RRC ID 57089
Author Wolfstetter G, Dahlitz I, Pfeifer K, Töpfer U, Alt JA, Pfeifer DC, Lakes-Harlan R, Baumgartner S, Palmer RH, Holz A.
Title Characterization of Drosophila Nidogen/entactin reveals roles in basement membrane stability, barrier function and nervous system patterning.
Journal Development
Abstract Basement membranes (BMs) are specialized layers of extracellular matrix (ECM) mainly composed of Laminin, type IV Collagen, Perlecan and Nidogen/entactin (NDG). Recent in vivo studies challenged the initially proposed role of NDG as a major ECM linker molecule by revealing dispensability for viability and BM formation. Here, we report the characterization of the single Ndg gene in Drosophila. Embryonic Ndg expression was primarily observed in mesodermal tissues and the chordotonal organs, whereas NDG protein localized to all BMs. Although loss of Laminin strongly affected BM localization of NDG, Ndg-null mutants exhibited no overt changes in the distribution of BM components. Although Drosophila Ndg mutants were viable, loss of NDG led to ultrastructural BM defects that compromised barrier function and stability in vivo Moreover, loss of NDG impaired larval crawling behavior and reduced responses to vibrational stimuli. Further morphological analysis revealed accompanying defects in the larval peripheral nervous system, especially in the chordotonal organs and the neuromuscular junction (NMJ). Taken together, our analysis suggests that NDG is not essential for BM assembly but mediates BM stability and ECM-dependent neural plasticity during Drosophila development.
Volume 146(2)
Published 2019-1-16
DOI 10.1242/dev.168948
PII dev.168948
PMID 30567930
MeSH Animals Basement Membrane / metabolism* Basement Membrane / ultrastructure Behavior, Animal Biomechanical Phenomena Body Patterning* Calcium-Binding Proteins / genetics Calcium-Binding Proteins / metabolism* Drosophila Proteins / genetics Drosophila Proteins / metabolism* Drosophila melanogaster / embryology* Drosophila melanogaster / genetics Drosophila melanogaster / metabolism* Embryo, Nonmammalian / metabolism Embryonic Development / genetics Extracellular Matrix Proteins / genetics Extracellular Matrix Proteins / metabolism* Gene Deletion Gene Expression Regulation, Developmental Laminin / metabolism Larva / genetics Nervous System / embryology* Nervous System / metabolism* Neuromuscular Junction / pathology Peripheral Nervous System / embryology Peripheral Nervous System / pathology Permeability Phenotype RNA, Messenger / genetics RNA, Messenger / metabolism Vibration
IF 5.763
Times Cited 3