dc.contributor.author | Boult, JKR | |
dc.contributor.author | Apps, JR | |
dc.contributor.author | Hölsken, A | |
dc.contributor.author | Hutchinson, JC | |
dc.contributor.author | Carreno, G | |
dc.contributor.author | Danielson, LS | |
dc.contributor.author | Smith, LM | |
dc.contributor.author | Bäuerle, T | |
dc.contributor.author | Buslei, R | |
dc.contributor.author | Buchfelder, M | |
dc.contributor.author | Virasami, AK | |
dc.contributor.author | Koers, A | |
dc.contributor.author | Arthurs, OJ | |
dc.contributor.author | Jacques, TS | |
dc.contributor.author | Chesler, L | |
dc.contributor.author | Martinez-Barbera, JP | |
dc.contributor.author | Robinson, SP | |
dc.date.accessioned | 2017-04-18T12:37:38Z | |
dc.date.issued | 2018-07-01 | |
dc.identifier.citation | Brain pathology (Zurich, Switzerland), 2018, 28 (4), pp. 475 - 483 | |
dc.identifier.issn | 1015-6305 | |
dc.identifier.uri | https://repository.icr.ac.uk/handle/internal/605 | |
dc.identifier.eissn | 1750-3639 | |
dc.identifier.doi | 10.1111/bpa.12525 | |
dc.description.abstract | To assess the clinical relevance of transgenic and patient-derived xenograft models of adamantinomatous craniopharyngioma (ACP) using serial magnetic resonance imaging (MRI) and high resolution post-mortem microcomputed tomography (μ-CT), with correlation with histology and human ACP imaging. The growth patterns and radiological features of tumors arising in Hesx1Cre/+ ;Ctnnb1lox(ex3)/+ transgenic mice, and of patient-derived ACP xenografts implanted in the cerebral cortex, were monitored longitudinally in vivo with anatomical and functional MRI, and by ex vivo μ-CT at study end. Pathological correlates with hematoxylin and eosin stained sections were investigated. Early enlargement and heterogeneity of Hesx1Cre/+ ;Ctnnb1lox(ex3)/+ mouse pituitaries was evident at initial imaging at 8 weeks, which was followed by enlargement of a solid tumor, and development of cysts and hemorrhage. Tumors demonstrated MRI features that recapitulated those of human ACP, specifically, T1 -weighted signal enhancement in the solid tumor component following Gd-DTPA administration, and in some animals, hyperintense cysts on FLAIR and T1 -weighted images. Ex vivo μ-CT correlated with MRI findings and identified smaller cysts, which were confirmed by histology. Characteristic histological features, including wet keratin and calcification, were visible on μ-CT and verified by histological sections of patient-derived ACP xenografts. The Hesx1Cre/+ ;Ctnnb1lox(ex3)/+ transgenic mouse model and cerebral patient-derived ACP xenografts recapitulate a number of the key radiological features of the human disease and provide promising foundations for in vivo trials of novel therapeutics for the treatment of these tumors. | |
dc.format | Print-Electronic | |
dc.format.extent | 475 - 483 | |
dc.language | eng | |
dc.language.iso | eng | |
dc.publisher | WILEY | |
dc.rights.uri | https://creativecommons.org/licenses/by/4.0 | |
dc.subject | Animals | |
dc.subject | Mice, Transgenic | |
dc.subject | Humans | |
dc.subject | Craniopharyngioma | |
dc.subject | Disease Models, Animal | |
dc.subject | Homeodomain Proteins | |
dc.subject | Repressor Proteins | |
dc.subject | Magnetic Resonance Imaging | |
dc.subject | Middle Aged | |
dc.subject | Male | |
dc.subject | beta Catenin | |
dc.subject | X-Ray Microtomography | |
dc.subject | Heterografts | |
dc.title | Preclinical transgenic and patient-derived xenograft models recapitulate the radiological features of human adamantinomatous craniopharyngioma. | |
dc.type | Journal Article | |
dcterms.dateAccepted | 2017-04-18 | |
rioxxterms.versionofrecord | 10.1111/bpa.12525 | |
rioxxterms.licenseref.uri | https://creativecommons.org/licenses/by/4.0 | |
rioxxterms.licenseref.startdate | 2018-07 | |
rioxxterms.type | Journal Article/Review | |
dc.relation.isPartOf | Brain pathology (Zurich, Switzerland) | |
pubs.issue | 4 | |
pubs.notes | Not known | |
pubs.organisational-group | /ICR | |
pubs.organisational-group | /ICR/Primary Group | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Cancer Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Cancer Therapeutics/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Clinical Studies | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Clinical Studies/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Molecular Pathology | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Molecular Pathology/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging/Pre-Clinical MRI | |
pubs.organisational-group | /ICR | |
pubs.organisational-group | /ICR/Primary Group | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Cancer Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Cancer Therapeutics/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Clinical Studies | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Clinical Studies/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Molecular Pathology | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Molecular Pathology/Paediatric Solid Tumour Biology and Therapeutics | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging | |
pubs.organisational-group | /ICR/Primary Group/ICR Divisions/Radiotherapy and Imaging/Pre-Clinical MRI | |
pubs.publication-status | Published | |
pubs.volume | 28 | |
pubs.embargo.terms | Not known | |
icr.researchteam | Paediatric Solid Tumour Biology and Therapeutics | |
icr.researchteam | Pre-Clinical MRI | |
dc.contributor.icrauthor | Boult, Jessica | |
dc.contributor.icrauthor | Chesler, Louis | |
dc.contributor.icrauthor | Robinson, Simon | |