Development and validation of a predictive model of drug-resistant genetic generalized epilepsy.

Adolescent Adult Age of Onset Case-Control Studies Child Child, Preschool Drug Resistant Epilepsy / diagnosis Epilepsy, Generalized / diagnosis Female Humans Longitudinal Studies Male Models, Statistical Young Adult

Journal

Neurology
ISSN: 1526-632X
Titre abrégé: Neurology
Pays: United States
ID NLM: 0401060

Informations de publication

Date de publication:
13 10 2020
Historique:
received: 24 09 2019
accepted: 15 05 2020
pubmed: 8 8 2020
medline: 15 12 2020
entrez: 8 8 2020
Statut: ppublish

Résumé

To develop and validate a clinical prediction model for antiepileptic drug (AED)-resistant genetic generalized epilepsy (GGE). We performed a case-control study of patients with and without drug-resistant GGE, nested within ongoing longitudinal observational studies of AED response at 2 tertiary epilepsy centers. Using a validation dataset, we tested the predictive performance of 3 candidate models, developed from a training dataset. We then tested the candidate models' predictive ability on an external testing dataset. Of 5,189 patients in the ongoing longitudinal study, 121 met criteria for AED-resistant GGE and 468 met criteria for AED-responsive GGE. There were 66 patients with GGE in the external dataset, of whom 17 were cases. Catamenial epilepsy, history of a psychiatric condition, and seizure types were strongly related with drug-resistant GGE case status. Compared to women without catamenial epilepsy, women with catamenial epilepsy had about a fourfold increased risk for AED resistance. The calibration of 3 models, assessing the agreement between observed outcomes and predictions, was adequate. Discriminative ability, as measured with area under the receiver operating characteristic curve (AUC), ranged from 0.58 to 0.65. Catamenial epilepsy, history of a psychiatric condition, and the seizure type combination of generalized tonic clonic, myoclonic, and absence seizures are negative prognostic factors of drug-resistant GGE. The AUC of 0.6 is not consistent with truly effective separation of the groups, suggesting other unmeasured variables may need to be considered in future studies to improve predictability.

Identifiants

DOI: 10.1212/WNL.0000000000010597 PMID: 32759205 PMC: PMC7713754
pubmed: 32759205
pii: WNL.0000000000010597
doi: 10.1212/WNL.0000000000010597
pmc: PMC7713754
doi:

Types de publication

Journal Article Observational Study Research Support, N.I.H., Extramural Validation Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

e2150-e2160

Subventions

Organisme : NCATS NIH HHS
ID : UL1 TR001863
Pays : United States
Organisme : NINDS NIH HHS
ID : K23 NS054981
Pays : United States

Investigateurs

Jane Adcock (J)
Danielle Andrade (D)
Gianpiero Cavalleri (G)
Daniel Costello (D)
Normal Delanty (N)
Patricia Dugan (P)
David Goldstein (D)
Patrick Kwan (P)
Fabio Nascimento (F)
Terence O'Brien (T)
Rod Radthke (R)
Philip Smith (P)
Rhys Thomas (R)

Informations de copyright

© 2020 American Academy of Neurology.

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Auteurs

Hyunmi Choi (H)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ. hc323@columbia.edu.

Kamil Detyniecki (K)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.
ORCID: 0000-0002-8334-1403

Carl Bazil (C)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Suzanne Thornton (S)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Peter Crosta (P)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Hatem Tolba (H)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Manahil Muneeb (M)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Lawrence J Hirsch (LJ)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Erin L Heinzen (EL)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Arjune Sen (A)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Chantal Depondt (C)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.

Piero Perucca (P)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.
ORCID: 0000-0002-7855-7066

Gary A Heiman (GA)

From the Department of Neurology (H.C., C.B., P.C., M.M.) and Institute for Genomic Medicine (E.L.H.), Columbia University Medical Center, New York, NY; Department of Neurology (K.D.), University of Miami, FL; Department of Statistics and Biostatistics (S.T.), Rutgers University, Piscataway, NJ; Department of Neurology (H.T., L.J.H.), Yale University, New Haven, CT; Nuffield Department of Clinical Neurosciences (A.S.), NIHR Biomedical Research Centre, University of Oxford, UK; Department of Neurology (C.D.), Free University of Brussels, Belgium; Department of Neuroscience (P.P.), Monash University; Departments of Medicine and Neurology (P.P.), The Royal Melbourne Hospital, The University of Melbourne; Department of Neurology (P.P.), Alfred Health, Melbourne, Australia; and Department of Genetics (G.A.H.), Human Genetics Institute of New Jersey, Rutgers, The State University of New Jersey, Piscataway, NJ.
ORCID: 0000-0001-5859-0259

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Classifications MeSH

questionsmedicales.fr › Personnes › Tranches d'âge › Adolescent › Development and validation of a predictive...
questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Development and validation of a predictive...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Facteurs épidémiologiques › Facteurs âges › Âge de début › Development and validation of a predictive...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études cas-témoins › Development and validation of a predictive...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Development and validation of a predictive...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Enfant d'âge préscolaire › Development and validation of a predictive...
questionsmedicales.fr › Maladies du système nerveux › Maladies du système nerveux central › Encéphalopathies › Épilepsie › Épilepsie pharmacorésistante › Development and validation of a predictive...
questionsmedicales.fr › Maladies du système nerveux › Maladies du système nerveux central › Encéphalopathies › Épilepsie › Épilepsie généralisée › Development and validation of a predictive...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Development and validation of a predictive...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Études longitudinales › Development and validation of a predictive...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Statistiques comme sujet › Modèles statistiques › Development and validation of a predictive...
questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Jeune adulte › Development and validation of a predictive...