Preliminary assessment of the reliability and validity of infrared skin temperature measurements in Rett syndrome.
Behavioural measurement methods
Behavioural phenotypes
Methodology in research
Rett syndrome
Treatment and services
Journal
Journal of intellectual disability research : JIDR
ISSN: 1365-2788
Titre abrégé: J Intellect Disabil Res
Pays: England
ID NLM: 9206090
Informations de publication
Date de publication:
04 2023
04 2023
Historique:
revised:
02
12
2022
received:
06
06
2022
accepted:
04
01
2023
pmc-release:
01
04
2024
pubmed:
7
2
2023
medline:
7
3
2023
entrez:
6
2
2023
Statut:
ppublish
Résumé
As clinical trials for Rett syndrome are underway, there is a need to validate potential supplemental outcome measures that reflect important signs and symptoms. Autonomic dysfunction, particularly vasomotor dysfunction, is one potential area for which biomarkers could be developed. In the current study, infrared thermal images of hands and feet from 26 females with Rett syndrome (aged 62 months to 39 years), and 17 females without known intellectual, genetic or neurological disorders (aged 55 months to 39 years) were collected. Between-group differences in skin temperature, and temporal stability of skin temperature measures in the Rett syndrome group, and relationships between skin temperature measures and parent-reported and researcher-evaluated indicators of autonomic dysfunction were evaluated. Between-group differences showed lower hand and foot temperatures in the Rett syndrome group. Hand temperature measurements were stable over time and were moderately correlated with parent-reported autonomic symptoms. Foot temperature measurements were more variable than hand temperatures but showed stronger correlations with parent-reported autonomic symptoms. The results provide preliminary support for the reliability and validity of hand and foot skin temperature measures in Rett syndrome. Additional research is needed to replicate these results and evaluate the temporal stability of these measures over shorter time scales.
Sections du résumé
BACKGROUND
As clinical trials for Rett syndrome are underway, there is a need to validate potential supplemental outcome measures that reflect important signs and symptoms. Autonomic dysfunction, particularly vasomotor dysfunction, is one potential area for which biomarkers could be developed.
METHODS
In the current study, infrared thermal images of hands and feet from 26 females with Rett syndrome (aged 62 months to 39 years), and 17 females without known intellectual, genetic or neurological disorders (aged 55 months to 39 years) were collected. Between-group differences in skin temperature, and temporal stability of skin temperature measures in the Rett syndrome group, and relationships between skin temperature measures and parent-reported and researcher-evaluated indicators of autonomic dysfunction were evaluated.
RESULTS
Between-group differences showed lower hand and foot temperatures in the Rett syndrome group. Hand temperature measurements were stable over time and were moderately correlated with parent-reported autonomic symptoms. Foot temperature measurements were more variable than hand temperatures but showed stronger correlations with parent-reported autonomic symptoms.
CONCLUSIONS
The results provide preliminary support for the reliability and validity of hand and foot skin temperature measures in Rett syndrome. Additional research is needed to replicate these results and evaluate the temporal stability of these measures over shorter time scales.
Identifiants
pubmed: 36744445
doi: 10.1111/jir.13010
pmc: PMC10251747
mid: NIHMS1868774
doi:
Types de publication
Journal Article
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
387-395Subventions
Organisme : NICHD NIH HHS
ID : R21 HD101075
Pays : United States
Organisme : NICHD NIH HHS
ID : R21HD101075-01
Pays : United States
Informations de copyright
© 2023 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd.
Références
J Biomed Inform. 2009 Apr;42(2):377-81
pubmed: 18929686
Mol Syndromol. 2012 Apr;2(3-5):113-127
pubmed: 22670134
Diagnostics (Basel). 2021 Mar 20;11(3):
pubmed: 33804657
J Neurodev Disord. 2017 Jun 12;9:14
pubmed: 28616097
Pediatr Neurol. 2015 Aug;53(2):169-72
pubmed: 26003587
J Am Podiatr Med Assoc. 2010 Jul-Aug;100(4):258-64
pubmed: 20660876
Ann Neurol. 2010 Dec;68(6):944-50
pubmed: 21154482
Sci Transl Med. 2016 Jan 13;8(321):321fs1
pubmed: 26764154
Eur J Paediatr Neurol. 2008 Mar;12(2):89-96
pubmed: 17662628
Sleep Med. 2013 Jan;14(1):5-11
pubmed: 21470909
Nat Rev Drug Discov. 2018 Dec 20;:
pubmed: 30936503
J Therm Biol. 2017 Oct;69:155-162
pubmed: 29037377
Brain Dev. 2001 Jul;23(4):208-11
pubmed: 11376997
Arch Dermatol. 2006 Aug;142(8):1085-6
pubmed: 16924077
Vasa. 2010 Feb;39(1):33-41
pubmed: 20186674
Nat Genet. 1999 Oct;23(2):185-8
pubmed: 10508514
Scoliosis. 2009 Oct 31;4:24
pubmed: 19878575
PLoS One. 2012;7(4):e35396
pubmed: 22523589
J Psychiatry Neurosci. 2019 Nov 08;45(3):150-181
pubmed: 31702122
Stem Cell Res Ther. 2015 May 09;6:92
pubmed: 25956259
Reumatologia. 2017;55(1):38-43
pubmed: 28386141
Curr Opin Neurol. 2021 Apr 1;34(2):197-205
pubmed: 33664205