Congenital myasthenia gravis syndrome (CMS) is a rare genetic heterogeneous disorder due to abnormal neuromuscular transmission caused by several genes. CMS caused by choline acetyltransferase (ChAT) gene mutations have been reported in 53 cases since 2020, and only three of them have been identified as hemizygous mutations. A 1-day-old term neonate was transferred from the nursery to the neonatal intensive care unit on 10 L of oxygen by face mask due to respiratory distress. Family history includes two ChAT gene variants in the elder brother and carrier genes in both parents. She was transitioned to continuous positive airway pressure +5, and chest X-ray showed poor chest expansion. Sepsis workup was negative. She was intubated due to worsening respiratory distress 3 days later. She was started on pyridostigmine, and genetic testing was done, which confirmed two ChAT gene mutations present. The patient had a tracheostomy placed on day 50 and was discharged on day 94 of life on tracheostomy and G-tube, given feeding intolerance. This case illustrates the value of genetic testing in determining the type of CMS to guide management and the importance of obtaining family history to have such syndromes earlier in the differential diagnosis to initiate treatment promptly.
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http://dx.doi.org/10.1055/a-2363-6319 | DOI Listing |
Sci Rep
January 2025
Institute for Antiviral Research, Department of Animal, Dairy, and Veterinary Sciences, Utah State University, Logan, UT, 84321-5600, USA.
Zika virus (ZIKV) causes a variety of peripheral and central nervous system complications leading to neurological symptoms such as limb weakness. We used a mouse model to identify candidate genes potentially involved in causation or recovery from ZIKV-induced acute flaccid paralysis. Using Zikv and Chat chromogenic and fluorescence in situ RNA hybridization, electron microscopy, immunohistochemistry, and ZIKV RT-qPCR, we determined that some paralyzed mice had infected motor neurons, but motor neurons are not reduced in number and the infection was not present in all paralyzed mice; hence infection of motor neurons were not strongly correlated with paralysis.
View Article and Find Full Text PDFFront Cell Infect Microbiol
December 2024
Institut Pasteur de la Guadeloupe, Les Abymes, Guadeloupe, France.
The free-living amoeba (NF) causes a rare but lethal parasitic meningoencephalitis (PAM) in humans. Currently, this disease lacks effective treatments and the specific molecular mechanisms that govern NF pathogenesis and host brain response remain unknown. To address some of these issues, we sought to explore naturally existing virulence diversity within environmental NF isolates.
View Article and Find Full Text PDFJ Inflamm Res
December 2024
Department of Vascular Surgery, The First Affiliated Hospital of Chongqing Medical University, Chongqing, 400016, People's Republic of China.
Purpose: The relationship between macrophages and the progression of abdominal aortic aneurysms (AAA) remains unclear, and effective biomarkers are lacking. In this study, we elucidated the mechanism whereby macrophages promote AAA development and identified associated biomarkers, with the goal of developing new targeted therapies and improving patient outcomes.
Patients And Methods: Differential expression analysis, weighted gene co-expression network analysis, and single-cell analysis were used to identify macrophage-related genes in an AAA dataset.
Harefuah
December 2024
Maccabi Healthcare Services Sharon Region, Hebrew University Faculty of Medicine, and Medint Medical Intelligence, Tel Aviv, Israel.
Adrenoleukodystrophy is a genetic metabolic disorder characterized by a heterogeneous phenotype. Its severe form, known as cerebral adrenoleukodystrophy, involves unpredictable cerebral damage and progressive central nervous system deterioration. This rare condition became famous because of a Hollywood movie in which the Italian parents of a child with the condition supposedly discovered a medication for treating the condition.
View Article and Find Full Text PDFNeuroendocrinology
December 2024
School of Biomedical Sciences and Pharmacy, University of Newcastle, Callaghan, New South Wales, Australia.
Introduction: The efferent vestibular system (EVS) originates in brainstem efferent vestibular nuclei (EVN) and modifies afferent vestibular signals at their source, in peripheral vestibular organs. Recent evidence suggests that EVS is also involved in the development of motion sickness symptoms, including vertigo and nausea, but the underlying mechanism is unknown. One possible link between EVN and motion sickness symptoms is through the neuropeptide calcitonin gene-related peptide (CGRP).
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