Evidence of decreased adhesion between the neural retina and retinal pigmented epithelium of the Mitf(vit) (vitiligo) mutant mouse

N. Bora, D. Defoe, Sylvia B Smith

Research output: Contribution to journalArticle

7 Citations (Scopus)

Abstract

In order for the retina to function properly, photoreceptor cell outer segments must be in contact with the adjacent retinal pigmented epithelium (RPE). A mouse model homozygous for the vitiligo mutation of the microphthalmia (Mitf) gene manifests disruption of the outer segment/RPE interdigitation and demonstrates progressive loss of the photoreceptor cells. The mouse nevertheless has near normal levels of rhodopsin for many weeks and it is not known whether there is an in vivo loss of adhesion or whether the disruption is visible following tissue processing for histology. To assess this, a mechanical separation experiment was performed in which neural retinas were peeled free from the RPE and examined for the amount of pigment adherent to them. The peeling experiment indicated that control neural retinas retained significant amounts of adherent pigment at all ages examined. Neural retinas of mutant mice at age 2 weeks demonstrated adherent pigment, but older animals retained minimal pigment. Scanning electron microscopy indicated that the RPE cells of control mice were markedly damaged upon peeling and displayed different planes of cleavage, whereas those of mutants showed minimal cellular damage upon peeling, suggestive of decreased adhesion. A recombination experiment revealed that the mutant RPE/eyecup could reappose mutant and control retinas under in vitro conditions, suggesting that RPE fluid transport abilities were intact. The data provide the first direct experimental evidence that the Mitf(vit) mutant mouse has a naturally occurring retinal detachment and hence support its value as a model for studies of retina/RPE adhesion.

Original languageEnglish (US)
Pages (from-to)65-75
Number of pages11
JournalCell and Tissue Research
Volume295
Issue number1
DOIs
StatePublished - Jan 11 1999

Fingerprint

Vitiligo
Retina
Epithelium
Photoreceptor Cells
Retinal Photoreceptor Cell Outer Segment
Microphthalmos
Rhodopsin
Retinal Detachment
Electron Scanning Microscopy
Genetic Recombination
Histology
Mutation
Genes

Keywords

  • Microphthalmia transcription factor
  • Mouse
  • Retinal degeneration
  • Retinal pigment epithelium
  • Vitiligo (C57BL/6-Mitf(vit))

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Histology
  • Cell Biology

Cite this

Evidence of decreased adhesion between the neural retina and retinal pigmented epithelium of the Mitf(vit) (vitiligo) mutant mouse. / Bora, N.; Defoe, D.; Smith, Sylvia B.

In: Cell and Tissue Research, Vol. 295, No. 1, 11.01.1999, p. 65-75.

Research output: Contribution to journalArticle

@article{a52e42acfd284f2db5babbec7660ca29,
title = "Evidence of decreased adhesion between the neural retina and retinal pigmented epithelium of the Mitf(vit) (vitiligo) mutant mouse",
abstract = "In order for the retina to function properly, photoreceptor cell outer segments must be in contact with the adjacent retinal pigmented epithelium (RPE). A mouse model homozygous for the vitiligo mutation of the microphthalmia (Mitf) gene manifests disruption of the outer segment/RPE interdigitation and demonstrates progressive loss of the photoreceptor cells. The mouse nevertheless has near normal levels of rhodopsin for many weeks and it is not known whether there is an in vivo loss of adhesion or whether the disruption is visible following tissue processing for histology. To assess this, a mechanical separation experiment was performed in which neural retinas were peeled free from the RPE and examined for the amount of pigment adherent to them. The peeling experiment indicated that control neural retinas retained significant amounts of adherent pigment at all ages examined. Neural retinas of mutant mice at age 2 weeks demonstrated adherent pigment, but older animals retained minimal pigment. Scanning electron microscopy indicated that the RPE cells of control mice were markedly damaged upon peeling and displayed different planes of cleavage, whereas those of mutants showed minimal cellular damage upon peeling, suggestive of decreased adhesion. A recombination experiment revealed that the mutant RPE/eyecup could reappose mutant and control retinas under in vitro conditions, suggesting that RPE fluid transport abilities were intact. The data provide the first direct experimental evidence that the Mitf(vit) mutant mouse has a naturally occurring retinal detachment and hence support its value as a model for studies of retina/RPE adhesion.",
keywords = "Microphthalmia transcription factor, Mouse, Retinal degeneration, Retinal pigment epithelium, Vitiligo (C57BL/6-Mitf(vit))",
author = "N. Bora and D. Defoe and Smith, {Sylvia B}",
year = "1999",
month = "1",
day = "11",
doi = "10.1007/s004410051213",
language = "English (US)",
volume = "295",
pages = "65--75",
journal = "Cell and Tissue Research",
issn = "0302-766X",
publisher = "Springer Verlag",
number = "1",

}

TY - JOUR

T1 - Evidence of decreased adhesion between the neural retina and retinal pigmented epithelium of the Mitf(vit) (vitiligo) mutant mouse

AU - Bora, N.

AU - Defoe, D.

AU - Smith, Sylvia B

PY - 1999/1/11

Y1 - 1999/1/11

N2 - In order for the retina to function properly, photoreceptor cell outer segments must be in contact with the adjacent retinal pigmented epithelium (RPE). A mouse model homozygous for the vitiligo mutation of the microphthalmia (Mitf) gene manifests disruption of the outer segment/RPE interdigitation and demonstrates progressive loss of the photoreceptor cells. The mouse nevertheless has near normal levels of rhodopsin for many weeks and it is not known whether there is an in vivo loss of adhesion or whether the disruption is visible following tissue processing for histology. To assess this, a mechanical separation experiment was performed in which neural retinas were peeled free from the RPE and examined for the amount of pigment adherent to them. The peeling experiment indicated that control neural retinas retained significant amounts of adherent pigment at all ages examined. Neural retinas of mutant mice at age 2 weeks demonstrated adherent pigment, but older animals retained minimal pigment. Scanning electron microscopy indicated that the RPE cells of control mice were markedly damaged upon peeling and displayed different planes of cleavage, whereas those of mutants showed minimal cellular damage upon peeling, suggestive of decreased adhesion. A recombination experiment revealed that the mutant RPE/eyecup could reappose mutant and control retinas under in vitro conditions, suggesting that RPE fluid transport abilities were intact. The data provide the first direct experimental evidence that the Mitf(vit) mutant mouse has a naturally occurring retinal detachment and hence support its value as a model for studies of retina/RPE adhesion.

AB - In order for the retina to function properly, photoreceptor cell outer segments must be in contact with the adjacent retinal pigmented epithelium (RPE). A mouse model homozygous for the vitiligo mutation of the microphthalmia (Mitf) gene manifests disruption of the outer segment/RPE interdigitation and demonstrates progressive loss of the photoreceptor cells. The mouse nevertheless has near normal levels of rhodopsin for many weeks and it is not known whether there is an in vivo loss of adhesion or whether the disruption is visible following tissue processing for histology. To assess this, a mechanical separation experiment was performed in which neural retinas were peeled free from the RPE and examined for the amount of pigment adherent to them. The peeling experiment indicated that control neural retinas retained significant amounts of adherent pigment at all ages examined. Neural retinas of mutant mice at age 2 weeks demonstrated adherent pigment, but older animals retained minimal pigment. Scanning electron microscopy indicated that the RPE cells of control mice were markedly damaged upon peeling and displayed different planes of cleavage, whereas those of mutants showed minimal cellular damage upon peeling, suggestive of decreased adhesion. A recombination experiment revealed that the mutant RPE/eyecup could reappose mutant and control retinas under in vitro conditions, suggesting that RPE fluid transport abilities were intact. The data provide the first direct experimental evidence that the Mitf(vit) mutant mouse has a naturally occurring retinal detachment and hence support its value as a model for studies of retina/RPE adhesion.

KW - Microphthalmia transcription factor

KW - Mouse

KW - Retinal degeneration

KW - Retinal pigment epithelium

KW - Vitiligo (C57BL/6-Mitf(vit))

UR - http://www.scopus.com/inward/record.url?scp=0032895119&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=0032895119&partnerID=8YFLogxK

U2 - 10.1007/s004410051213

DO - 10.1007/s004410051213

M3 - Article

C2 - 9931354

AN - SCOPUS:0032895119

VL - 295

SP - 65

EP - 75

JO - Cell and Tissue Research

JF - Cell and Tissue Research

SN - 0302-766X

IS - 1

ER -