Universität zu Köln

Beck, Susanne C., Karlstetter, Marcus, Garrido, Marina Garcia, Feng, Yuxi, Dannhausen, Katharina, Muehlfriedel, Regine, Sothilingam, Vithiyanjali, Seebauer, Britta, Berger, Wolfgang, Hammes, Hans-Peter, Seeliger, Mathias W. and Langmann, Thomas (2018). Cystoid edema, neovascularization and inflammatory processes in the murine Norrin-deficient retina. Sci Rep, 8. LONDON: NATURE PUBLISHING GROUP. ISSN 2045-2322

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Abstract

Mutations in the Norrin (NDP) gene cause severe developmental blood vessel defects in the retina leading to congenital blindness. In the retina of Ndph-knockout mice only the superficial capillary network develops. Here, a detailed characterization of this mouse model at late stages of the disease using in vivo retinal imaging revealed cystoid structures that closely resemble the ovoid cysts in the inner nuclear layer of the human retina with cystoid macular edema (CME). In human CME an involvement of Muller glia cells is hypothesized. In Ndph-knockout retinae we could demonstrate that activated Muller cells were located around and within these cystoid spaces. In addition, we observed extensive activation of retinal microglia and development of neovascularization. Furthermore, ex vivo analyses detected extravasation of monocytic cells suggesting a breakdown of the blood retina barrier. Thus, we could demonstrate that also in the developmental retinal vascular pathology present in the Ndph-knockout mouse inflammatory processes are active and may contribute to further retinal degeneration. This observation delivers a new perspective for curative treatments of retinal vasculopathies. Modulation of inflammatory responses might reduce the symptoms and improve visual acuity in these diseases.

Item Type:	Journal Article
Creators:	Creators Email ORCID ORCID Put Code Beck, Susanne C. UNSPECIFIED UNSPECIFIED UNSPECIFIED Karlstetter, Marcus UNSPECIFIED UNSPECIFIED UNSPECIFIED Garrido, Marina Garcia UNSPECIFIED UNSPECIFIED UNSPECIFIED Feng, Yuxi UNSPECIFIED UNSPECIFIED UNSPECIFIED Dannhausen, Katharina UNSPECIFIED UNSPECIFIED UNSPECIFIED Muehlfriedel, Regine UNSPECIFIED UNSPECIFIED UNSPECIFIED Sothilingam, Vithiyanjali UNSPECIFIED UNSPECIFIED UNSPECIFIED Seebauer, Britta UNSPECIFIED UNSPECIFIED UNSPECIFIED Berger, Wolfgang UNSPECIFIED UNSPECIFIED UNSPECIFIED Hammes, Hans-Peter UNSPECIFIED UNSPECIFIED UNSPECIFIED Seeliger, Mathias W. UNSPECIFIED UNSPECIFIED UNSPECIFIED Langmann, Thomas UNSPECIFIED UNSPECIFIED UNSPECIFIED
URN:	urn:nbn:de:hbz:38-189373
DOI:	10.1038/s41598-018-24476-y
Journal or Publication Title:	Sci Rep
Volume:	8
Date:	2018
Publisher:	NATURE PUBLISHING GROUP
Place of Publication:	LONDON
ISSN:	2045-2322
Language:	English
Faculty:	Unspecified
Divisions:	Unspecified
Subjects:	no entry
Uncontrolled Keywords:	Keywords Language OPTICAL COHERENCE TOMOGRAPHY; MACULAR EDEMA; DISEASE GENE; MULLER CELLS; VASCULAR DEVELOPMENT; REACTIVE MICROGLIA; DEGENERATION; PATHOPHYSIOLOGY; ANGIOGENESIS; PATHOGENESIS Multiple languages Multidisciplinary Sciences Multiple languages
Refereed:	Yes
URI:	http://kups.ub.uni-koeln.de/id/eprint/18937