Evidence map›Paper›PMID 38165365›Full record

ArticleNeurology2024

Role of Daytime Continuous Polysomnography in the Diagnosis of Pediatric Narcolepsy Type 1.

Fabio Pizza, Luca Vignatelli, Stefano Vandi, Corrado Zenesini, Francesco Biscarini, Christian Franceschini, Elena Antelmi, Francesca Ingravallo, Emmanuel Mignot, Oliviero Bruni and 4 more

Erratum issuedOpen access · hybridAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Neurology, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
3.2field-weighted citation impact, top 8% of its field
1 · What the graph read from it

What it found

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

5 citing papers in PubMed, 18 citations in OpenAlex.

  1. Article
  2. Article
  3. Review
  4. Narcolepsy and rapid eye movement sleep.Journal of sleep research · 2025
    Review
  5. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

14 authors at 1 institution in 1 country.

Fabio PizzaFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Luca VignatelliFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0002-9051-7091
Stefano VandiFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Corrado ZenesiniFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Francesco BiscariniFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0001-9756-6470
Christian FranceschiniFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0002-9307-5209
Elena AntelmiFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Francesca IngravalloFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Emmanuel MignotFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0002-6928-5310
Oliviero BruniFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0003-2207-1398
Lino NobiliFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0001-9317-5405
Pierangelo VeggiottiFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0003-2851-3441
Raffaele FerriFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.ORCID 0000-0001-6937-3065
Giuseppe PlazziFrom the Department of Biomedical and Neuromotor Sciences (DIBINEM) (F.P., S.V., F.B.), University of Bologna; IRCCS Istituto delle Scienze Neurologiche di Bologna (F.P., L.V., S.V., C.Z., G.P.); Department of Medicine and Surgery (C.F.), University of Parma; Neurology Unit (E.A.), Movement Disorders Division, Department of Neurosciences, Biomedicine and Movement Sciences, University of Verona; Department of Medical and Surgical Sciences (DIMEC) (F.I.), University of Bologna, Italy; Tanford University Center for Sleep Sciences (E.M.), Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Palo Alto, CA; Department of Developmental and Social Psychology (O.B.), Sapienza University, Rome; IRCCS Istituto Giannina Gaslini (L.N.), Genoa; Dipartimento di Neuroscienze, Riabilitazione, Oftalmologia, Genetica e Scienze Materno-Infantili (L.N.), DINOGMI, University of Genoa; University of Milan (P.V.), Milan; Clinical Neurophysiology Research Unit (R.F.), Oasi Research Institute-IRCCS, Troina; and Department of Biomedical, Metabolic and Neural Sciences (G.P.), University of Modena and Reggio-Emilia, Italy.
Istituto delle Scienze Neurologiche di Bologna · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND AND

objectivesNarcolepsy type 1 (NT1) is still largely underdiagnosed or diagnosed too late in children. Difficulties in obtaining rapid and reliable diagnostic evaluations of the condition in clinical practice partially explain this problem. Predictors of NT1 include cataplexy and sleep-onset REM periods (SOREMPs), documented during nocturnal polysomnography (N-PSG) or through the multiple sleep latency test (MSLT), although low CSF hypocretin-1 (CSF hcrt-1) is the definitive biological disease marker. Obtaining reliable MSLT results is not always feasible in children; therefore, this study aimed to validate daytime continuous polysomnography (D-PSG) as an alternative diagnostic tool.

methodsTwo hundred consecutive patients aged younger than 18 years (112 with NT1; 25 with other hypersomnias, including narcolepsy type 2 and idiopathic hypersomnia; and 63 with subjective excessive daytime sleepiness) were randomly split into 2 groups: group 1 (n = 133) for the identification of diagnostic markers and group 2 (n = 67) for the validation of the detected markers. The D-PSG data collected included the number of spontaneous naps, total sleep time, and the number of daytime SOREMPs (d-SOREMP). D-PSG data were tested against CSF hcrt-1 deficiency (NT1 diagnosis) as the gold standard using receiver operating characteristic (ROC) curve analysis in group 1. ROC diagnostic performances of single and combined D-PSG parameters were tested in group 1 and validated in group 2.

resultsIn group 1, the areas under the ROC curve (AUCs) were 0.91 (95% CI 0.86-0.96) for d-SOREMPs, 0.81 (95% CI 0.74-0.89) for the number of spontaneous naps, and 0.70 (95% CI 0.60-0.79) for total sleep time. A d-SOREMP count ≥1 (sensitivity of 95% and specificity of 72%), coupled with a diurnal total sleep time above 60 minutes (sensitivity of 89% and specificity of 91%), identified NT1 in group 1 with high reliability (area under the ROC curve of 0.93, 95% CI 0.88-0.97). These results were confirmed in the validation group with an AUC of 0.88 (95% CI 0.79-0.97). DISCUSSION: D-PSG recording is an easily performed, cost-effective, and reliable tool for identifying NT1 in children. Further studies should confirm its validity with home D-PSG monitoring. These alternative procedures could be used to confirm NT1 diagnosis and curtail diagnostic delay.

Indexed as

Disorders of Excessive SomnolenceNarcolepsyAgedChildDelayed DiagnosisHumansPolysomnographyReproducibility of Results

Identifiers

PMID38165365
PMCPMC10834121
OpenAlexW4389763499

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