Axon fasciculation defects and retinal dysplasias in mice lacking the immunoglobulin superfamily adhesion molecule BEN/ALCAM/SC1

Joshua A Weiner; Sonya J Koo; Stéphane Nicolas; Sandrine Fraboulet; Samuel L Pfaff; Olivier Pourquié; Joshua R Sanes

doi:10.1016/j.mcn.2004.06.005

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Axon fasciculation defects and retinal dysplasias in mice lacking the immunoglobulin superfamily adhesion molecule BEN/ALCAM/SC1

Journal article

Peer reviewed

Axon fasciculation defects and retinal dysplasias in mice lacking the immunoglobulin superfamily adhesion molecule BEN/ALCAM/SC1

Joshua A Weiner, Sonya J Koo, Stéphane Nicolas, Sandrine Fraboulet, Samuel L Pfaff, Olivier Pourquié and Joshua R Sanes

Molecular and cellular neurosciences, Vol.27(1), pp.59-69

09/2004

DOI: 10.1016/j.mcn.2004.06.005

PMID: 15345243

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Abstract

The immunoglobulin superfamily adhesion molecule BEN (other names include ALCAM, SC1, DM-GRASP, neurolin, and CD166) has been implicated in the control of numerous developmental and pathological processes, including the guidance of retinal and motor axons to their targets. To test hypotheses about BEN function, we disrupted its gene via homologous recombination and analyzed the resulting mutant mice. Mice lacking BEN are viable and fertile, and display no external morphological defects. Despite grossly normal trajectories, both motor and retinal ganglion cell axons fasciculated poorly and were occasionally misdirected. In addition, BEN mutant retinae exhibited evaginated or invaginated regions with photoreceptor ectopias that resembled the "retinal folds" observed in some human retinopathies. Together, these results demonstrate that BEN promotes fasciculation of multiple axonal populations and uncover an unexpected function for BEN in retinal histogenesis.

Optic Nerve - pathology

Cell Adhesion - genetics

Retinal Dysplasia - metabolism

Cells, Cultured

Cell Communication - genetics

Growth Cones - pathology

Optic Nerve - abnormalities

Retinal Ganglion Cells - metabolism

Choristoma - physiopathology

Motor Neurons - pathology

Mice, Knockout

Retinal Ganglion Cells - pathology

Motor Neurons - metabolism

Cell Differentiation - genetics

Retinal Dysplasia - genetics

Animals

Growth Cones - metabolism

Choristoma - metabolism

Mice

Retinal Dysplasia - physiopathology

Choristoma - genetics

Activated-Leukocyte Cell Adhesion Molecule - genetics

Optic Nerve - metabolism

Details

Title: Subtitle: Axon fasciculation defects and retinal dysplasias in mice lacking the immunoglobulin superfamily adhesion molecule BEN/ALCAM/SC1
Creators: Joshua A Weiner - Department of Anatomy and Neurobiology, Washington University School of Medicine, St. Louis, MO 63110, USA. joshua-weiner@uiowa.edu
Sonya J Koo
Stéphane Nicolas
Sandrine Fraboulet
Samuel L Pfaff
Olivier Pourquié
Joshua R Sanes
Resource Type: Journal article
Publication Details: Molecular and cellular neurosciences, Vol.27(1), pp.59-69
DOI: 10.1016/j.mcn.2004.06.005
PMID: 15345243
NLM abbreviation: Mol Cell Neurosci
ISSN: 1044-7431
eISSN: 1095-9327
Publisher: United States
Language: English
Date published: 09/2004
Academic Unit: Liberal Arts and Science Admin; Psychiatry; Iowa Neuroscience Institute; Biology
Record Identifier: 9983997987602771

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