000043378 001__ 43378
000043378 005__ 20240614124957.0
000043378 0247_ $$2doi$$a10.6083/bpxhc43378
000043378 037__ $$aETD
000043378 041__ $$aeng
000043378 245__ $$aStudying LCHADD chorioretinopathy using a novel LCHADD mouse model
000043378 260__ $$bOregon Health and Science University
000043378 269__ $$a2024-06-13
000043378 336__ $$aDissertation
000043378 502__ $$bPh.D.
000043378 502__ $$gMolecular & Medical Genetics
000043378 520__ $$aThe goal of this dissertation is to improve understanding and treatment for the chorioretinopathy seen in long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency (LCHADD) patients by examining the molecular mechanisms and testing potential treatments for LCHADD chorioretinopathy using a novel LCHADD mouse model. 
000043378 536__ $$oNational Eye Institute$$cR01EY032889
000043378 540__ $$fCC BY
000043378 542__ $$fIn copyright - single owner
000043378 650__ $$aMitochondrial Trifunctional Protein$$040282
000043378 650__ $$aGenetic Therapy$$028314
000043378 650__ $$aLong-Chain-3-Hydroxyacyl-CoA Dehydrogenase$$040280
000043378 650__ $$aRetinal Pigment Epithelium$$038211
000043378 6531_ $$aLCHADD
000043378 6531_ $$achorioretinopathy
000043378 6531_ $$agene addition
000043378 6531_ $$afatty acid oxidation
000043378 6531_ $$aRPE
000043378 691__ $$aSchool of Medicine$$041369
000043378 692__ $$aDepartment of Molecular and Medical Genetics$$041428
000043378 7001_ $$aBabcock, Shannon$$uOregon Health and Science University$$041354$$10000-0003-2365-2119
000043378 7201_ $$aGillingham, Melanie$$uOregon Health and Science University$$041354$$10000-0003-0897-8268$$ePI$$7Personal
000043378 789__ $$eIs part of$$w10.1016/j.ymgme.2024.108351$$2DOI
000043378 789__ $$eIs part of$$w10.1038/s42003-023-05268-1$$2DOI
000043378 792__ $$a<p>1. Babcock, S. J., et al. (2024). "A review of fatty acid oxidation disorder mouse models." Mol Genet Metab 142(1): 108351.</p>

<p> 2. Gaston G, Babcock S, Ryals R, Elizondo G, DeVine T, Wafai D, Packwood W, Holden S, Raber J, Lindner JR, Pennesi ME, Harding CO, Gillingham MB. A G1528C Hadha knock-in mouse model recapitulates aspects of human clinical phenotypes for long-chain 3-hydroxyacyl-CoA dehydrogenase deficiency. Commun Biol. 2023 Aug 29;6(1):890. doi: 10.1038/s42003-023-05268-1. PMID: 37644104; PMCID: PMC10465608.</p>

000043378 8564_ $$9fac2522c-907d-4dbd-8fbf-79ad4ccc68a3$$s27223095$$uhttps://digitalcollections.ohsu.edu/record/43378/files/Babcock.Shannon.2024.pdf
000043378 909CO $$ooai:digitalcollections.ohsu.edu:43378$$pstudent-work
000043378 980__ $$aTheses and Dissertations
000043378 981__ $$aPublished$$b2024-06-14