The Unfolded Protein Response in the Human Infant Brain and Dysregulation Seen in Sudden Infant Death Syndrome (SIDS).


Journal

Molecular neurobiology
ISSN: 1559-1182
Titre abrégé: Mol Neurobiol
Pays: United States
ID NLM: 8900963

Informations de publication

Date de publication:
May 2021
Historique:
received: 24 06 2020
accepted: 02 12 2020
pubmed: 9 1 2021
medline: 19 11 2021
entrez: 8 1 2021
Statut: ppublish

Résumé

Low orexin levels in the hypothalamus, and abnormal brainstem expression levels of many neurotransmitter and receptor systems in infants who died suddenly during a sleep period and diagnosed as sudden infant death syndrome (SIDS), may be linked to abnormal protein unfolding. We studied neuronal expression of the three unfolded protein response (UPR) pathways in the human infant brainstem, hypothalamus, and cerebellum: activating transcription factor 6 (ATF6), phosphorylated inositol-requiring enzyme 1 (IRE1), and phosphorylated protein-kinase (PKR)-like endoplasmic reticulum (ER) kinase (pPERK). Percentages of positively stained neurons were examined via immunohistochemistry and compared between SIDS (n = 28) and non-SIDS (n = 12) infant deaths. Further analysis determined the effects of the SIDS risk factors including cigarette smoke exposure, bed-sharing, prone sleeping, and an upper respiratory tract infection (URTI). Compared to non-SIDS, SIDS infants had higher ATF6 in the inferior olivary and hypoglossal nuclei of the medulla, higher pIRE1 in the dentate nucleus of the cerebellum, and higher pPERK in the cuneate nucleus and hypothalamus. Infants who were found prone had higher ATF6 in the hypoglossal and the locus coeruleus of the pons. Infants exposed to cigarette smoke had higher ATF6 in the vestibular and cuneate nuclei of the medulla. Infants who were bed-sharing had higher pPERK in the dorsal raphe nuclei of the pons and the Purkinje cells of the cerebellum. This study indicates that subgroups of SIDS infants, defined by risk exposure, had activation of the UPR in several nuclei relating to proprioception and motor control, suggesting that the UPR underlies the neuroreceptor system changes responsible for these physiological functions, leading to compromise in the pathogenesis of SIDS.

Identifiants

pubmed: 33417217
doi: 10.1007/s12035-020-02244-2
pii: 10.1007/s12035-020-02244-2
doi:

Substances chimiques

Activating Transcription Factor 6 0
Orexins 0
ERN1 protein, human EC 2.7.11.1
Protein Serine-Threonine Kinases EC 2.7.11.1
Endoribonucleases EC 3.1.-

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

2242-2255

Références

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Auteurs

Shannon Thomson (S)

Discipline of Medicine, Central Clinical School, Children's Hospital Westmead Clinical School, Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, 2006, Australia.

Karen A Waters (KA)

Discipline of Medicine, Central Clinical School, Children's Hospital Westmead Clinical School, Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, 2006, Australia.
Discipline of Child and Adolescent Health, Children's Hospital Westmead Clinical School, Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, 2006, Australia.

Rita Machaalani (R)

Discipline of Medicine, Central Clinical School, Children's Hospital Westmead Clinical School, Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, 2006, Australia. rita.machaalani@sydney.edu.au.
Discipline of Child and Adolescent Health, Children's Hospital Westmead Clinical School, Faculty of Medicine and Health, The University of Sydney, Sydney, NSW, 2006, Australia. rita.machaalani@sydney.edu.au.

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Classifications MeSH