Characterization of rapid weight gain phenotype in children with narcolepsy.
children
narcolepsy
obesity
rapid weight gain
sleep
Journal
CNS neuroscience & therapeutics
ISSN: 1755-5949
Titre abrégé: CNS Neurosci Ther
Pays: England
ID NLM: 101473265
Informations de publication
Date de publication:
06 2022
06 2022
Historique:
revised:
20
01
2022
received:
16
10
2021
accepted:
21
01
2022
pubmed:
26
2
2022
medline:
6
5
2022
entrez:
25
2
2022
Statut:
ppublish
Résumé
To characterize the rapid weight gain (RWG) phenotype associated with the onset of childhood narcolepsy and to determine whether it could constitute a marker of severity of the disease. RWG was defined using the BMI z-score slope reported to one year (>0.67 SD) from symptom onset to disease diagnosis. We compared the clinical, metabolic, and sleep characteristics between patients with or without RWG at diagnosis. Pharmacological management, anthropometric, and clinical progression were also evaluated during the follow-up. A total of 84 de novo narcoleptic pediatric patients were included; their median age at diagnosis was 12.0 years; 59.5% boys, 90.5% cataplexy, and 98.7% HLA-DQB1*06:02, 57% had RWG profile. RWG patients were younger at diagnosis than non-RWG patients, despite a shorter diagnostic delay. They had a higher BMI z-score and a higher prevalence of obesity at diagnosis, but not at symptom onset, and higher adapted Epworth Sleepiness Scale and Insomnia Severity Index scores than non-RWG patients. No differences on nocturnal polysomnography and multiple sleep latency tests were found between groups at disease diagnosis. After a median follow-up of 5 years, RWG patients still had a higher BMI z-score and a higher prevalence of obesity despite benefiting from the same therapeutic management and displaying improvement in sleepiness and school difficulties. Narcoleptic RWG patients were younger, sleepier, and the prevalence of obesity was higher at diagnosis despite a shorter diagnostic delay than that of non-RWG patients. These patients had also a higher risk of developing a long-term obesity, despite a positive progression of their narcoleptic symptoms. RGW could then represent a maker of a more severe phenotype of childhood narcolepsy, which should inspire a prompt and more offensive management to prevent obesity and its complications.
Identifiants
pubmed: 35212159
doi: 10.1111/cns.13811
pmc: PMC9062543
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
829-841Informations de copyright
© 2022 The Authors. CNS Neuroscience & Therapeutics Published by John Wiley & Sons Ltd.
Références
Paediatr Respir Rev. 2018 Jan;25:14-18
pubmed: 28108192
Nat Commun. 2019 Feb 19;10(1):837
pubmed: 30783092
Sleep. 2005 Oct;28(10):1289-96
pubmed: 16295214
J Abnorm Child Psychol. 1978 Jun;6(2):221-36
pubmed: 670589
Nat Sci Sleep. 2021 May 11;13:557-577
pubmed: 34007229
Acta Paediatr. 2006 Aug;95(8):904-8
pubmed: 16882560
Obes Res. 2003 Mar;11(3):457-60
pubmed: 12634445
Sleep Med. 2001 Jul;2(4):297-307
pubmed: 11438246
Sleep. 2013 Feb 01;36(2):175-81
pubmed: 23372264
CNS Neurosci Ther. 2022 Jun;28(6):829-841
pubmed: 35212159
Rev Neurol (Paris). 2017 Jan - Feb;173(1-2):8-18
pubmed: 27865546
Pediatr Obes. 2012 Aug;7(4):284-94
pubmed: 22715120
Eur J Neurol. 2021 Sep;28(9):2815-2830
pubmed: 34173695
Nature. 2018 Oct;562(7725):63-68
pubmed: 30232458
Nat Med. 2000 Sep;6(9):991-7
pubmed: 10973318
J Clin Sleep Med. 2016 Dec 15;12(12):1649-1657
pubmed: 27707443
Sleep. 2006 Aug;29(8):1025-9
pubmed: 16944670
Pediatrics. 2004 Aug;114(2 Suppl 4th Report):555-76
pubmed: 15286277
J Clin Invest. 2010 Mar;120(3):713-9
pubmed: 20160349
Sleep. 2020 Jun 15;43(6):
pubmed: 31993661
J Clin Sleep Med. 2007 Dec 15;3(7):752
pubmed: 18198811
J Neurosci. 2002 Sep 1;22(17):7695-711
pubmed: 12196593
J Neurosci. 2009 Nov 18;29(46):14423-38
pubmed: 19923277
Eur J Endocrinol. 2012 Feb;166(2):301-6
pubmed: 22065856
CNS Neurosci Ther. 2020 Oct;26(10):1092-1102
pubmed: 32761857
BMJ. 2000 May 6;320(7244):1240-3
pubmed: 10797032
Obes Rev. 2005 May;6(2):143-54
pubmed: 15836465
Curr Treat Options Neurol. 2020 Jan 30;22(1):2
pubmed: 31997035
CNS Neurosci Ther. 2013 Jul;19(7):521-8
pubmed: 23574649
BMJ. 2000 Apr 8;320(7240):967-71
pubmed: 10753147
Am J Clin Nutr. 2003 Jun;77(6):1374-8
pubmed: 12791612
Arch Gen Psychiatry. 2008 Aug;65(8):924-32
pubmed: 18678797
Int J Pediatr Obes. 2010 Dec;5(6):458-60
pubmed: 20233144
J Autoimmun. 2015 Jun;60:20-31
pubmed: 25892508
Nat Rev Neurol. 2019 Sep;15(9):519-539
pubmed: 31324898
Sleep. 2007 Oct;30(10):1267-73
pubmed: 17969460
Neurology. 2001 Dec 11;57(11):2029-33
pubmed: 11739821
Arch Pediatr. 2006 Jan;13(1):104-10
pubmed: 16257193
Diabetologia. 2011 Jul;54(7):1841-52
pubmed: 21505958
Rev Port Pneumol (2006). 2016 Nov - Dec;22(6):331-336
pubmed: 27339391
Sleep. 2010 Nov;33(11):1457-64
pubmed: 21102987
Int J Obes Relat Metab Disord. 2003 Oct;27(10):1274-82
pubmed: 14513077
Pediatrics. 2002 Dec;110(6):e74
pubmed: 12456941
Sleep. 2009 Aug;32(8):979-83
pubmed: 19725248
Psychopharmacol Bull. 1985;21(4):995-8
pubmed: 4089116
Sleep. 2009 Jul;32(7):962-4
pubmed: 19639760
Lancet. 2005 Apr 16-22;365(9468):1415-28
pubmed: 15836891
Neurology. 2019 Sep 10;93(11):e1034-e1044
pubmed: 31405906
Sleep. 2012 Oct 01;35(10):1359-66
pubmed: 23024434
CNS Neurosci Ther. 2019 Mar;25(3):386-395
pubmed: 30225986
Neuropsychopharmacology. 2017 Jan;42(1):129-155
pubmed: 27510422
Am J Hum Genet. 2001 Mar;68(3):686-99
pubmed: 11179016
Nutr Metab Cardiovasc Dis. 2020 Apr 12;30(4):683-693
pubmed: 32008915
Curr Opin Neurobiol. 2011 Dec;21(6):897-903
pubmed: 21963829
Sleep Med. 2016 Sep;25:139-144
pubmed: 27823707