Evidence map›Paper›PMID 41657285›Full record

ArticleJournal of sleep research2026

iSPHYNCS: Unsupervised Clustering in Questionnaires and Metadata Reveals Distinct Subtypes in the Narcolepsy Borderland.

Rafael Morand, Livia Fregolente, Julia van der Meer, Elena S Wenz, Annina Helmy, Lorenzo Brigato, Jan D Warncke, Kseniia Zub, Ramin Khatami, Zhongxing Zhang and 10 more

Registry-linked trialAbstract readMulticenter Study
In one paragraph

Article in Journal of sleep research, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04330963 (International Swiss Primary Hypersomnolence and Narcolepsy Cohort Study), which is not on this map. Cited by 1 paper, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed, 1 pooled it
–field-weighted citation impact
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.

NCT04330963 unknown statusnot on this map

International Swiss Primary Hypersomnolence and Narcolepsy Cohort Study

Typeobservational_patient_registrySponsorInsel Gruppe AG, University Hospital BernRan2020 to 2026Enrolled600ConditionsNarcolepsy, Idiopathic Hypersomnia, Hypersomnolence Disorder
3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

20 authors.

Rafael MorandSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Livia FregolenteSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.ORCID 0000-0001-8793-0961
Julia van der MeerSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Elena S WenzSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.ORCID 0000-0002-6231-3358
Annina HelmySleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.ORCID 0009-0003-1221-7581
Lorenzo BrigatoARTORG Center for Biomedical Engineering Research, University of Bern, Bern, Switzerland.
Jan D WarnckeSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Kseniia ZubSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Ramin KhatamiSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Zhongxing ZhangClinic Barmelweid, Center for Sleep Medicine and Sleep Research, Barmelweid, Switzerland.
Sigrid von ManitiusDepartment of Neurology, Health Ostschweiz (HOCH), Kantonsspital St. Gallen St, St. Gallen, Switzerland.
Silvia MianoSleep and Epilepsy Center, Neurocenter of Southern Switzerland, Regional Hospital (EOC) of Lugano, Lugano, Switzerland.
Jens AckerZurzachCare, Clinic for Sleep Medicine, Bad Zurzach, Switzerland.
Mathias StrubCentre for Sleep Medicine Basel, Basel, Switzerland.
Ulf KallweitCenter for Narcolepsy and Hypersomnias, Professorship for Narcolepsy and Hypersomnolence Research, Department of Medicine, University Witten/Herdecke, Witten, Germany.ORCID 0000-0003-1975-6919
Gert Jan LammersSleep Wake Centre SEIN, Stichting Epilepsie Instellingen Nederland, Heemstede, the Netherlands.
Athina TzovaraSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Claudio L A BassettiSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.
Stavroula MougiakakouARTORG Center for Biomedical Engineering Research, University of Bern, Bern, Switzerland.
Markus H SchmidtSleep-Wake Epilepsy Center, NeuroTec, Department of Neurology, Inselspital, Bern University Hospital, University of Bern, Bern, Switzerland.

Funding

DLF Bern Biobank Call 2017Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung 320030_185362Schweizerischer Nationalfonds zur Förderung der Wissenschaftlichen Forschung 3203B_215721
6 · The paper itself

Abstract

The international Swiss Primary Hypersomnolence and Narcolepsy Cohort Study (iSPHYNCS) is a multicentre study aimed at identifying novel biomarkers for central disorders of hypersomnolence (CDH). We analysed questionnaires and metadata to uncover distinct clusters of participants and explore phenotypic variability within CDH. Data were collected from 227 patients with CDH and 33 healthy controls. Participants completed validated clinical questionnaires and study-specific questions addressing CDH-related symptoms such as excessive daytime sleepiness, fatigue, cataplexy, disrupted sleep, and sleep paralysis. Demographic metadata (age, gender, BMI) were included. After excluding participants with missing over 30% of data (n = 40), missing values were imputed using a multiple random forest algorithm. A robust clustering pipeline was employed: (1) random sampling of 60% of the dataset, (2) dimensionality reduction via UMAP, (3) K-means clustering, and (4) consensus clustering across 500 iterations. Post hoc analysis was performed to identify biomarkers in data not used for clustering. We identified four distinct clusters. One predominantly comprised healthy controls, while another primarily contained individuals with narcolepsy type 1 (NT1). Two clusters represented predominantly the narcolepsy borderland group (NBL), with one distinctly characterised by higher symptom severity and psychiatric comorbidities. The clustering pipeline produced reproducible results, with the NT1 and healthy control clusters serving as internal validation. The differentiation between the two NBL clusters aligns with prior studies, suggesting a possible NBL subtype marked by increased fatigue and psychiatric comorbidities. These findings emphasise the phenotypic heterogeneity of CDH and the potential for cluster-based approaches in management. Trial Registration: ClinicalTrials.gov identifier: NCT04330963.

Indexed as

Disorders of Excessive SomnolenceNarcolepsyAdultBiomarkersCluster AnalysisClustering AlgorithmsCohort StudiesFemaleHumansMaleMiddle AgedSurveys and QuestionnairesSwitzerlandBiomarkerscentral disorders of hypersomnolenceclustering analysisfatigueiSPHYNCSnarcolepsypsychiatric burdensleepiness

Identifiers

PMID41657285
PMCPMC13357946

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

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.