Rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation (ROHHAD) syndrome is a rare disease of concurrent respiratory dysfunction and autonomic dysregulation with endocrine abnormalities. ROHHADNET includes ROHHAD plus coexisting neuroendocrine tumors (NETs). We describe an eight-year-old boy, who originally presented at four years of age with rapid weight gain and hyperhidrosis and who developed mild obstructive sleep apnea (OSA). His clinical course was eventually complicated by hypoxic respiratory failure requiring admission to the pediatric intensive care unit (PICU). Echocardiogram at that time demonstrated dilated cardiomyopathy left ventricular ejection fraction (LVEF) of 28% at time of admission. His respiratory failure persisted despite average volume-assured pressure support (AVAPS) around the clock leading to tracheostomy placement for cardiopulmonary support. He also demonstrated autonomic instability with multiple pituitary hormone deficiencies. Computed tomography (CT) imaging of the abdomen and pelvis demonstrated a presacral soft tissue mass consistent with a tumor of neural crest origin. Daytime somnolence and confusion progressed and a low cerebrospinal fluid hypocretin level revealed a diagnosis of narcolepsy type 1.
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http://dx.doi.org/10.7759/cureus.50341 | DOI Listing |
Neurology
February 2025
From the Autonomic Medicine Section, Clinical Neurosciences Program, Division of Intramural Research, National Institute of Neurological Disorders and Stroke, NIH, Bethesda, MD.
Background And Objectives: Lewy body diseases (LBDs) such as Parkinson disease (PD) feature increased deposition of α-synuclein (α-syn) in cutaneous sympathetic noradrenergic nerves. The pathophysiologic significance of sympathetic intraneuronal α-syn is unclear. We reviewed data about immunoreactive α-syn, tyrosine hydroxylase (TH, a marker of catecholaminergic fibers), and the sympathetic neurotransmitter norepinephrine (NE) in skin biopsies from control participants and patients with PD, the related LBD pure autonomic failure (PAF), the non-LBD synucleinopathy multiple system atrophy (MSA), or neurologic postacute sequelae of severe acute respiratory syndrome coronavirus 2 (neuro-PASC).
View Article and Find Full Text PDFBrain Commun
December 2024
Division of Psychiatry, University College London, London W1T 7NF, UK.
Hyperphosphorylated tau accumulation is seen in the noradrenergic locus coeruleus from the earliest stages of Alzheimer's disease onwards and has been associated with symptoms of agitation. It is hypothesized that compensatory locus coeruleus-noradrenaline system overactivity and impaired emotion regulation could underlie agitation propensity, but to our knowledge this has not previously been investigated. A better understanding of the neurobiological underpinnings of agitation would help the development of targeted prevention and treatment strategies.
View Article and Find Full Text PDFPhysiol Rep
January 2025
Department of Cell Physiology, Kumamoto University, Kumamoto, Japan.
Transient receptor potential (TRP) channels with temperature sensitivities (thermo-TRPs) are involved in various physiological processes. Thermo-TRPs that detect temperature changes in peripheral sensory neurons possess indispensable functions in thermosensation, eliciting defensive behavior against noxious temperatures and driving autonomic/behavioral thermoregulatory responses to maintain body temperature in mammals. Moreover, most thermo-TRPs are functionally expressed in cells and tissues where the temperature is maintained at a constant core body temperature.
View Article and Find Full Text PDFNeurosci Biobehav Rev
January 2025
Sapienza University of Rome and CLN(2)S@Sapienza, Istituto Italiano di Tecnologia, Viale Regina Elena, 291, 00161, Rome, Italy; IRCCS, Santa Lucia Foundation, 00142 Rome, Italy.
This review examines the role of thermal interoception-awareness of body temperature-in shaping physiological states and its impact on physical, emotional, and social well-being. We describe the neural pathways and mechanisms involved in thermal processing and environmental heat exchange and how thermal interoception influences both autonomic and behavioral responses, contributing to survival and homeostasis. Additionally, the review highlights the significance of thermal signals and thermoregulation in determining higher-order cognitive and emotional functions, such as regulating sleep patterns and activity levels, monitoring physical well-being, regulating emotions, and even social interactions.
View Article and Find Full Text PDFInt J Legal Med
January 2025
Faculty of Medicine, Lucian Blaga University of Sibiu, Sibiu, 550169, Romania.
The burnout phenomenon is a subject of considerable interest due to its impact on both employee well-being and scientific inquiry. Workplace factors, both intrinsic and extrinsic, play a pivotal role in its development, often leading to job dissatisfaction and heightened burnout risk. Chronic stress and burnout induce significant dysregulation in the autonomic nervous system and hormonal pathways, alongside structural brain changes.
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