Idling for Decades: A European Study on Risk Factors Associated with the Delay Before a Narcolepsy Diagnosis

Result code in IS VaVaI

<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00023752%3A_____%2F22%3A43920913" target="_blank" >RIV/00023752:_____/22:43920913 - isvavai.cz</a>

Alternative codes found

RIV/00064165:_____/22:10444796 RIV/00216208:11110/22:10444796

Result on the web

<a href="https://www.dovepress.com/idling-for-decades-a-european-study-on-risk-factors-associated-with-th-peer-reviewed-fulltext-article-NSS" target="_blank" >https://www.dovepress.com/idling-for-decades-a-european-study-on-risk-factors-associated-with-th-peer-reviewed-fulltext-article-NSS</a>

DOI - Digital Object Identifier

<a href="http://dx.doi.org/10.2147/NSS.S359980" target="_blank" >10.2147/NSS.S359980</a>

Result language

angličtina

Original language name

Idling for Decades: A European Study on Risk Factors Associated with the Delay Before a Narcolepsy Diagnosis

Original language description

Purpose: Narcolepsy type-1 (NT1) is a rare chronic neurological sleep disorder with excessive daytime sleepiness (EDS) as usual first and cataplexy as pathognomonic symptom. Shortening the NT1 diagnostic delay is the key to reduce disease burden and related low quality of life. Here we investigated the changes of diagnostic delay over the diagnostic years (1990-2018) and the factors associated with the delay in Europe. Patients and Methods: We analyzed 580 NT1 patients (male: 325, female: 255) from 12 European countries using the European Narcolepsy Network database. We combined machine learning and linear mixed-effect regression to identify factors associated with the delay. Results: The mean age at EDS onset and diagnosis of our patients was 20.9 +/- 11.8 (mean +/- standard deviation) and 30.5 +/- 14.9 years old, respectively. Their mean and median diagnostic delay was 9.7 +/- 11.5 and 5.3 (interquartile range: 1.7-13.2 years) years, respectively. We did not find significant differences in the diagnostic delay over years in either the whole dataset or in individual countries, although the delay showed significant differences in various countries. The number of patients with short (&lt;= 2-year) and long (&gt;= 13-year) diagnostic delay equally increased over decades, suggesting that subgroups of NT1 patients with variable disease progression may co-exist. Younger age at cataplexy onset, longer interval between EDS and cataplexy onsets, lower cataplexy frequency, shorter duration of irresistible daytime sleep, lower daytime REM sleep propensity, and being female are associated with longer diagnostic delay. Conclusion: Our findings contrast the results of previous studies reporting shorter delay over time which is confounded by calendar year, because they characterized the changes in diagnostic delay over the symptom onset year. Our study indicates that new strategies such as increasing media attention/awareness and developing new biomarkers are needed to better detect EDS, cataplexy, and changes of nocturnal sleep in narcolepsy, in order to shorten the diagnostic interval.

Czech name

—

Czech description

—

Type

J_imp - Article in a specialist periodical, which is included in the Web of Science database

CEP classification

—

OECD FORD branch

30210 - Clinical neurology

Project

—

Continuities

V - Vyzkumna aktivita podporovana z jinych verejnych zdroju

Publication year

2022

Confidentiality

S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Name of the periodical

Nature and Science of Sleep

ISSN

1179-1608

e-ISSN

1179-1608

Volume of the periodical

14

Issue of the periodical within the volume

May

Country of publishing house

GB - UNITED KINGDOM

Number of pages

17

Pages from-to

1031-1047

UT code for WoS article

000810518700001

EID of the result in the Scopus database

2-s2.0-85131794714