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Frontiers in Pediatrics 2022While heart rate variability (HRV) is a relevant non-invasive tool to assess the autonomic nervous system (ANS) functioning with recognized diagnostic and therapeutic...
While heart rate variability (HRV) is a relevant non-invasive tool to assess the autonomic nervous system (ANS) functioning with recognized diagnostic and therapeutic implications, the lack of knowledge on its interest in neonatal medicine is certain. This review aims to briefly describe the algorithms used to decompose variations in the length of the RR interval and better understand the physiological autonomic maturation data of the newborn. Assessing newborns' autonomous reactivity can identify dysautonomia situations and discriminate children with a high risk of life-threatening events, which should benefit from cardiorespiratory monitoring at home. Targeted monitoring of HRV should provide an objective reflection of the newborn's intrinsic capacity for cardiorespiratory self-regulation.
PubMed: 35529337
DOI: 10.3389/fped.2022.860145 -
Orphanet Journal of Rare Diseases Apr 2022Stuve-Wiedemann syndrome (SWS) is a rare and severe genetic disease characterized by skeletal anomalies and dysautonomic disturbances requiring appropriate care. Peer... (Review)
Review
BACKGROUND
Stuve-Wiedemann syndrome (SWS) is a rare and severe genetic disease characterized by skeletal anomalies and dysautonomic disturbances requiring appropriate care. Peer support is mandatory to fill the lack of clinical recommendations in such rare diseases. We report a new case and provide the first systematic review of all previous published cases.
OBJECTIVE
To better describe the timeline of SWS and to improve paediatric management.
DATA SOURCES
SWS English publications available on Pubmed until 31/03/2021.
STUDY SELECTION
Case description combining typical osteo-articular and dysautonomic involvement (with 2 items by categories required for children < 2 years and 3 items > 2 years).
DATA EXTRACTION
Demographic, clinical, genetics and outcome data.
RESULTS
In our cohort of 69 patients, the median age at report was 32 months. Only 46% presented antenatal signs. Mortality rate is higher during the first 2 years (42% < 2 years; 10% > 2 years) mainly due to respiratory failure, pulmonary arterial hypertension appearing to be a poor prognosis factor (mortality rate 63%). After 2 years, orthopaedic symptoms significantly increase including joint mobility restriction (81%), spinal deformations (77%) and fractures (61%).
CONCLUSIONS
Natural history of SWS is marked by a high mortality rate before 2 years due to dysautonomic disturbances. A specialized multidisciplinary approach is needed to address these early mortality risks and then adapt to the specific, mainly orthopaedic, needs of patients after 2 years of age. Further research is required to provide clinical guidelines and improve pre-natal counselling.
Topics: Abnormalities, Multiple; Child; Exostoses, Multiple Hereditary; Female; Humans; Osteochondrodysplasias; Pregnancy; Respiratory Insufficiency
PubMed: 35461249
DOI: 10.1186/s13023-022-02323-8 -
Medicina (Kaunas, Lithuania) Mar 2022Multiple system atrophy (MSA) and progressive supranuclear palsy (PSP) are rare atypical parkinsonian syndromes, characterized by motor and cognitive symptoms. Their... (Review)
Review
Multiple system atrophy (MSA) and progressive supranuclear palsy (PSP) are rare atypical parkinsonian syndromes, characterized by motor and cognitive symptoms. Their clinical diagnosis is challenging because there are no established biomarkers. Dysregulation of microRNAs (miRNAs/miRs) has been reported to serve an important role in neurodegenerative diseases. However, the miRNA profiles of MSA and PSP patients are rarely reported. The aim of this study was to critically review the role of miRNAs as diagnostic biomarkers to differentiate these atypical parkinsonian disorders and their role in disease pathogenesis. A systematic literature search of PubMed was conducted up to February 2022 according the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. A total of 15 studies were analyzed. Three studies have shown that miR-9-3p, miR-19a, miR-19b, and miR-24 are potential biomarkers for MSA. In two studies, miR-132 was downregulated, whereas miR-147a and miR-518e were upregulated in the brain tissue of PSP patients. The potential of miRNA is still uncertain as a potential differential diagnostic marker to identify these disorders. Pre-analytical and analytical factors of included studies were important limitations to justify the introduction of miRNAs into clinical practice.
Topics: Biomarkers; Humans; MicroRNAs; Multiple System Atrophy; Parkinsonian Disorders; Supranuclear Palsy, Progressive
PubMed: 35454322
DOI: 10.3390/medicina58040483 -
Movement Disorders : Official Journal... Jun 2022The second consensus criteria for the diagnosis of multiple system atrophy (MSA) are widely recognized as the reference standard for clinical research, but lack... (Review)
Review
BACKGROUND
The second consensus criteria for the diagnosis of multiple system atrophy (MSA) are widely recognized as the reference standard for clinical research, but lack sensitivity to diagnose the disease at early stages.
OBJECTIVE
To develop novel Movement Disorder Society (MDS) criteria for MSA diagnosis using an evidence-based and consensus-based methodology.
METHODS
We identified shortcomings of the second consensus criteria for MSA diagnosis and conducted a systematic literature review to answer predefined questions on clinical presentation and diagnostic tools relevant for MSA diagnosis. The criteria were developed and later optimized using two Delphi rounds within the MSA Criteria Revision Task Force, a survey for MDS membership, and a virtual Consensus Conference.
RESULTS
The criteria for neuropathologically established MSA remain unchanged. For a clinical MSA diagnosis a new category of clinically established MSA is introduced, aiming for maximum specificity with acceptable sensitivity. A category of clinically probable MSA is defined to enhance sensitivity while maintaining specificity. A research category of possible prodromal MSA is designed to capture patients in the earliest stages when symptoms and signs are present, but do not meet the threshold for clinically established or clinically probable MSA. Brain magnetic resonance imaging markers suggestive of MSA are required for the diagnosis of clinically established MSA. The number of research biomarkers that support all clinical diagnostic categories will likely grow.
CONCLUSIONS
This set of MDS MSA diagnostic criteria aims at improving the diagnostic accuracy, particularly in early disease stages. It requires validation in a prospective clinical and a clinicopathological study. © 2022 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.
Topics: Brain; Consensus; Humans; Magnetic Resonance Imaging; Multiple System Atrophy; Prospective Studies
PubMed: 35445419
DOI: 10.1002/mds.29005 -
European Journal of Pain (London,... Jul 2022To improve CRPS treatment, it is imperative to understand the nature, degree and relative importance of ongoing problems associated with CRPS. The objective of this... (Review)
Review
BACKGROUND AND OBJECTIVE
To improve CRPS treatment, it is imperative to understand the nature, degree and relative importance of ongoing problems associated with CRPS. The objective of this systematic review was to summarize the published data concerning measures of function and impact including occupational parameters, of CRPS at 12 months from symptom onset and beyond.
DATABASES AND DATA TREATMENT
MEDLINE, EmBase and PsychINFO were searched (inception to May 2021). Study cohorts were eligible if they included; adult patients with the primary complaint of CRPS ≥12 months duration, outcomes that reported change in CRPS signs and symptoms, and physical and social function. Prospero registration: CRD42021241785.
RESULTS
Twenty-two included studies suggest that pain and motor dysfunction are the most dominant long-term features of CRPS, persisting for 51%-89% of patients at ≥12 months. On average for all patients who had CRPS at baseline, grip strength was found to be reduced by 25%-66%, and range of motion reduced by 20%-25% at ≥12 months. Such losses were associated with physical and social disability. Thirty to forty percent of all patients did not return to work and a further 27%-35% of persons returned to work but required some form of workplace adaptation, although the quality of this data was poor. Quality assessment highlighted limitations in the literature, such as high attrition bias and variations in diagnostic criteria.
CONCLUSIONS
Results provide first-time quantitative data including specific evidence about losses to motor function and long-term compromises to work status. Results demonstrate that the ongoing impact of one episode of CRPS on limb function and work status is relatively high.
SIGNIFICANCE
This review provides first-time clarity in relation to outcomes of limb function and work status associated with an episode of CRPS, beyond 12 months from onset. Results demonstrate that the long-term impact of an episode of CRPS on these outcomes is much larger than previously described, and thus also illustrates how the wider health economic impact of CRPS is not yet fully understood. We additionally highlight the need for future research that identifies long-term predictors, and treatments that can foster good functional and occupational recovery.
Topics: Adult; Complex Regional Pain Syndromes; Disabled Persons; Extremities; Humans; Pain; Pain Measurement
PubMed: 35435302
DOI: 10.1002/ejp.1953 -
International Journal of Environmental... Mar 2022Objective: Cardiovascular effects of thyroid hormones may be measured through heart rate variability (HRV). We sought to determine the impact of hyperthyroidism on HRV.... (Meta-Analysis)
Meta-Analysis Review
Objective: Cardiovascular effects of thyroid hormones may be measured through heart rate variability (HRV). We sought to determine the impact of hyperthyroidism on HRV. Design: A systematic review and meta-analysis on the impact of hyperthyroidism on HRV. Methods: PubMed, Cochrane, Embase and Google Scholar were searched until 20 August 2021 for articles reporting HRV parameters in untreated hyperthyroidism and healthy controls. Random-effects meta-analysis was stratified by degree of hyperthyroidism for each HRV parameter: RR intervals (or Normal-to-Normal intervals—NN), SDNN (standard deviation of RR intervals), RMSSD (square root of the mean difference of successive RR intervals), pNN50 (percentage of RR intervals with >50 ms of variation), total power (TP), LFnu (low-frequency normalized unit) and HFnu (high-frequency), VLF (very low-frequency), and LF/HF ratio. Results: We included 22 studies with 10,811 patients: 1002 with hyperthyroidism and 9809 healthy controls. There was a decrease in RR (effect size = −4.63, 95% CI −5.7 to −3.56), SDNN (−6.07, −7.42 to −4.71), RMSSD (−1.52, −2.18 to −0.87), pNN50 (−1.36, −1.83 to −0.88), TP (−2.05, −2.87 to −1.24), HFnu (−3.51, −4.76 to −2.26), and VLF power (−2.65, −3.74 to −1.55), and an increase in LFnu (2.66, 1.55 to 3.78) and LF/HF ratio (1.75, 1.02 to 2.48) (p < 0.01). Most parameters had ES that was twice as high in overt compared to subclinical hyperthyroidism. Increased peripheral thyroid hormones and decreased TSH levels were associated with lower RR intervals. Conclusions: Hyperthyroidism is associated with a decreased HRV, which may be explained by the deleterious effect of thyroid hormones and TSH. The increased sympathetic and decreased parasympathetic activity may have clinical implications.
Topics: Cardiovascular System; Heart; Heart Rate; Humans; Hyperthyroidism; Thyrotropin
PubMed: 35329294
DOI: 10.3390/ijerph19063606 -
Biomedical Engineering Online Mar 2022Mental illness represents a major global burden of disease worldwide. It has been hypothesised that individuals with mental illness have greater blood pressure... (Review)
Review
BACKGROUND
Mental illness represents a major global burden of disease worldwide. It has been hypothesised that individuals with mental illness have greater blood pressure fluctuations that lead to increased cardiovascular risk and target organ damage. This systematic review aims to (i) investigate the association between mental illness and blood pressure variability (BPV) and (ii) describe methods of BPV measurements and analysis which may affect pattern and degree of variability.
METHODS
Four electronic databases were searched from inception until 2020. The quality assessment was performed using STROBE criteria. Studies were included if they investigated BPV (including either frequency or time domain analysis) in individuals with mental illness (particularly anxiety/generalised anxiety disorder, depression/major depressive disorder, panic disorder and hostility) and without hypertension. Two authors independently screened titles, abstracts and full texts. A third author resolved any disagreements.
RESULTS
Twelve studies met the inclusion criteria. Three studies measured short-term BPV, two measured long-term BPV and seven measured ultra-short-term BPV. All studies related to short-term BPV using ambulatory and home blood pressure monitoring found a higher BPV in individuals with depression or panic disorder. The two studies measuring long-term BPV were limited to the older population and found mixed results. Mental illness is significantly associated with an increased BPV in younger and middle-aged adults. All studies of ultra-short-term BPV using standard cardiac autonomic assessment; non-invasive continuous finger blood pressure and heart rate signals found significant association between BPV and mental illness. A mixed result related to degree of tilt during tilt assessment and between controlled and spontaneous breathing were observed in patients with psychological state.
CONCLUSIONS
Current review found that people with mental illness is significantly associated with an increased BPV regardless of age. Since mental illness can contribute to the deterioration of autonomic function (HRV, BPV), early therapeutic intervention in mental illness may prevent diseases associated with autonomic dysregulation and reduce the likelihood of negative cardiac outcomes. Therefore, these findings may have important implications for patients' future physical health and well-being, highlighting the need for comprehensive cardiovascular risk reduction.
Topics: Adult; Blood Pressure; Blood Pressure Monitoring, Ambulatory; Depressive Disorder, Major; Humans; Hypertension; Mental Disorders; Middle Aged
PubMed: 35313918
DOI: 10.1186/s12938-022-00985-w -
[Nihon Koshu Eisei Zasshi] Japanese... May 2022Objectives Minor health complaints related to stress, mental health, sleep, and fatigue are closely associated with each other, and their deterioration may cause...
Objectives Minor health complaints related to stress, mental health, sleep, and fatigue are closely associated with each other, and their deterioration may cause lifestyle diseases. The health status of people can be predicted through a questionnaire by exploring the relationship between their state of minor health complaints and objective health status indices. Therefore, we conducted a systematic review of the relationship between a questionnaire on the state of minor health complaints and health status indices among Japanese people who have a high level of stress, which they experience on a daily basis, using epidemiological literature. Additionally, we considered items for the questionnaire which were necessary for an index development.Methods The PubMed database was searched for papers on "autonomic nervous system," "sleep disorders," "mental health and stress," and "fatigue," using keywords mentioned in previous studies on minor health complaints. The extracted research papers were screened according to the following inclusion criteria: 1) the participants were healthy Japanese people; 2) descriptions included characteristics of the target population; 3) use of analytic epidemiological study design, intervention studies, and systematic reviews; 4) minor health complaints assessed by a questionnaire; 5) evaluation of the relationship between the questionnaires for minor health complaints and the health index; and 6) written in Japanese or English. Based on this, ten papers were adopted.Results Of the 10 papers collected, one was a cohort study, three were case-control studies, and six were cross-sectional studies. The participants in five of them were working adults. Reports on three out of six questionnaires on stress, four out of seven on sleep, and all two on comprehensive health status showed significant associations between minor health complaints assessed by the questionnaires and the index for health status. The increase in responses about work-related stress from the questionnaire was associated with an increase in the "risk of developing depression" [odds ratio 2.96 (confidence interval: 1.04-8.42)]. Poor sleep quality was associated with an increase in "changes in autonomic index," "number of comorbidities and the rate of depression," and the "risk of work-related injuries." Moreover, the health score was associated with the "autonomic nervous system index".Conclusion These results suggest that a questionnaire evaluating minor health complaints should include questions about "stress," "sleep quality," and "comprehensive health status." Since studies that appropriately adopted for risk of bias were limited, it is necessary to further examine these relationships by applying prospective studies such as cohort studies and intervention studies.
Topics: Adult; Cohort Studies; Fatigue; Health Status; Health Status Indicators; Humans; Japan; Prospective Studies; Surveys and Questionnaires
PubMed: 35296592
DOI: 10.11236/jph.21-099 -
European Journal of Pediatrics May 2022Patients who undergo salivary gland, neck, or facelift surgery or suffer from diabetes mellitus often develop Frey syndrome (also known as auriculotemporal syndrome or...
Patients who undergo salivary gland, neck, or facelift surgery or suffer from diabetes mellitus often develop Frey syndrome (also known as auriculotemporal syndrome or gustatory sweating). Frey syndrome has been occasionally reported to occur in subjects without history of surgery or diabetes but this variant of Frey syndrome has not been systematically investigated. We searched for original articles of Frey syndrome unrelated to surgery or diabetes without date and language restriction. Article selection and data extraction were performed in duplicate. Our systematic review included 76 reports describing 121 individual cases (67 males and 54 females) of Frey syndrome not associated with surgery or diabetes. The age at onset of symptoms was ≤ 18 years in 113 (93%) cases. The time to diagnosis was 12 months or more in 55 (45%) cases. On the other hand, an allergy evaluation was performed in half of the cases. A possible cause for Frey syndrome was detected in 85 (70%) cases, most frequently history of forceps birth (N = 63; 52%). The majority of the remaining 22 cases occurred after a blunt face trauma, following an auriculotemporal nerve neuritis or in association with a neurocutaneous syndrome. The cause underlying Frey syndrome was unknown in 36 cases. Conclusion: Frey syndrome not associated with surgery or diabetes almost exclusively affects subjects in pediatric age and is uncommon and underrecognized. Most cases occur after forceps birth. There is a need to expand awareness of this pseudo-allergic reaction among pediatricians and allergists. What is Known: • Pre-auricular reddening, sweating, and warmth in response to mastication or a salivary stimulus characterize Frey syndrome. • It usually occurs after salivary gland surgery and in diabetes. What is New: • In children, Frey syndrome is rare, and most cases occur after a forceps-assisted birth. • In childhood, this condition is often erroneously attributed to food allergy.
Topics: Child; Diabetes Mellitus; Female; Food Hypersensitivity; Humans; Male; Neck; Sweating, Gustatory
PubMed: 35182195
DOI: 10.1007/s00431-022-04415-w -
Medicina (Kaunas, Lithuania) Dec 2021: Migraine headaches are chronic neurological diseases that reduce the quality of life by causing severe headaches and autonomic nervous system dysfunction, such as... (Meta-Analysis)
Meta-Analysis Review
: Migraine headaches are chronic neurological diseases that reduce the quality of life by causing severe headaches and autonomic nervous system dysfunction, such as facial flushing, nasal stuffiness, and sweating. Their major treatment methods include medication and cognitive behavioral therapy (CBT). CBT has been used for pain treatment and various psychogenic neurological diseases by reducing pain, disability, and emotional disorders caused by symptoms of mental illness and improving the understanding of mental health. This study aimed to evaluate the effectiveness and safety of CBT in treating migraines. : Seven electronic databases were searched from the date of inception to December 2020. Randomized controlled studies (RCTs) using CBT as an intervention for migraine were included. The primary outcome of this study was to determine the frequency of migraines and the intensity of migraines on Visual Analog Scale (VAS), the frequency of drug use, Migraine Disability Assessment (MIDAS), and Headache Impact Test (HIT-6) index. The two authors independently conducted the data extraction and quality assessment of the included RCTs, and conducted meta-analysis with RevMan V.5.4. : Among the 373 studies, 11 RCTs were included in this systematic review. Seven out of the 11 RCTs were conducted in the USA, and four were conducted in the UK, Germany, Iran, and Italy, respectively. Headache frequency and MIDAS scores were statistically significant reduced. In the subgroup analysis, headache strength was significantly reduced. Two of the included studies reported adverse effects, including worsening of migraine intensity and frequency, respiratory symptoms, and vivid memory of a traumatic event. : CBT for migraine effectively reduced headache frequency and MIDAS score in meta-analysis and headache intensity subgroup analysis, with few adverse events. Additional RCTs with CBT for migraine headaches are needed for a more accurate analysis.
Topics: Cognitive Behavioral Therapy; Disability Evaluation; Headache; Humans; Migraine Disorders; Pain Measurement
PubMed: 35056352
DOI: 10.3390/medicina58010044