Evidence map›Paper›PMID 26707700›Full record

ArticleNeurobiology of disease2016

Gene co-expression networks shed light into diseases of brain iron accumulation.

Conceição Bettencourt, Paola Forabosco, Sarah Wiethoff, Moones Heidari, Daniel M Johnstone, Juan A Botía, Joanna F Collingwood, John Hardy, UK Brain Expression Consortium (UKBEC), Elizabeth A Milward and 2 more

Abstract readValidation Study
In one paragraph

Article in Neurobiology of disease, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.

0numbers the graph read from it
0cells of the map it votes in
18citing papers in PubMed
–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.

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

18 citing papers in PubMed.

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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

12 authors.

Conceição BettencourtDepartment of Molecular Neuroscience, UCL Institute of Neurology, London, UK; Department of Clinical and Experimental Epilepsy, UCL Institute of Neurology, London, UK. Electronic address: c.bettencourt@ucl.ac.uk.
Paola ForaboscoIstituto di Ricerca Genetica e Biomedica CNR, Cagliari, Italy.
Sarah WiethoffDepartment of Molecular Neuroscience, UCL Institute of Neurology, London, UK; Center for Neurology and Hertie Institute for Clinical Brain Research, Eberhard-Karls-University, Tübingen, Germany.
Moones HeidariSchool of Biomedical Sciences and Pharmacy, The University of Newcastle, Callaghan, NSW, Australia; Centre for Bioinformatics, Biomarker Discovery and Information-Based Medicine, The University of Newcastle, Callaghan, NSW, Australia.
Daniel M JohnstoneBosch Institute and Discipline of Physiology, University of Sydney, NSW, Australia;
Juan A BotíaDepartment of Medical and Molecular Genetics, King's College London, London, UK.
Joanna F CollingwoodSchool of Engineering, University of Warwick, Coventry, UK.
John HardyDepartment of Molecular Neuroscience, UCL Institute of Neurology, London, UK.
UK Brain Expression Consortium (UKBEC)
Elizabeth A MilwardSchool of Biomedical Sciences and Pharmacy, The University of Newcastle, Callaghan, NSW, Australia; Centre for Bioinformatics, Biomarker Discovery and Information-Based Medicine, The University of Newcastle, Callaghan, NSW, Australia.
Mina RytenDepartment of Molecular Neuroscience, UCL Institute of Neurology, London, UK; Department of Medical and Molecular Genetics, King's College London, London, UK.
Henry HouldenDepartment of Molecular Neuroscience, UCL Institute of Neurology, London, UK.

Funding

Medical Research Council G0802462Medical Research Council G0802760Medical Research Council G0901254Medical Research Council G1001253Medical Research Council G108/638Medical Research Council MR/J004758/1Wellcome Trust 104033Wellcome Trust WT089698Wellcome Trust WT093205MAWellcome Trust WT104033/Z/14/Z
6 · The paper itself

Abstract

Aberrant brain iron deposition is observed in both common and rare neurodegenerative disorders, including those categorized as Neurodegeneration with Brain Iron Accumulation (NBIA), which are characterized by focal iron accumulation in the basal ganglia. Two NBIA genes are directly involved in iron metabolism, but whether other NBIA-related genes also regulate iron homeostasis in the human brain, and whether aberrant iron deposition contributes to neurodegenerative processes remains largely unknown. This study aims to expand our understanding of these iron overload diseases and identify relationships between known NBIA genes and their main interacting partners by using a systems biology approach. We used whole-transcriptome gene expression data from human brain samples originating from 101 neuropathologically normal individuals (10 brain regions) to generate weighted gene co-expression networks and cluster the 10 known NBIA genes in an unsupervised manner. We investigated NBIA-enriched networks for relevant cell types and pathways, and whether they are disrupted by iron loading in NBIA diseased tissue and in an in vivo mouse model. We identified two basal ganglia gene co-expression modules significantly enriched for NBIA genes, which resemble neuronal and oligodendrocytic signatures. These NBIA gene networks are enriched for iron-related genes, and implicate synapse and lipid metabolism related pathways. Our data also indicates that these networks are disrupted by excessive brain iron loading. We identified multiple cell types in the origin of NBIA disorders. We also found unforeseen links between NBIA networks and iron-related processes, and demonstrate convergent pathways connecting NBIAs and phenotypically overlapping diseases. Our results are of further relevance for these diseases by providing candidates for new causative genes and possible points for therapeutic intervention.

Indexed as

TranscriptomeAdolescentAgedAged, 80 and overAnimalsBasal GangliaChildChild, PreschoolDisease Models, AnimalFemaleGene Expression ProfilingHemochromatosis ProteinHistocompatibility Antigens Class IHumansInfantIronHemochromatosis ProteinHfe protein, mouseHistocompatibility Antigens Class IIronMembrane ProteinsReceptors, TransferrinTFR2 protein, mouseHuman brainIron metabolismNBIAWGCNAWhole-transcriptome analysis

Identifiers

PMID26707700
PMCPMC4731015

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Registered trials

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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.