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Guide to preclinical models used to study the pathophysiology of idiopathic intracranial hypertension

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Alimajstorovic, Z, Westgate, CSJ, Jensen, RH, Eftekhari, S, Mitchell, J, Vijay, V, Seneviratne, SY, Mollan, SP & Sinclair, AJ 2020, 'Guide to preclinical models used to study the pathophysiology of idiopathic intracranial hypertension', Eye, vol. 34, no. 8, pp. 1321-1333. https://doi.org/10.1038/s41433-019-0751-1

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Alimajstorovic, Zerin ; Westgate, Connar S J ; Jensen, Rigmor H ; Eftekhari, Sajedeh ; Mitchell, James ; Vijay, Vivek ; Seneviratne, Senali Y ; Mollan, Susan P ; Sinclair, Alexandra J. / Guide to preclinical models used to study the pathophysiology of idiopathic intracranial hypertension. In: Eye. 2020 ; Vol. 34, No. 8. pp. 1321-1333.

Bibtex

@article{66ab0d542e484df68476781f427ec222,
title = "Guide to preclinical models used to study the pathophysiology of idiopathic intracranial hypertension",
abstract = "Idiopathic intracranial hypertension (IIH) is characterised by raised intracranial pressure (ICP) and papilloedema in the absence of an identifiable secondary cause typically occurring in young women with obesity. The impact is considerable with the potential for blindness, chronic disabling headaches, future risk of cardiovascular disease and marked healthcare utilisation. There have been marked advances in our understanding the pathophysiology of IIH including the role of androgen excess. Insight into pathophysiological underpinnings has arisen from astute clinical observations, studies, and an array of preclinical models. This article summarises the current literature pertaining to the pathophysiology of IIH. The current preclinical models relevant to gaining mechanistic insights into IIH are then discussed. In vitro and in vivo models which study CSF secretion and the effect of potentially pathogenic molecules have started to glean important mechanistic insights. These models are also useful to evaluate novel therapeutic targets to abrogate CSF secretion. Importantly, in vitro CSF secretion assays translate into relevant changes in ICP in vivo. Models of CSF absorption pertinent to IIH, are less well established but highly relevant and of future interest. There is no fully developed in vivo model of IIH but this remains an area of importance. Progress is being made to improve our understanding of the underlying aetiology in IIH including the characterisation of disease biomarkers and their mechanistic role in driving disease pathology. Preclinical models, used to evaluate IIH mechanisms are yielding important mechanistic insights. Further work to refine these techniques will provide translatable insights into disease aetiology.",
author = "Zerin Alimajstorovic and Westgate, {Connar S J} and Jensen, {Rigmor H} and Sajedeh Eftekhari and James Mitchell and Vivek Vijay and Seneviratne, {Senali Y} and Mollan, {Susan P} and Sinclair, {Alexandra J}",
year = "2020",
doi = "10.1038/s41433-019-0751-1",
language = "English",
volume = "34",
pages = "1321--1333",
journal = "Eye",
issn = "0950-222X",
publisher = "Nature Publishing Group",
number = "8",

}

RIS

TY - JOUR

T1 - Guide to preclinical models used to study the pathophysiology of idiopathic intracranial hypertension

AU - Alimajstorovic, Zerin

AU - Westgate, Connar S J

AU - Jensen, Rigmor H

AU - Eftekhari, Sajedeh

AU - Mitchell, James

AU - Vijay, Vivek

AU - Seneviratne, Senali Y

AU - Mollan, Susan P

AU - Sinclair, Alexandra J

PY - 2020

Y1 - 2020

N2 - Idiopathic intracranial hypertension (IIH) is characterised by raised intracranial pressure (ICP) and papilloedema in the absence of an identifiable secondary cause typically occurring in young women with obesity. The impact is considerable with the potential for blindness, chronic disabling headaches, future risk of cardiovascular disease and marked healthcare utilisation. There have been marked advances in our understanding the pathophysiology of IIH including the role of androgen excess. Insight into pathophysiological underpinnings has arisen from astute clinical observations, studies, and an array of preclinical models. This article summarises the current literature pertaining to the pathophysiology of IIH. The current preclinical models relevant to gaining mechanistic insights into IIH are then discussed. In vitro and in vivo models which study CSF secretion and the effect of potentially pathogenic molecules have started to glean important mechanistic insights. These models are also useful to evaluate novel therapeutic targets to abrogate CSF secretion. Importantly, in vitro CSF secretion assays translate into relevant changes in ICP in vivo. Models of CSF absorption pertinent to IIH, are less well established but highly relevant and of future interest. There is no fully developed in vivo model of IIH but this remains an area of importance. Progress is being made to improve our understanding of the underlying aetiology in IIH including the characterisation of disease biomarkers and their mechanistic role in driving disease pathology. Preclinical models, used to evaluate IIH mechanisms are yielding important mechanistic insights. Further work to refine these techniques will provide translatable insights into disease aetiology.

AB - Idiopathic intracranial hypertension (IIH) is characterised by raised intracranial pressure (ICP) and papilloedema in the absence of an identifiable secondary cause typically occurring in young women with obesity. The impact is considerable with the potential for blindness, chronic disabling headaches, future risk of cardiovascular disease and marked healthcare utilisation. There have been marked advances in our understanding the pathophysiology of IIH including the role of androgen excess. Insight into pathophysiological underpinnings has arisen from astute clinical observations, studies, and an array of preclinical models. This article summarises the current literature pertaining to the pathophysiology of IIH. The current preclinical models relevant to gaining mechanistic insights into IIH are then discussed. In vitro and in vivo models which study CSF secretion and the effect of potentially pathogenic molecules have started to glean important mechanistic insights. These models are also useful to evaluate novel therapeutic targets to abrogate CSF secretion. Importantly, in vitro CSF secretion assays translate into relevant changes in ICP in vivo. Models of CSF absorption pertinent to IIH, are less well established but highly relevant and of future interest. There is no fully developed in vivo model of IIH but this remains an area of importance. Progress is being made to improve our understanding of the underlying aetiology in IIH including the characterisation of disease biomarkers and their mechanistic role in driving disease pathology. Preclinical models, used to evaluate IIH mechanisms are yielding important mechanistic insights. Further work to refine these techniques will provide translatable insights into disease aetiology.

U2 - 10.1038/s41433-019-0751-1

DO - 10.1038/s41433-019-0751-1

M3 - Review

C2 - 31896803

VL - 34

SP - 1321

EP - 1333

JO - Eye

JF - Eye

SN - 0950-222X

IS - 8

ER -

ID: 59474455