Repeated measures of hypocretin-1 in Danish and Italian patients with narcolepsy and in controls
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Repeated measures of hypocretin-1 in Danish and Italian patients with narcolepsy and in controls. / Torstensen, Eva Wiberg; Haubjerg Østerby, Niels Christian; Pizza, Fabio; Plazzi, Giuseppe; Antelmi, Elena; Moresco, Monica; Mignot, Emmanuel; Jørgensen, Niklas Rye; Christensen, Gitte Lund; Wanscher, Benedikte; Kornum, Birgitte Rahbek; Jennum, Poul Jørgen.
I: Sleep Medicine, Bind 101, 2023, s. 213-220.Publikation: Bidrag til tidsskrift › Tidsskriftartikel › Forskning › fagfællebedømt
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TY - JOUR
T1 - Repeated measures of hypocretin-1 in Danish and Italian patients with narcolepsy and in controls
AU - Torstensen, Eva Wiberg
AU - Haubjerg Østerby, Niels Christian
AU - Pizza, Fabio
AU - Plazzi, Giuseppe
AU - Antelmi, Elena
AU - Moresco, Monica
AU - Mignot, Emmanuel
AU - Jørgensen, Niklas Rye
AU - Christensen, Gitte Lund
AU - Wanscher, Benedikte
AU - Kornum, Birgitte Rahbek
AU - Jennum, Poul Jørgen
N1 - Publisher Copyright: © 2022
PY - 2023
Y1 - 2023
N2 - Study objectives: The assay currently used worldwide to measure cerebrospinal fluid hypocretin-1 (CSF-hcrt-1) for diagnosing narcolepsy uses a competitive radioimmunoassay with polyclonal anti-hcrt-1 antibodies. This assay detects multiple hypocretin-1 immunoreactive species in the CSF that are all derived from full-length hcrt-1. We aimed to revalidate CSF-hcrt-1 cut-offs for narcolepsy type 1 (NT1) diagnosis and to evaluate temporal changes in CSF-hcrt-1 levels in patients suspected of having central hypersomnia. Method: We carried out a repeat lumbar puncture with a mean follow-up of 4.0 years, to measure CSF-hcrt-1 in patients suspected of having central hypersomnia in a follow-up study. Data from CSF samples of patients with NT1 and of controls without known hypersomnia, from the Italian–Stanford and Danish populations, were examined using a receiver-operating characteristic analysis. Results: The optimal CSF-hcrt-1 cut-offs for identifying NT1 were 129 pg/ml and 179 pg/ml for the Italian-Stanford and Danish populations, respectively. The sensitivity was 0.93–0.99 and the specificity was 1. Follow-up lumbar puncture measurements of CSF-hcrt-1 were obtained from 73 patients. 30 of 32 patients with low CSF-hcrt-1 levels continued to be categorized as low, with an unaltered diagnosis; two patients showed a marked increase in CSF-hcrt-1, attaining normal values at follow-up. One of these patients relapsed to low CSF-hcrt-1 after follow-up. All 41 patients with normal CSF-hcrt-1 at baseline had normal CSF-hcrt-1 at follow-up. Conclusion: CSF-hcrt-1 measurement can provide an accurate test for diagnosing NT1, although it is important to validate the CSF-hcrt-1 cut-off for specific testing locations. Stable CSF-hcrt-1 levels support the already established prognosis of narcolepsy as permanent once the disorder has fully developed.
AB - Study objectives: The assay currently used worldwide to measure cerebrospinal fluid hypocretin-1 (CSF-hcrt-1) for diagnosing narcolepsy uses a competitive radioimmunoassay with polyclonal anti-hcrt-1 antibodies. This assay detects multiple hypocretin-1 immunoreactive species in the CSF that are all derived from full-length hcrt-1. We aimed to revalidate CSF-hcrt-1 cut-offs for narcolepsy type 1 (NT1) diagnosis and to evaluate temporal changes in CSF-hcrt-1 levels in patients suspected of having central hypersomnia. Method: We carried out a repeat lumbar puncture with a mean follow-up of 4.0 years, to measure CSF-hcrt-1 in patients suspected of having central hypersomnia in a follow-up study. Data from CSF samples of patients with NT1 and of controls without known hypersomnia, from the Italian–Stanford and Danish populations, were examined using a receiver-operating characteristic analysis. Results: The optimal CSF-hcrt-1 cut-offs for identifying NT1 were 129 pg/ml and 179 pg/ml for the Italian-Stanford and Danish populations, respectively. The sensitivity was 0.93–0.99 and the specificity was 1. Follow-up lumbar puncture measurements of CSF-hcrt-1 were obtained from 73 patients. 30 of 32 patients with low CSF-hcrt-1 levels continued to be categorized as low, with an unaltered diagnosis; two patients showed a marked increase in CSF-hcrt-1, attaining normal values at follow-up. One of these patients relapsed to low CSF-hcrt-1 after follow-up. All 41 patients with normal CSF-hcrt-1 at baseline had normal CSF-hcrt-1 at follow-up. Conclusion: CSF-hcrt-1 measurement can provide an accurate test for diagnosing NT1, although it is important to validate the CSF-hcrt-1 cut-off for specific testing locations. Stable CSF-hcrt-1 levels support the already established prognosis of narcolepsy as permanent once the disorder has fully developed.
KW - Catalepsy
KW - Central hypersomnia
KW - Diagnostic-validation
KW - Orexin
KW - Radioimmunoassay
U2 - 10.1016/j.sleep.2022.10.004
DO - 10.1016/j.sleep.2022.10.004
M3 - Journal article
C2 - 36427467
AN - SCOPUS:85143167031
VL - 101
SP - 213
EP - 220
JO - Sleep Medicine
JF - Sleep Medicine
SN - 1389-9457
ER -
ID: 331639555