Search

Your search keyword '"Giousoh, Aminah"' showing total 8 results
Start Over Author "Giousoh, Aminah"
8 results on '"Giousoh, Aminah"'

1. Zebrafish models for nemaline myopathy reveal a spectrum of nemaline bodies contributing to reduced muscle function

Author

Sztal, Tamar E., Zhao, Mo, Williams, Caitlin, Oorschot, Viola, Parslow, Adam C., Giousoh, Aminah, Yuen, Michaela, Hall, Thomas E., Costin, Adam, Ramm, Georg, Bird, Phillip I., Busch-Nentwich, Elisabeth M., Stemple, Derek L., Currie, Peter D., Cooper, Sandra T., Laing, Nigel G., Nowak, Kristen J., and Bryson-Richardson, Robert J.

Published
2015
Full Text
View/download PDF

3. Investigating the role of brinp1 in zebrafish development

Author

Giousoh, Aminah

Subjects
embryonic structures, Uncategorized
Abstract

The Membrane Attack Complex - PerForin (MACPF) superfamily is known to have a diverse array of roles in immunity, embryonic development and neural function. BRINPs (Bone Morphogenetic Protein – Retinoic Acid Inducible Neural specific Proteins) are a family of three MACPF domain-containing proteins that are highly expressed during rodent neural development. Each contains a MACPF domain, but the remainder of the molecule has no significant sequence similarity to any other structurally or functionally characterised protein. BRINP orthologues are highly conserved across vertebrates, suggesting they perform essential functions. In rodents, all three Brinps are ubiquitously expressed in the brain at early embryonic stages, with Brinp1 most highly expressed. In postnatal to adult stages Brinp1 expression is restricted to the hippocampus, olfactory bulb, cerebellum and cerebral cortex. Only low expression of Brinp2 and Brinp3 is observed at these stages. In this study, the zebrafish animal model was used to study the function of brinps in the developing brain, and to determine the effect of disrupting their function. A bioinformatic approach was applied to explore the evolutionary relationships between BRINP family members. The locus arrangement of BRINP genes is conserved in mammals and teleosts. Full-length zebrafish orthologues were identified and cloned, and two additional brinp3 paralogues were discovered. Brinp gene expression in zebrafish was reminiscent of the expression in rodents. Expression was unrestricted in the brain at early stages (24 hpf) and localised to specific brain structures in older embryos (48-72 hpf). Expression was also observed in the primordial fin buds. In addition to the broad and overlapping expression pattern of all brinps, brinp1 was exclusively detected in the lateral line primordium. Through morpholino oligonucleotide gene silencing, loss of function of the brinp1 gene was examined and determined to result in pleiotropic defects in the developing embryo. At 48 hpf, embryos exhibited a curved body axis, aberrant neuromast formation and defects in axonal projections in the head and tail. Finally, this study has led to the identification of a pathway in which brinp1 may function. It was possible to reverse the effect of the curved tail and loss of neuromasts by 72 hpf using a Tyrosine kinase B (TrkB) receptor agonist, providing preliminary evidence that brinp1 functions in the BDNF/ TrkB receptor pathway.

Published
2017
Full Text
View/download PDF

7. Brinp1-/- mice exhibit autism-like behaviour, altered memory, hyperactivity and increased parvalbumin-positive cortical interneuron density.

Author

Berkowicz, Susan R., Featherby, Travis J., Zhengdong Qu, Giousoh, Aminah, Borg, Natalie A., Heng, Julian I., Whisstock, James C., and Bird, Phillip I.

Subjects
AUTISM research, PARVALBUMINS, KNOCKOUT mice
Abstract

Background: BMP/RA-inducible neural-specific protein 1 (Brinp1) is highly conserved in vertebrates, and continuously expressed in the neocortex, hippocampus, olfactory bulb and cerebellum from mid-embryonic development through to adulthood. Methods: Brinp1 knock-out (Brinp1-/-) mice were generated by Cre-recombinase-mediated removal of the third exon of Brinp1. Knock-out mice were characterised by behavioural phenotyping, immunohistochemistry and expression analysis of the developing and adult brain. Results: Absence of Brinp1 during development results in a behavioural phenotype resembling autism spectrum disorder (ASD), in which knock-out mice show reduced sociability and changes in vocalisation capacity. In addition, Brinp1-/- mice exhibit hyper-locomotor activity, have impaired short-term memory, and exhibit poor reproductive success. Brinp1-/- mice show increased density of parvalbumin-expressing interneurons in the adult mouse brain. Brinp1-/- mice do not show signs of altered neural precursor proliferation or increased apoptosis during late embryonic brain development. The expression of the related neuronal migration genes Astn1 and Astn2 is increased in the brains of Brinp1-/- mice, suggesting that they may ameliorate the effects of Brinp1 loss. Conclusions: Brinp1 plays an important role in normal brain development and function by influencing neuronal distribution within the cortex. The increased cortical PV-positive interneuron density and altered behaviour of Brinp1-/- mice resemble features of a subset of human neurological disorders; namely autism spectrum disorder (ASD) and the hyperactivity aspect of attention deficit hyperactivity disorder (ADHD). [ABSTRACT FROM AUTHOR]

Published
2016
Full Text
View/download PDF

8. Brinp1(-/-) mice exhibit autism-like behaviour, altered memory, hyperactivity and increased parvalbumin-positive cortical interneuron density.

Author

Berkowicz SR, Featherby TJ, Qu Z, Giousoh A, Borg NA, Heng JI, Whisstock JC, and Bird PI

Subjects
Animals, Attention Deficit Disorder with Hyperactivity metabolism, Attention Deficit Disorder with Hyperactivity pathology, Autism Spectrum Disorder metabolism, Behavior, Animal, Brain metabolism, Brain pathology, Cell Cycle Proteins, Disease Models, Animal, Female, Genotype, Glycoproteins genetics, Glycoproteins metabolism, Interneurons metabolism, Male, Memory, Short-Term, Mice, Mice, Inbred C57BL, Mice, Knockout, Motor Activity, Nerve Tissue Proteins deficiency, Nerve Tissue Proteins metabolism, Parvalbumins genetics, Parvalbumins metabolism, Phenotype, Real-Time Polymerase Chain Reaction, Vocalization, Animal, Autism Spectrum Disorder pathology, Nerve Tissue Proteins genetics
Abstract

Background: BMP/RA-inducible neural-specific protein 1 (Brinp1) is highly conserved in vertebrates, and continuously expressed in the neocortex, hippocampus, olfactory bulb and cerebellum from mid-embryonic development through to adulthood., Methods: Brinp1 knock-out (Brinp1(-/-)) mice were generated by Cre-recombinase-mediated removal of the third exon of Brinp1. Knock-out mice were characterised by behavioural phenotyping, immunohistochemistry and expression analysis of the developing and adult brain., Results: Absence of Brinp1 during development results in a behavioural phenotype resembling autism spectrum disorder (ASD), in which knock-out mice show reduced sociability and changes in vocalisation capacity. In addition, Brinp1(-/-) mice exhibit hyper-locomotor activity, have impaired short-term memory, and exhibit poor reproductive success. Brinp1(-/-) mice show increased density of parvalbumin-expressing interneurons in the adult mouse brain. Brinp1(-/-) mice do not show signs of altered neural precursor proliferation or increased apoptosis during late embryonic brain development. The expression of the related neuronal migration genes Astn1 and Astn2 is increased in the brains of Brinp1(-/-) mice, suggesting that they may ameliorate the effects of Brinp1 loss., Conclusions: Brinp1 plays an important role in normal brain development and function by influencing neuronal distribution within the cortex. The increased cortical PV-positive interneuron density and altered behaviour of Brinp1(-/-) mice resemble features of a subset of human neurological disorders; namely autism spectrum disorder (ASD) and the hyperactivity aspect of attention deficit hyperactivity disorder (ADHD).

Published
2016
Full Text
View/download PDF

Catalog

Books, media, physical & digital resources
See catalog results