Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading

Severe disruption of brain iron homeostasis can cause fatal neurodegenerative disease, however debate surrounds the neurologic effects of milder, more common iron loading disorders such as hereditary hemochromatosis, which is usually caused by loss-of-function polymorphisms in the HFE gene. There is...

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Main Authors: Johnstone, D., Graham, Ross, Trinder, D., Delima, R., Riveros, C., Olynyk, John, Scott, R., Moscato, P., Milward, E.
Format: Journal Article
Published: Elsevier 2012
Subjects:
Online Access:http://hdl.handle.net/20.500.11937/40786
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author Johnstone, D.
Graham, Ross
Trinder, D.
Delima, R.
Riveros, C.
Olynyk, John
Scott, R.
Moscato, P.
Milward, E.
author_facet Johnstone, D.
Graham, Ross
Trinder, D.
Delima, R.
Riveros, C.
Olynyk, John
Scott, R.
Moscato, P.
Milward, E.
author_sort Johnstone, D.
building Curtin Institutional Repository
collection Online Access
description Severe disruption of brain iron homeostasis can cause fatal neurodegenerative disease, however debate surrounds the neurologic effects of milder, more common iron loading disorders such as hereditary hemochromatosis, which is usually caused by loss-of-function polymorphisms in the HFE gene. There is evidence from both human and animal studies that HFE gene variants may affect brain function and modify risks of brain disease. To investigate how disruption of HFE influences brain transcript levels, we used microarray and real-time reverse transcription polymerase chain reaction to assess the brain transcriptome in Hfe-/-mice relative to wildtype AKR controls (age 10 weeks, n≥4/group). The Hfe-/- mouse brain showed numerous significant changes in transcript levels (p<0.05) although few of these related to proteins directly involved in iron homeostasis. There were robust changes of at least 2-fold in levels of transcripts for prominent genes relating to transcriptional regulation (FBJ osteosarcoma oncogene Fos, early growth response genes), neurotransmission (glutamate NMDA receptor Grin1, GABA receptor Gabbr1) and synaptic plasticity and memory (calcium/calmodulin-dependent protein kinase IIα Camk2a). As previously reported for dietary iron-supplemented mice, there were altered levels of transcripts for genes linked to neuronal ceroid lipofuscinosis, a disease characterized by excessive lipofuscin deposition. Labile iron is known to enhance lipofuscin generation which may accelerate brain aging. The findings provide evidence that iron loading disorders can considerably perturb levels of transcripts for genes essential for normal brain function and may help explain some of the neurologic signs and symptoms reported in hemochromatosis patients.
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spelling curtin-20.500.11937-407862017-09-13T16:05:30Z Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading Johnstone, D. Graham, Ross Trinder, D. Delima, R. Riveros, C. Olynyk, John Scott, R. Moscato, P. Milward, E. Microarray Mouse model HFE Hemochromatosis Iron Severe disruption of brain iron homeostasis can cause fatal neurodegenerative disease, however debate surrounds the neurologic effects of milder, more common iron loading disorders such as hereditary hemochromatosis, which is usually caused by loss-of-function polymorphisms in the HFE gene. There is evidence from both human and animal studies that HFE gene variants may affect brain function and modify risks of brain disease. To investigate how disruption of HFE influences brain transcript levels, we used microarray and real-time reverse transcription polymerase chain reaction to assess the brain transcriptome in Hfe-/-mice relative to wildtype AKR controls (age 10 weeks, n≥4/group). The Hfe-/- mouse brain showed numerous significant changes in transcript levels (p<0.05) although few of these related to proteins directly involved in iron homeostasis. There were robust changes of at least 2-fold in levels of transcripts for prominent genes relating to transcriptional regulation (FBJ osteosarcoma oncogene Fos, early growth response genes), neurotransmission (glutamate NMDA receptor Grin1, GABA receptor Gabbr1) and synaptic plasticity and memory (calcium/calmodulin-dependent protein kinase IIα Camk2a). As previously reported for dietary iron-supplemented mice, there were altered levels of transcripts for genes linked to neuronal ceroid lipofuscinosis, a disease characterized by excessive lipofuscin deposition. Labile iron is known to enhance lipofuscin generation which may accelerate brain aging. The findings provide evidence that iron loading disorders can considerably perturb levels of transcripts for genes essential for normal brain function and may help explain some of the neurologic signs and symptoms reported in hemochromatosis patients. 2012 Journal Article http://hdl.handle.net/20.500.11937/40786 10.1016/j.brainres.2012.02.006 Elsevier restricted
spellingShingle Microarray
Mouse model
HFE
Hemochromatosis
Iron
Johnstone, D.
Graham, Ross
Trinder, D.
Delima, R.
Riveros, C.
Olynyk, John
Scott, R.
Moscato, P.
Milward, E.
Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title_full Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title_fullStr Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title_full_unstemmed Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title_short Brain transcriptome perturbations in the Hfe -/- mouse model of genetic iron loading
title_sort brain transcriptome perturbations in the hfe -/- mouse model of genetic iron loading
topic Microarray
Mouse model
HFE
Hemochromatosis
Iron
url http://hdl.handle.net/20.500.11937/40786