A Case of Congenital Myasthenia Gravis due to ChAT Gene Mutation in a Term Neonate: A Case Report.

AJP Rep

Department of Neonatology, Corewell Health East Royal Oak, Royal Oak, Michigan.

Published: July 2024

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.

Download full-text PDF

Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC11259492PMC
http://dx.doi.org/10.1055/a-2363-6319DOI Listing

Publication Analysis

Top Keywords

chat gene
16
congenital myasthenia
8
myasthenia gravis
8
term neonate
8
gene mutations
8
respiratory distress
8
family history
8
genetic testing
8
case congenital
4
chat
4

Similar Publications

Identification of candidate genes involved in Zika virus-induced reversible paralysis of mice.

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 PDF

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 PDF

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.

View Article and Find Full Text PDF

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 PDF

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).

View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!