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The following handle holds various files of this Leiden University dissertation:

http://hdl.handle.net/1887/72412

Author: Quinn, P.M.J.

Title: The Retinal Crumbs Complex: from animal models and retinal organoids to

therapy

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The Retinal Crumbs Complex:

From Animal Models and Retinal Organoids to Therapy

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The printing of this thesis was financially supported by Stichting Blindenhulp and the LUMC.

Copyright © Peter M. Quinn, 2019.

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The Retinal Crumbs Complex:

From Animal Models and Retinal Organoids to Therapy

Proefschrift

ter verkrijging van

de graad van Doctor aan de Universiteit Leiden, op gezag van Rector Magnificus prof.mr. C.J.J.M. Stolker,

volgens besluit van het College voor Promoties te verdedigen op woensdag 15 mei 2019

klokke 13:45 uur

door

Peter Martin John Quinn

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Prof. dr. C.J.F. Boon Dr. D.A. Baker Dr. L. van der Weerd

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1.2 Aims and outline of this thesis 30

CHAPTER 2 Targeted ablation of CRB1 and CRB2 in retinal progenitor cells

mimics Leber congenital amaurosis

PLoS Genetics 2013; 9:e1003976

39

CHAPTER 3 CRB2 in immature photoreceptors determines the

superior-inferior symmetry of the developing retina to maintain retinal structure and function

Human Molecular Genetics 2018; 27:3137-3153

79

CHAPTER 4 Loss of CRB2 in immature Müller glial cells adjusts a retinitis

pigmentosa phenotype due to loss of CRB1 into a Leber congenital amaurosis phenotype

Human Molecular Genetics 2019; 28:105-123

119

CHAPTER 5 Gene therapy into photoreceptors and Müller glial cells

restores retinal structure and function in CRB1 retinitis pigmentosa mouse models

Human Molecular Genetics 2015; 24:3104-3118

161

CHAPTER 6 6.1 NTPDase2 as a surface marker to isolate flow

cytometrically a Müller glial cell enriched population from dissociated neural retinae

Journal of Neuroscience and Neurosurgery 2018; 1 (4): 117

6.2 Transplantation of sorted Müller glial cells into the mouse retina

Journal of Neuroscience and Neurosurgery 2018; 1 (5): 122

201

212

CHAPTER 7 Human iPS-derived retinas recapitulate the fetal CRB1 CRB2

complex and human photoreceptors and Müller glial cells are targets of AAV5

A modified version is published in Stem Cell Reports, 2019.

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CHAPTER 8 Discussion

8.1 Retinogenesis of the Human Fetal Retina: An Apical polarity perspective

In preparation

8.2 Summary and Discussion

269

300

CHAPTER 9 9.1 Appendix I.

Production of iPS-Derived Human Retinal Organoids for Use in Transgene Expression Assays.

Methods in Molecular Biology 2018; 1715:261-273

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