Evidence map›Paper›PMID 28095967›Full record

Observational studyJournal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine2017

Intravenous Immunoglobulin Therapy in Pediatric Narcolepsy: A Nonrandomized, Open-Label, Controlled, Longitudinal Observational Study.

Michel Lecendreux, Johanna Berthier, Jennifer Corny, Olivier Bourdon, Claire Dossier, Christophe Delclaux

Open access · bronzeAbstract readObservational Study
In one paragraph

Observational study in Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
19citing papers in PubMed, 1 pooled it
2.1field-weighted citation impact, top 13% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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

19 citing papers in PubMed, 1 synthesis or guideline pooled it, 45 citations in OpenAlex.

  1. Treatment of central disorders of hypersomnolence: an American Academy of Sleep Medicine systematic review, meta-analysis, and GRADE assessment.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2021
    Pooled it
  2. Review
  3. Article
  4. Article
  5. Article
  6. Pediatric Narcolepsy-A Practical Review.Children (Basel, Switzerland) · 2022
    Review
  7. Article
  8. Review
  9. IgG Epitopes Processed and Presented by IgGJournal of immunology (Baltimore, Md. : 1950) · 2021
    Observational
  10. Article
  11. Review
  12. A practical guide to the pharmacological and behavioral therapy of Narcolepsy.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2021
    Review
  13. Immunotherapy in Narcolepsy.Current treatment options in neurology · 2020
    Review
  14. Recent advances in treatment for narcolepsy.Therapeutic advances in neurological disorders · 2019
    Review
  15. Narcolepsy Associated with Pandemrix Vaccine.Current neurology and neuroscience reports · 2018
    Review
  16. Article
  17. Review
  18. Cataplexy and Its Mimics: Clinical Recognition and Management.Current treatment options in neurology · 2017
    Review
  19. Is it Time for Immunotherapy Trials in Narcolepsy?Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2017
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

6 authors at 5 institutions in 2 countries.

Michel LecendreuxAP-HP, Pediatric Sleep Center, Hospital Robert-Debré, Paris, France.
Johanna BerthierCarol Davila University of Medicine and Pharmacy, Bucharest, Romania.
Jennifer CornyPharmacy Department, Hospital Robert-Debré, Paris, France.
Olivier BourdonPharmacy Department, Hospital Robert-Debré, Paris, France.
Claire DossierPediatric Nephrology Department, Hospital Robert-Debré, Paris, France.
Christophe DelclauxAP-HP, Pediatric Sleep Center, Hospital Robert-Debré, Paris, France.
Hôpital Robert-Debré · FRAssistance Publique – Hôpitaux de Paris · FRCarol Davila University of Medicine and Pharmacy · RODélégation Paris 5 · FRDélégation Paris 7 · FR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

STUDY

objectivesPrevious case reports of intravenous immunoglobulins (IVIg) in pediatric narcolepsy have shown contradictory results.

methodsThis was a nonrandomized, open-label, controlled, longitudinal observational study of IVIg use in pediatric narcolepsy with retrospective data collection from medical files obtained from a single pediatric national reference center for the treatment of narcolepsy in France. Of 56 consecutively referred patients with narcolepsy, 24 received IVIg (3 infusions administered at 1-mo intervals) in addition to standard care (psychostimulants and/or anticataplectic agents), and 32 continued on standard care alone (controls).

resultsFor two patients in each group, medical files were unavailable. Of the 22 IVIg patients, all had cerebrospinal fluid (CSF) hypocretin ≤ 110 pg/mL and were HLA-DQB1*06:02 positive. Of the 30 control patients, 29 were HLA-DQB1*06:02 positive and of those with available CSF measurements, all 12 had hypocretin ≤ 110 pg/mL. Compared with control patients, IVIg patients had shorter disease duration, shorter latency to sleep onset, and more had received H1N1 vaccination. Mean (standard deviation) follow-up length was 2.4 (1.1) y in the IVIg group and 3.9 (1.7) y in controls. In multivariate-adjusted linear mixed-effects analyses of change from baseline in Ullanlinna Narcolepsy Scale (UNS) scores, high baseline UNS, but not IVIg treatment, was associated with a reduction in narcolepsy symptoms. On time-to-event analysis, among patients with high baseline UNS scores, control patients achieved a UNS score < 14 (indicating remission) less rapidly than IVIg patients (adjusted hazard ratio 0.18; 95% confidence interval: 95% confidence interval: 0.03, 0.95; p = 0.043). Shorter or longer disease duration did not influence treatment response in any analysis.

conclusionsOverall, narcolepsy symptoms were not significantly reduced by IVIg. However, in patients with high baseline symptoms, a subset of IVIg-treated patients achieved remission more rapidly than control patients. COMMENTARY: A commentary on this article appears in this issue on page 363.

Indexed as

ChildFemaleFranceHumansImmunoglobulins, IntravenousLongitudinal StudiesMaleNarcolepsyRetrospective StudiesTreatment OutcomeImmunoglobulins, Intravenouscataplexychildimmunoglobulinimmunomodulationnarcolepsy

Identifiers

PMID28095967
PMCPMC5337592
OpenAlexW2581782077

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.