Fang Xu, Hui Wang, Hongyan Huang, Qiuyan Shen, Dan Zhang, Yi Bao, Junying Zhou, Yanming Xu
{"title":"Impact of supine body position on sleep-disordered breathing in patients with MSA patients.","authors":"Fang Xu, Hui Wang, Hongyan Huang, Qiuyan Shen, Dan Zhang, Yi Bao, Junying Zhou, Yanming Xu","doi":"10.1007/s10286-024-01099-0","DOIUrl":"https://doi.org/10.1007/s10286-024-01099-0","url":null,"abstract":"","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":""},"PeriodicalIF":3.9,"publicationDate":"2024-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142799628","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
David S Goldstein, Patti Sullivan, Courtney Holmes
{"title":"Decreased urinary excretion of norepinephrine and dopamine in autonomic synucleinopathies.","authors":"David S Goldstein, Patti Sullivan, Courtney Holmes","doi":"10.1007/s10286-024-01093-6","DOIUrl":"https://doi.org/10.1007/s10286-024-01093-6","url":null,"abstract":"<p><strong>Background: </strong>Autonomic synucleinopathies feature autonomic failure and intracellular deposition of the protein alpha-synuclein. Three such conditions are the Lewy body diseases (LBDs) Parkinson's disease (PD) and pure autonomic failure (PAF) and the non-LBD synucleinopathy multiple system atrophy (MSA). These diseases all entail catecholaminergic abnormalities in the brain, sympathetically innervated organs, or both; however, little is known about renal catecholaminergic functions in autonomic synucleinopathies. We measured urinary excretion rates of the sympathetic neurotransmitter norepinephrine, the hormone epinephrine, the autocrine-paracrine substance dopamine, the catecholamine precursor 3,4-dihydroxyphenylalanine (DOPA), 3,4-dihydroxyphenylglycol (DHPG, the main neuronal metabolite of norepinephrine), and 3,4-dihydroxyphenylacetic acid (DOPAC, a major dopamine metabolite), in PD, PAF, and MSA groups and controls.</p><p><strong>Methods: </strong>Data were reviewed from all research participants who had urine collections (usually 3.5 h) at the National Institutes of Health (NIH) Clinical Center from 1995 to 2024. The control cohort had neither autonomic failure nor a movement disorder.</p><p><strong>Results: </strong>Norepinephrine excretion rates were decreased compared with controls in PD (p = 0.0001), PAF (p < 0.0001), and MSA (p < 0.0001). Dopamine excretion was also decreased in the three groups (PD: p = 0.0136, PAF: p = 0.0027, MSA: p = 0.0344). DHPG excretion was decreased in PD (p = 0.0004) and PAF (p = 0.0004) but not in MSA. DOPA and epinephrine excretion did not differ among the study groups.</p><p><strong>Conclusions: </strong>Autonomic synucleinopathies involve decreased urinary excretion rates of norepinephrine and dopamine. Since virtually all of urinary dopamine in humans is derived from circulating DOPA, the low rates of urinary norepinephrine and dopamine excretion may reflect dysfunctions in the renal sympathetic noradrenergic system, the DOPA-dopamine autocrine-paracrine system, or both systems.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":""},"PeriodicalIF":3.9,"publicationDate":"2024-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142799627","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Author Response: \"iSTAND trial of IVIG in POTS: a step in the right direction, but more studies are needed\".","authors":"Steven Vernino, Steve Hopkins, Meredith Bryarly","doi":"10.1007/s10286-024-01098-1","DOIUrl":"https://doi.org/10.1007/s10286-024-01098-1","url":null,"abstract":"","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":""},"PeriodicalIF":3.9,"publicationDate":"2024-12-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142799626","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Silvia Galli, Lanfranco De Carolis, Edoardo Bianchini, Marika Alborghetti, Bianca Caliò, Pierre Pacilio, Alessandra Fanciulli, Francesco E Pontieri, Domiziana Rinaldi
{"title":"Effects of levodopa/carbidopa intestinal gel infusion on autonomic symptoms in advanced Parkinson's disease: a systematic review.","authors":"Silvia Galli, Lanfranco De Carolis, Edoardo Bianchini, Marika Alborghetti, Bianca Caliò, Pierre Pacilio, Alessandra Fanciulli, Francesco E Pontieri, Domiziana Rinaldi","doi":"10.1007/s10286-024-01090-9","DOIUrl":"https://doi.org/10.1007/s10286-024-01090-9","url":null,"abstract":"<p><strong>Purpose: </strong>Autonomic failure has a major impact on the quality of life of individuals with Parkinson's disease (PD), especially in advanced stages of the disease. Levodopa/carbidopa intestinal gel (LCIG) infusion is a well-established treatment for advanced PD with severe motor fluctuations and provides substantial benefit in managing some non-motor symptoms (NMS), such as sleep, fatigue, and neuropsychiatric issues. The effect of LCIG on autonomic symptoms is by contrast not well known. Here we performed a systematic review on the influence of LCIG therapy on autonomic dysfunction in PD individuals.</p><p><strong>Methods: </strong>Following the PRISMA guidelines, we systematically searched for studies that included autonomic outcome measures in LCIG-treated PD individuals, limiting the search to articles written in English and published between January 2005 and June 2023. We evaluated improvement, stability, or worsening of gastrointestinal, urinary, and cardiovascular symptoms at six different timepoints according to clinimetric scale changes compared to baseline. Data on autonomic adverse events (AEs) possibly related to LCIG treatment were also collected.</p><p><strong>Results: </strong>Of the 1476 studies identified in the initial search, 16 ultimately met the inclusion criteria and underwent quality assessment and data extraction, with data from 1361 PD patients (18.3 months mean follow-up). Thirteen studies reported improvement or stability of gastrointestinal, urinary, and cardiovascular symptoms over the interventional period. One study found a worsening of cardiovascular symptoms and two of urological symptoms. Regarding safety, seven studies reported gastrointestinal (8.4%), urinary (0.5%), and cardiovascular (1.1%) autonomic LCIG-related AEs.</p><p><strong>Conclusions: </strong>LCIG infusion may help to reduce the burden of autonomic symptoms in advanced PD. Prospective studies specifically addressing the effect of LCIG on autonomic function in advanced PD are warranted.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":""},"PeriodicalIF":3.9,"publicationDate":"2024-12-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142779379","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Rachel Debs, Gwenaëlle Diene, Julie Cortadellas, Catherine Molinas, Marc Kermorgant, Maïthé Tauber, Anne Pavy Le Traon
{"title":"Cardiovascular autonomic dysfunction and sleep abnormalities in children with Prader-Willi syndrome.","authors":"Rachel Debs, Gwenaëlle Diene, Julie Cortadellas, Catherine Molinas, Marc Kermorgant, Maïthé Tauber, Anne Pavy Le Traon","doi":"10.1007/s10286-024-01083-8","DOIUrl":"https://doi.org/10.1007/s10286-024-01083-8","url":null,"abstract":"<p><strong>Purpose: </strong>Prader-Willi syndrome (PWS) is a rare genetic neurodevelopmental condition characterized by cognitive disabilities, behavioral problems, hypothalamic dysfunction with obesity, and sleep disorders. A few studies have reported autonomic nervous system dysfunction. Our aim was to investigate dysautonomia by combining sleep studies and standard autonomic testing in regularly followed children with PWS.</p><p><strong>Methods: </strong>In this retrospective study, heart rate variability was analyzed during each sleep stage (polysomnography) using time and frequency domains in PWS children (N = 37) compared with age-matched controls (N = 20). Cardiovascular autonomic testing (Ewing tests) and sweating assessment (electrochemical skin conductance) were also performed in patients over 6 years (N = 23).</p><p><strong>Results: </strong>Autonomic testing: Heart rate changes with active standing and with deep breathing were impaired in 47% and 22% of the children, respectively. Asymptomatic orthostatic hypotension (OH) was found in 26%. Baroreflex sensitivity in supine position was in normal range (14.1 ± 6.7 ms/mmHg). Electrochemical skin conductance was normal. Sleep study: 46% of the children with PWS had obstructive sleep apnea and 24% had central sleep apnea. None of these events were observed in the control group. Mean R-R and time domain heart rate variability parameters were significantly lower compared with controls in N2 and Rapid Eye Movement (REM) sleep stages. Narcoleptic-like phenotype was found in 47% associated with lower low-frequency (LF) power (sympathetic index) in REM sleep.</p><p><strong>Conclusion: </strong>Our study confirms a decreased vagal modulation during both wakefulness and sleep in children with PWS. OH in some patients suggests a sympathetic dysfunction. These changes may contribute to the increased cardiovascular risk in PWS.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":""},"PeriodicalIF":3.9,"publicationDate":"2024-12-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142779364","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Autonomic failure associated with 16p11.2 duplication in two siblings.","authors":"Cole P Denkensohn, Glen A Cook","doi":"10.1007/s10286-024-01058-9","DOIUrl":"10.1007/s10286-024-01058-9","url":null,"abstract":"","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":"603-605"},"PeriodicalIF":3.9,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141981846","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Responses to Valsalva's maneuver in spinal cord injury do not broadly relate to vasoconstrictor capacity.","authors":"Kathryn Burns, Adina E Draghici, J Andrew Taylor","doi":"10.1007/s10286-024-01060-1","DOIUrl":"10.1007/s10286-024-01060-1","url":null,"abstract":"<p><strong>Purpose: </strong>A blood pressure stabilization during late phase II of Valsalva's maneuver may be utilized to confirm sympathetic vasoconstrictor control after a spinal cord injury. This study investigated whether Valsalva response was predictive of hemodynamics during tilt or isometric handgrip.</p><p><strong>Methods: </strong>Presence/absence of Valsalva response was compared to heart rate, mean arterial pressure, leg blood flow, and vascular resistance during head-up tilt and isometric handgrip to fatigue in 14 adults with spinal cord injury from C7 to T12 and 14 controls. Statistics were performed with two-way repeated measure analysis of variance (ANOVA), post hoc t-tests for between-group comparisons, and Mann-Whitney U tests for within-group.</p><p><strong>Results: </strong>In total, six participants with spinal cord injury lacked a blood pressure stabilization for Valsalva's maneuver. However, this was not related to vasoconstrictor responses during the other tests. The groups had similar heart rate and blood pressure changes during tilt, though leg blood flow decreases and vascular resistance increases tended to be smaller at 20° tilt in those with spinal cord injury (p = 0.07 and p = 0.11, respectively). Participants with spinal cord injury had lower heart rates and markedly smaller blood pressure increases during handgrip (both p < 0.05). There were no group differences in leg blood flow, but those with spinal cord injury demonstrated a blunted vascular resistance increase by the final 10% of the handgrip (p < 0.01).</p><p><strong>Conclusions: </strong>Valsalva response was not consistent with hemodynamics during other stimuli, but some individuals evidence increases in sub-lesional vascular resistance to isometric handgrip comparable to controls, suggesting a sympathoexcitatory stimulus may be critical to provoke hemodynamic responses after spinal cord injury.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":"571-581"},"PeriodicalIF":3.9,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142281159","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
E L Williams, C J Mathias, S Sanatani, M J Tipton, V E Claydon
{"title":"In at the deep end: the physiological challenges associated with artistic swimming.","authors":"E L Williams, C J Mathias, S Sanatani, M J Tipton, V E Claydon","doi":"10.1007/s10286-024-01070-z","DOIUrl":"10.1007/s10286-024-01070-z","url":null,"abstract":"","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":"619-624"},"PeriodicalIF":3.9,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11543717/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142371128","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Christopher W Austelle, Stewart S Cox, Kristin E Wills, Bashar W Badran
{"title":"Vagus nerve stimulation (VNS): recent advances and future directions.","authors":"Christopher W Austelle, Stewart S Cox, Kristin E Wills, Bashar W Badran","doi":"10.1007/s10286-024-01065-w","DOIUrl":"10.1007/s10286-024-01065-w","url":null,"abstract":"<p><strong>Purpose: </strong>Vagus nerve stimulation (VNS) is emerging as a unique and potent intervention, particularly within neurology and psychiatry. The clinical value of VNS continues to grow, while the development of noninvasive options promises to change a landscape that is already quickly evolving. In this review, we highlight recent progress in the field and offer readers a glimpse of the future for this bright and promising modality.</p><p><strong>Methods: </strong>We compiled a narrative review of VNS literature using PubMed and organized the discussion by disease states with approved indications (epilepsy, depression, obesity, post-stroke motor rehabilitation, headache), followed by a section highlighting novel, exploratory areas of VNS research. In each section, we summarized the current role, recent advancements, and future directions of VNS in the treatment of each disease.</p><p><strong>Results: </strong>The field continues to gain appreciation for the clinical potential of this modality. VNS was initially developed for treatment-resistant epilepsy, with the first depression studies following shortly thereafter. Overall, VNS has gained approval or clearance in the treatment of medication-refractory epilepsy, treatment-resistant depression, obesity, migraine/cluster headache, and post-stroke motor rehabilitation.</p><p><strong>Conclusion: </strong>Noninvasive VNS represents an opportunity to bridge the translational gap between preclinical and clinical paradigms and may offer the same therapeutic potential as invasive VNS. Further investigation into how VNS parameters modulate behavior and biology, as well as how to translate noninvasive options into the clinical arena, are crucial next steps for researchers and clinicians studying VNS.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":"529-547"},"PeriodicalIF":3.9,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11543756/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142371129","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Marie-Béatrice Saade, Samuel Holden, Lisa Kakinami, Jennifer J McGrath, Marie-Ève Mathieu, Paul Poirier, Tracie A Barnett, Pierre Beaucage, Mélanie Henderson
{"title":"Adiposity and cardiac autonomic function in children with a family history of obesity.","authors":"Marie-Béatrice Saade, Samuel Holden, Lisa Kakinami, Jennifer J McGrath, Marie-Ève Mathieu, Paul Poirier, Tracie A Barnett, Pierre Beaucage, Mélanie Henderson","doi":"10.1007/s10286-024-01063-y","DOIUrl":"10.1007/s10286-024-01063-y","url":null,"abstract":"<p><strong>Purpose: </strong>Data on associations between adiposity and heart rate variability (HRV) in prepubertal children are limited. We examined the associations between adiposity indices and HRV, independent of lifestyle behaviors, comparing multiple indicators of adiposity, and explored differences between boys and girls.</p><p><strong>Methods: </strong>Data stem from 469 participants of the QUALITY cohort (630 children aged 8-10 years with a parental history of obesity). Adiposity indices included waist-to-height ratio, body mass index (BMI) percentiles and categories (overweight, obesity), dual-energy x-ray absorptiometry (DEXA) defined fat mass percentage and android/gynoid ratio. HRV indices in the frequency and the spectral domain were derived from a daytime 3-h Holter recording. Multivariable linear regression models were adjusted for age, sex, Tanner stage, physical activity, screen time, and fitness. Interactions between sex and adiposity were tested.</p><p><strong>Results: </strong>Greater adiposity was associated with decreased parasympathetic modulation and increased sympathetic dominance. Waist-to-height ratio was associated with lower parasympathetic activity: root mean square of the successive differences (RMSSD) [B = -23.32, 95% confidence interval (CI) -42.42, -4.22], pNN50 (B = -16.93, 95% CI - 28.58, - 5.27), LF/HF ratio (B = 1.83, 95% CI 0.97-2.70). Patterns of association were similar for android/gynoid ratio. Overweight was not associated with altered HRV. Obesity was negatively associated with RMSSD and pNN50 and positively with LF/HF ratio. Greater fat mass percentage was associated with lower RMSSD, pNN50, and HF, and increased LF/HF ratio. There were no differences between boys and girls.</p><p><strong>Conclusions: </strong>Specific markers of adiposity relate to altered HRV in childhood, with waist-to-height ratio being potentially a more relevant marker of HRV than BMI and more pragmatic than percent body fat.</p><p><strong>Trial registration: </strong>NCT03356262, 11 November 2017.</p>","PeriodicalId":10168,"journal":{"name":"Clinical Autonomic Research","volume":" ","pages":"583-592"},"PeriodicalIF":3.9,"publicationDate":"2024-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142281157","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}