ID: mdl-33860871

RESUMO

PURPOSE: Post-COVID-19 syndrome is a poorly understood aspect of the current pandemic, with clinical features that overlap with symptoms of autonomic/small fiber dysfunction. An early systematic analysis of autonomic dysfunction following COVID-19 is lacking and may provide initial insights into the spectrum of this condition. METHODS: We conducted a retrospective review of all patients with confirmed history of COVID-19 infection referred for autonomic testing for symptoms concerning for para-/postinfectious autonomic dysfunction at Mayo Clinic Rochester or Jacksonville between March 2020 and January 2021. RESULTS: We identified 27 patients fulfilling the search criteria. Symptoms developed between 0 and 122 days following the acute infection and included lightheadedness (93%), orthostatic headache (22%), syncope (11%), hyperhidrosis (11%), and burning pain (11%). Sudomotor function was abnormal in 36%, cardiovagal function in 27%, and cardiovascular adrenergic function in 7%. The most common clinical scenario was orthostatic symptoms without tachycardia or hypotension (41%); 22% of patients fulfilled the criteria for postural tachycardia syndrome (POTS), and 11% had borderline findings to support orthostatic intolerance. One patient each was diagnosed with autoimmune autonomic ganglionopathy, inappropriate sinus tachycardia, vasodepressor syncope, cough/vasovagal syncope, exacerbation of preexisting orthostatic hypotension, exacerbation of sensory and autonomic neuropathy, and exacerbation of small fiber neuropathy. CONCLUSION: Abnormalities on autonomic testing were seen in the majority of patients but were mild in most cases. The most common finding was orthostatic intolerance, often without objective hemodynamic abnormalities on testing. Unmasking/exacerbation of preexisting conditions was seen. The temporal association between infection and autonomic symptoms implies a causal relationship, which however cannot be proven by this study.


Assuntos
Doenças do Sistema Nervoso Autônomo/etiologia , COVID-19/complicações , Adulto , Idoso , Disreflexia Autonômica/etiologia , Fibras Autônomas Pós-Ganglionares/patologia , Doenças do Sistema Nervoso Autônomo/diagnóstico , Doenças do Sistema Nervoso Autônomo/fisiopatologia , Tontura , Feminino , Hemodinâmica , Humanos , Masculino , Pessoa de Meia-Idade , Exame Neurológico , Intolerância Ortostática/diagnóstico , Síndrome da Taquicardia Postural Ortostática/etiologia , Estudos Retrospectivos , Síndrome de Shy-Drager/etiologia , Adulto Jovem
11.
Obes Surg ; 31(5): 2250-2254, 2021 May.
Artigo em Inglês | MEDLINE | ID: mdl-33655427

RESUMO

Predisposing factors of new-onset orthostatic intolerance (OI) after bariatric surgery (BS) are unknown. The purpose of this study is to summarize current existing data on new-onset OI after BS. Materials and methods were considered for a search of articles that were published by the 30th of July 2020. A systematic review in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines and in line with the protocol agreed by all authors was conducted. Of the 604 initially identified articles, four studies were found to match the established criteria and were extracted for eligibility. 83.3% were female. Hypertension, type 2 diabetes mellitus, and obstructive sleep apnea syndrome were the most frequently reported comorbidities. Surgical intervention such as revision, conversion, or reversal was not documented in these studies. Awareness of this issue must be raised due to the possibility of reduced quality of life and the risk of syncope.


Assuntos
Cirurgia Bariátrica , Diabetes Mellitus Tipo 2 , Obesidade Mórbida , Intolerância Ortostática , Cirurgia Bariátrica/efeitos adversos , Diabetes Mellitus Tipo 2/epidemiologia , Feminino , Humanos , Masculino , Obesidade Mórbida/cirurgia , Intolerância Ortostática/epidemiologia , Intolerância Ortostática/etiologia , Qualidade de Vida
12.
Med Biol Eng Comput ; 59(3): 621-632, 2021 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-33582941

RESUMO

Imbalance in the autonomic nervous system can lead to orthostatic intolerance manifested by dizziness, lightheadedness, and a sudden loss of consciousness (syncope); these are common conditions, but they are challenging to diagnose correctly. Uncertainties about the triggering mechanisms and the underlying pathophysiology have led to variations in their classification. This study uses machine learning to categorize patients with orthostatic intolerance. We use random forest classification trees to identify a small number of markers in blood pressure, and heart rate time-series data measured during head-up tilt to (a) distinguish patients with a single pathology and (b) examine data from patients with a mixed pathophysiology. Next, we use Kmeans to cluster the markers representing the time-series data. We apply the proposed method analyzing clinical data from 186 subjects identified as control or suffering from one of four conditions: postural orthostatic tachycardia (POTS), cardioinhibition, vasodepression, and mixed cardioinhibition and vasodepression. Classification results confirm the use of supervised machine learning. We were able to categorize more than 95% of patients with a single condition and were able to subgroup all patients with mixed cardioinhibitory and vasodepressor syncope. Clustering results confirm the disease groups and identify two distinct subgroups within the control and mixed groups. The proposed study demonstrates how to use machine learning to discover structure in blood pressure and heart rate time-series data. The methodology is used in classification of patients with orthostatic intolerance. Diagnosing orthostatic intolerance is challenging, and full characterization of the pathophysiological mechanisms remains a topic of ongoing research. This study provides a step toward leveraging machine learning to assist clinicians and researchers in addressing these challenges. Graphical abstract Machine learning tools utilized to analyze heart rate (HR) and blood pressure (BP) time-series data from syncope and control patients. Results show that machine learning can provide accurate classification of disease groups for 98% of patients and we identified two subgroups within the control patients differentiated by their BP response.


Assuntos
Intolerância Ortostática , Sistema Nervoso Autônomo , Pressão Sanguínea , Ciência de Dados , Frequência Cardíaca , Humanos , Intolerância Ortostática/diagnóstico , Síncope/diagnóstico , Teste da Mesa Inclinada
13.
Clin Auton Res ; 31(3): 433-441, 2021 06.
Artigo em Inglês | MEDLINE | ID: mdl-33550497

RESUMO

PURPOSE: Postural tachycardia syndrome (POTS) and vasovagal syncope (VVS) are two disorders of orthostatic intolerance which are often misdiagnosed as the other. In each case, patients experience a reduced health-related quality of life (HRQoL) compared to healthy populations. This study was conducted to test the hypothesis that HRQoL is worse in POTS. METHODS: POTS patients were recruited from the Dysautonomia International Annual Patient and Caregiver Conference. VVS patient data came from those enrolled in the Second Prevention of Syncope Trial. Participants aged ≥ 18 years (177 POTS and 72 VVS) completed the RAND 36-Item Health Survey, a generic and coherent health-related quality of life survey. RESULTS: POTS patients reported reduced HRQoL compared to VVS patients in physical functioning (42.5 ± 1.7 vs. 76.5 ± 2.9, p < 0.001), role limitations due to physical health (11.4 ± 1.9 vs. 33.0 ± 5.0, p < 0.001), energy and fatigue (27.2 ± 1.3 vs. 50.7 ± 2.6, p < 0.001), social functioning (45.2 ± 1.8 vs. 71.2 ± 2.9, p < 0.001), pain (48.8 ± 1.9 vs. 67.7 ± 2.9, p < 0.001), and general health (31.2 ± 1.5 vs. 60.5 ± 2.6, p < 0.001) domains. Scores did not differ significantly in the role limitations due to emotional health (p = 0.052) and emotional well-being (p = 0.271) domains. Physical and general health composite scores were lower in the POTS population, while mental health composite scores were not different. CONCLUSION: Differences in HRQoL exist between these patient populations. POTS patients report lower scores in physical and general health domains than VVS patients, but emotional health domains do not differ significantly. Targeting physical functioning in these patients may help improve quality of life.


Assuntos
Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Síncope Vasovagal , Humanos , Qualidade de Vida , Síncope
14.
Auton Neurosci ; 231: 102770, 2021 03.
Artigo em Inglês | MEDLINE | ID: mdl-33388686

RESUMO

INTRODUCTION: Patients with postural orthostatic tachycardia syndrome (POTS) have been shown to exhibit comorbid joint hypermobility manifested as Ehlers-Danlos syndrome (EDS) or hypermobility spectrum disorder (HSD). The prevalence of EDS and HSD in POTS has been demonstrated in smaller studies combining adult and pediatric patients. We examined a large series of pediatric patients to determine their prevalence in children with POTS. MATERIALS AND METHODS: Patients 18 years old, or less, at initial evaluation at our clinic were included. POTS was diagnosed based on at least six months of frequent debilitating symptoms of orthostatic intolerance, plus a consistent heart rate increase of at least 40 beats per minute without orthostatic hypotension on standing test. Patients with a Beighton score of at least 5/9 plus other systemic findings suggestive of EDS were further evaluated in Connective Tissue Disorders clinics. RESULTS: There were 362 patients meeting inclusion criteria, of which 82 patients had EDS (22.7%) and 141 patients had HSD (39.0%). Patients with EDS had an earlier median age at symptom onset (12.1 vs. 13.5 years, p=0.004) and longer median symptom duration (2.5 vs. 1.5 years, p=0.0008) compared to patients without hypermobility. CONCLUSIONS: Our evaluation of a large series of pediatric patients with POTS revealed that over one-fifth of patients had EDS and over one-third of patients had HSD. The awareness of the prevalence of comorbidities such as hypermobility disorders may help inform providers diagnosing and caring for these patients.


Assuntos
Síndrome de Ehlers-Danlos , Instabilidade Articular , Intolerância Ortostática , Síndrome da Taquicardia Postural Ortostática , Adolescente , Adulto , Criança , Síndrome de Ehlers-Danlos/complicações , Síndrome de Ehlers-Danlos/diagnóstico , Síndrome de Ehlers-Danlos/epidemiologia , Humanos , Instabilidade Articular/epidemiologia , Síndrome da Taquicardia Postural Ortostática/diagnóstico , Síndrome da Taquicardia Postural Ortostática/epidemiologia , Prevalência
15.
Ann Neurol ; 89(4): 753-768, 2021 04.
Artigo em Inglês | MEDLINE | ID: mdl-33438240

RESUMO

OBJECTIVE: The objective of this study was to evaluate patients with ganglionic acetylcholine receptor antibody (gAChR-Ab) positive autoimmune autonomic ganglionopathy using a multimodal testing protocol to characterize their full clinical phenotype and explore biomarkers to quantify immunotherapy response. METHODS: We conducted a cohort study of 13 individuals (7 women, 21-69 years of age) with autonomic failure and gAChR-Ab >100 pM identified between 2005 and 2019. From 2018, all patients were longitudinally assessed with cardiovascular, pupillary, urinary, sudomotor, lacrimal and salivary testing, and Composite Autonomic Symptom Score (COMPASS-31) autonomic symptom questionnaires. The orthostatic intolerance ratio was calculated by dividing change in systolic blood pressure over time tolerated on head-up tilt. Eleven patients received immunotherapy. RESULTS: At first assessment, all 13 patients had cardiovascular and pupillary impairments, 7 of 8 had postganglionic sudomotor dysfunction, 9 of 11 had urinary retention and xeropthalmia, and 6 of 8 had xerostomia. After immunotherapy, there were significant improvements in orthostatic intolerance ratio (33.3 [17.8-61.3] to 5.2 [1.4-8.2], p = 0.007), heart rate response to deep breathing (1.5 [0.0-3.3] to 4.5 [3.0-6.3], p = 0.02), pupillary constriction to light (12.0 [5.5-18.0] to 19.0 [10.6-23.8]%, p = 0.02), saliva production (0.01 [0.01-0.05] to 0.08 [0.02-0.20] g/min, p = 0.03), and COMPASS-31 scores (52 to 17, p = 0.03). Orthostatic intolerance ratio correlated with autonomic symptoms at baseline (r = 0.841, p = 0.01) and following immunotherapy (r = 0.889, p = 0.02). Immunofluorescence analyses of skin samples from a patient 32 years after disease onset showed loss of nerve fibers supplying the dermal autonomic adnexa and epidermis, with clear improvements following immunotherapy. INTERPRETATION: Patients with autoimmune autonomic ganglionopathy demonstrated objective evidence of widespread sympathetic and parasympathetic autonomic failure, with significant improvements after immunotherapy. Quantitative autonomic biomarkers should be used to define initial deficits, guide therapeutic decisions, and document treatment response. ANN NEUROL 2021;89:753-768.


Assuntos
Doenças Autoimunes do Sistema Nervoso/diagnóstico , Doenças do Sistema Nervoso Autônomo/diagnóstico , Biomarcadores/análise , Gânglios Autônomos , Adulto , Idoso , Doenças Autoimunes do Sistema Nervoso/terapia , Doenças do Sistema Nervoso Autônomo/terapia , Pressão Sanguínea , Estudos de Coortes , Feminino , Humanos , Imunoterapia , Estudos Longitudinais , Masculino , Pessoa de Meia-Idade , Fibras Nervosas/patologia , Intolerância Ortostática , Prognóstico , Receptores Colinérgicos/imunologia , Pele/patologia , Resultado do Tratamento , Adulto Jovem
16.
Eur J Appl Physiol ; 121(1): 265-277, 2021 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-33047259

RESUMO

PURPOSE: While the possible ergogenic benefits of remote ischemic preconditioning (RIPC) make it an attractive training modality, the mechanisms of action remain unclear. Alterations in neural tone have been demonstrated in conjunction with circulatory occlusion, yet investigation of the autonomic nervous system following RIPC treatment has received little attention. We sought to characterize alterations in autonomic balance to both RIPC and augmented RIPC (RIPCaug) performed while cycling, using acute and sustained autonomic indices. METHODS: Thirteen participants (8M:5F) recorded baseline waking heart rate variability (HRV) for 5 days prior to treatment. Participants then completed control exercise (CON), RIPC, and RIPCaug interventions in a randomized cross-over design. Cardiovascular measurements were recorded immediately before and after each intervention at rest, and during an orthostatic challenge. Waking HRV was repeated the morning after each intervention. RESULTS: RIPC resulted in acutely reduced resting heart rates (HR) (∆ - 4 ± 6 bpm, P = 0.02) and suppressed HR 30 s following the orthostatic challenge compared to CON (64 ± 10 vs 74 ± 9 bpm, P = 0.003). RIPCaug yielded elevated HRs compared to CON and RIPC prior to (P = 0.003) and during the orthostatic challenge (P = 0.002). RIPCaug reduced LnSDNN (Baseline 4.39 ± 0.27; CON 4.44 ± 0.39; RIPC 4.41 ± 0.34; RIPCaug 4.22 ± 0.29, P = 0.02) and LnHfa power (Baseline 7.82 ± 0.54; CON 7.73 ± 1.11; RIPC 7.89 ± 0.78; RIPCaug 7.23 ± 0.87, P = 0.04) the morning after treatment compared to all other conditions. CONCLUSIONS: Our data suggest that RIPC may influence HR acutely, possibly through a reduction in cardiac sympathetic activity, and that RIPCaug reduces HRV through cardiac vagal withdrawal or increased cardiac sympathetic modulation, with alterations persisting until the following morning. These findings imply a dose-response relationship with potential for optimization of performance.


Assuntos
Precondicionamento Isquêmico/métodos , Sistema Nervoso Simpático/fisiologia , Adulto , Feminino , Coração/fisiologia , Frequência Cardíaca , Humanos , Precondicionamento Isquêmico/efeitos adversos , Masculino , Intolerância Ortostática/etiologia
17.
World Neurosurg ; 145: 256-259, 2021 01.
Artigo em Inglês | MEDLINE | ID: mdl-32992060

RESUMO

BACKGROUND: Posterior fossa arachnoid cysts are often asymptomatic, but can rarely cause postural headache, the mechanism of which remains unknown. CASE DESCRIPTION: We present a 40-year-old woman with an asymptomatic supracerebellar arachnoid cyst. Upright computed tomography (CT) showed enlargement of the arachnoid cyst and caudal descent of the cerebellum compared with supine CT with narrowing of the craniocervical junction cerebrospinal fluid space. CONCLUSIONS: This finding aids in understanding the association of posterior fossa arachnoid cysts with orthostatic headache. Clinicians should be aware of possible posture-related changes in intracranial structures.


Assuntos
Cistos Aracnóideos/diagnóstico por imagem , Doenças Cerebelares/diagnóstico por imagem , Tomografia Computadorizada por Raios X/métodos , Adulto , Cistos Aracnóideos/cirurgia , Neoplasias Encefálicas/cirurgia , Doenças Cerebelares/cirurgia , Cerebelo/diagnóstico por imagem , Fossa Craniana Posterior/diagnóstico por imagem , Feminino , Humanos , Imageamento por Ressonância Magnética , Oligodendroglioma/cirurgia , Intolerância Ortostática , Resultado do Tratamento
19.
J Am Geriatr Soc ; 69(3): 767-772, 2021 03.
Artigo em Inglês | MEDLINE | ID: mdl-33314116

RESUMO

BACKGROUND: Exacerbation of or new onset orthostatic hypotension in perioperative patients can occur. There is complex underlying pathophysiology with further derailment likely caused by acute cardiovascular changes associated with surgery. The implications for post-operative recovery are unclear, particularly in frail and older patients. We retrospectively explored patient notes for evidence of post-operative orthostatic intolerance in relation to pre-operative orthostatic hypotension. METHODS: Supine and 1-minute and 3-minute standing blood pressure measures obtained from adult patients before mainly general, orthopedic or uro/gynecology surgery were compared to post-operative outcome, specifically, evidence in patient notes about falls, feeling dizzy/unsteady and/or fearful to stand. Orthostatic hypotension was defined as a 20 mmHg or more and/or 10 mmHg or more fall in systolic and diastolic blood pressure, respectively, within ~3 minutes of standing after lying supine for an electrocardiogram. RESULTS: Whilst all patients included had a 1-minute standing blood pressure assessment (N = 170), 3-minute assessment was performed less commonly (N = 113). Nevertheless, one-quarter (23.5%; N = 40) of 170 patients had pre-operative orthostatic hypotension. This was not clearly explained by cardiac or neurological disease or by common medications, but did occur more frequently in older patients and in those aged 65 years or more with higher clinical frailty scale scores. The COVID-19 pandemic reduced the number of patients progressing to surgery within the planned study timescale (N = 143/170; 84.1%). Nevertheless, patients with orthostatic hypotension stayed longer in hospital post-operatively and were more likely to have an episode of fall, unsteadiness and/or dizziness documented (un-prompted) in their notes. CONCLUSIONS: These data provide further impetus for research into modifiable perioperative risk factors associated with orthostatic hypotension. These risks are not confined to those with a pre-existing dysautonomia diagnosis.


Assuntos
Pressão Sanguínea , Fragilidade/fisiopatologia , Hipotensão Ortostática/diagnóstico , Intolerância Ortostática/etiologia , Complicações Pós-Operatórias/etiologia , Idoso , Idoso de 80 Anos ou mais , Determinação da Pressão Arterial , COVID-19 , Feminino , Fragilidade/complicações , Fragilidade/cirurgia , Avaliação Geriátrica , Humanos , Hipotensão Ortostática/etiologia , Masculino , Período Pré-Operatório , Estudos Retrospectivos , Medição de Risco , Fatores de Risco , SARS-CoV-2 , Resultado do Tratamento
20.
J Pediatr ; 231: 141-147, 2021 04.
Artigo em Inglês | MEDLINE | ID: mdl-33338494

RESUMO

OBJECTIVES: To report the intermediate-term outcome following surgical intervention for median arcuate ligament syndrome (MALS) in adolescents and young adults with orthostatic intolerance (OI) to assess clinical improvement in the gastrointestinal and 5 other functional domains and if relief of arterial obstruction is associated with resolution of clinical symptoms. STUDY DESIGN: Thirty-one patients were given 2 dysautonomia-designed questionnaires to assess changes in symptoms following operative intervention in 6 functional domains and underwent postoperative repeat abdominal ultrasound examinations. RESULTS: Average follow-up after surgery was 22.4 ± 14.8 months. Self-assessed quality of health on a Likert scale (1-10 with 10 being normal) improved from 4.5 ± 2.1 preoperatively to 5.3 ± 2.4 postoperatively (P = not significant). Gastrointestinal symptoms of abdominal pain, nausea, and vomiting improved in 63% (P = .007), 53% (P = .040), and 62% (P = .014) of patients, respectively. Cardiovascular symptoms of dizziness, syncope, chest pain, and palpitations improved in 45% (P = not significant), 50% (P = not significant), 54% (P = .043), and 54% (P = .037) of patients, respectively. Transabdominal ultrasound peak supine expiratory velocity decreased from 348 ± 105 cm/s preoperatively to 251 ± 109 cm/s at 6 months or more after a ligament release procedure. Decrease of the postoperative celiac artery Doppler velocity was not associated with an improvement in gastrointestinal symptoms (P = .075). CONCLUSIONS: Adolescent and young adult patients with median arcuate ligament syndrome and OI have a good response to surgical intervention. About two-thirds of patients report significant improvement in symptoms of abdominal pain, nausea, and vomiting. Despite these encouraging data, many patients with MALS and OI continue to have an impaired quality of health.


Assuntos
Síndrome do Ligamento Arqueado Mediano/cirurgia , Intolerância Ortostática/cirurgia , Adolescente , Feminino , Humanos , Masculino , Síndrome do Ligamento Arqueado Mediano/complicações , Intolerância Ortostática/complicações , Síndrome da Taquicardia Postural Ortostática/complicações , Síndrome da Taquicardia Postural Ortostática/cirurgia , Estudos Prospectivos , Fatores de Tempo , Resultado do Tratamento , Adulto Jovem
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