DNA methylation-based classification of malformations of cortical development in the human brain.


Journal

Acta neuropathologica
ISSN: 1432-0533
Titre abrégé: Acta Neuropathol
Pays: Germany
ID NLM: 0412041

Informations de publication

Date de publication:
01 2022
Historique:
received: 08 10 2021
accepted: 15 11 2021
revised: 26 10 2021
pubmed: 20 11 2021
medline: 8 3 2022
entrez: 19 11 2021
Statut: ppublish

Résumé

Malformations of cortical development (MCD) comprise a broad spectrum of structural brain lesions frequently associated with epilepsy. Disease definition and diagnosis remain challenging and are often prone to arbitrary judgment. Molecular classification of histopathological entities may help rationalize the diagnostic process. We present a retrospective, multi-center analysis of genome-wide DNA methylation from human brain specimens obtained from epilepsy surgery using EPIC 850 K BeadChip arrays. A total of 308 samples were included in the study. In the reference cohort, 239 formalin-fixed and paraffin-embedded (FFPE) tissue samples were histopathologically classified as MCD, including 12 major subtype pathologies. They were compared to 15 FFPE samples from surgical non-MCD cortices and 11 FFPE samples from post-mortem non-epilepsy controls. We applied three different statistical approaches to decipher the DNA methylation pattern of histopathological MCD entities, i.e., pairwise comparison, machine learning, and deep learning algorithms. Our deep learning model, which represented a shallow neuronal network, achieved the highest level of accuracy. A test cohort of 43 independent surgical samples from different epilepsy centers was used to test the precision of our DNA methylation-based MCD classifier. All samples from the test cohort were accurately assigned to their disease classes by the algorithm. These data demonstrate DNA methylation-based MCD classification suitability across major histopathological entities amenable to epilepsy surgery and age groups and will help establish an integrated diagnostic classification scheme for epilepsy-associated MCD.

Identifiants

pubmed: 34797422
doi: 10.1007/s00401-021-02386-0
pii: 10.1007/s00401-021-02386-0
pmc: PMC8732912
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

93-104

Informations de copyright

© 2021. The Author(s).

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Auteurs

Samir Jabari (S)

Department of Neuropathology, Affiliated Partner of the ERN EpiCARE, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.

Katja Kobow (K)

Department of Neuropathology, Affiliated Partner of the ERN EpiCARE, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany. katja.kobow@uk-erlangen.de.

Tom Pieper (T)

Center for Pediatric Neurology, Neurorehabilitation and Epileptology, Vogtareuth, Germany.

Till Hartlieb (T)

Center for Pediatric Neurology, Neurorehabilitation and Epileptology, Vogtareuth, Germany.
Research Institute, Rehabilitation, Transition, Palliation", PMU Salzburg, Salzburg, Austria.

Manfred Kudernatsch (M)

Center for Neurosurgery and Epilepsy Surgery, Schön Klinik Vogtareuth, Vogtareuth, Germany.
Research Institute, Rehabilitation, Transition, Palliation", PMU Salzburg, Salzburg, Austria.

Tilman Polster (T)

Department of Epileptology (Krankenhaus Mara), Medical School, Bielefeld University, Bielefeld, Germany.

Christian G Bien (CG)

Department of Epileptology (Krankenhaus Mara), Medical School, Bielefeld University, Bielefeld, Germany.

Thilo Kalbhenn (T)

Department of Neurosurgery - Epilepsy Surgery, Evangelisches Klinikum Bethel, Universitätsklinikum OWL, Bielefeld University, Bielefeld, Germany.

Matthias Simon (M)

Department of Neurosurgery - Epilepsy Surgery, Evangelisches Klinikum Bethel, Universitätsklinikum OWL, Bielefeld University, Bielefeld, Germany.

Hajo Hamer (H)

Department of Neurology, Epilepsy Center, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.

Karl Rössler (K)

Department of Neurosurgery, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.
Department of Neurosurgery, Medical University Vienna, Vienna, Austria.

Martha Feucht (M)

Department of Pediatrics and Adolescent Medicine, Affiliated Partner of the ERN EpiCARE, Medical University Vienna, Vienna, Austria.

Angelika Mühlebner (A)

Department of Pathology, University Medical Center Utrecht, Utrecht, The Netherlands.
Department of (Neuro) Pathology, Amsterdam UMC, Location AMC, Amsterdam, The Netherlands.

Imad Najm (I)

Charles Shor Epilepsy Center, Cleveland Clinic, Cleveland, OH, USA.
Department of Neurology, Cleveland Clinic, Cleveland, OH, USA.

José Eduardo Peixoto-Santos (JE)

Department of Neurology and Neurosurgery, Paulista Medical School, UNIFESP, Sao Paulo, Brazil.

Antonio Gil-Nagel (A)

Epilepsy Program, Hospital Ruber Internacional, Madrid, Spain.

Rafael Toledano Delgado (RT)

Epilepsy Program, Hospital Ruber Internacional, Madrid, Spain.

Angel Aledo-Serrano (A)

Epilepsy Program, Hospital Ruber Internacional, Madrid, Spain.

Yanghao Hou (Y)

Department of Neuropathology, German Cancer Research Center (DKFZ), Universitätsklinikum Heidelberg, and CCU Neuropathology, Heidelberg, Germany.
Institute of Pathology and Southwest Cancer Center, Southwest Hospital, Third Military Medical University (Army Medical University), Chongqing, China.

Roland Coras (R)

Department of Neuropathology, Affiliated Partner of the ERN EpiCARE, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.

Andreas von Deimling (A)

Department of Neuropathology, German Cancer Research Center (DKFZ), Universitätsklinikum Heidelberg, and CCU Neuropathology, Heidelberg, Germany.

Ingmar Blümcke (I)

Department of Neuropathology, Affiliated Partner of the ERN EpiCARE, Universitätsklinikum Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.

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