Lama1 mutations lead to vitreoretinal blood vessel formation, persistence of fetal vasculature, and epiretinal membrane formation in miceReportar como inadecuado




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BMC Developmental Biology

, 11:60

Organogenesis

Abstract

BackgroundValuable insights into the complex process of retinal vascular development can be gained using models with abnormal retinal vasculature. Two such models are the recently described mouse lines with mutations in Lama1, an important component of the retinal internal limiting membrane ILM. These mutants have a persistence of the fetal vasculature of vitreous FVV but lack a primary retinal vascular plexus. The present study provides a detailed analysis of astrocyte and vascular development in these Lama1 mutants.

ResultsAlthough astrocytes and blood vessels initially migrate into Lama1 mutant retinas, both traverse the peripapillary ILM into the vitreous by P3. Once in the vitreous, blood vessels anastomose with vessels of the vasa hyaloidea propria, part of the FVV, and eventually re-enter the retina where they dive to form the inner and outer retinal capillary networks. Astrocytes continue proliferating within the vitreous to form a dense mesh that resembles epiretinal membranes associated with persistent fetal vasculature and proliferative vitreoretinopathy.

ConclusionsLama1 and a fully intact ILM are required for normal retinal vascular development. Mutations in Lama1 allow developing retinal vessels to enter the vitreous where they anastomose with vessels of the hyaloid system which persist and expand. Together, these vessels branch into the retina to form fairly normal inner retinal vascular capillary plexi. The Lama1 mutants described in this report are potential models for studying the human conditions persistent fetal vasculature and proliferative vitreoretinopathy.

List of AbbreviationsFVVfetal vasculature of vitreous

GFAPglial fibrillary acidic protein

GS isolectinGriffonia simplicifolia isolectin B4

ILMinternal limiting membrane

PASperiodic acid-Schiff

PFVpersistent fetal vasculature

PDGFRαplatelet-derived growth factor receptor alpha

Ppost natal day

PFAparaformaldehyde

PVRproliferative vitreoretinopathy

TBSTTris buffered saline containing 1% Triton X-100

TEMtransmission electron microscopy

VHPvasa hyaloidea propria

WTwild type

Electronic supplementary materialThe online version of this article doi:10.1186-1471-213X-11-60 contains supplementary material, which is available to authorized users.

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Autor: Malia M Edwards - D Scott McLeod - Rhonda Grebe - Céline Heng - Olivier Lefebvre - Gerard A Lutty

Fuente: https://link.springer.com/article/10.1186/1471-213X-11-60







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