PMID: 8962054Dec 10, 1996Paper

A fusion pore phenotype in mast cells of the ruby-eye mouse

Proceedings of the National Academy of Sciences of the United States of America
A F Oberhauser, J M Fernandez

Abstract

Using patch-clamp capacitance and amperometric techniques, we have identified an exocytotic phenotype that affects the function of the fusion pore, the molecular structure that connects the lumen of a secretory vesicle with the extracellular environment during exocytosis. Direct observation of individual exocytotic events in mast cells from the ruby-eye mouse (ru/ru) showed a 3-fold increase in the fraction and duration of transient fusion events with respect to wild-type mice. The fraction of the total fusion events that were transient increased from 0.22 +/- 0.02 (wild type) to 0.65 +/- 0.02 (ru/ru), and the average duration of these events increased from 418 +/- 32 ms (wild type) to 1207 +/- 89 ms (ru/ru). We also show that this phenotype can reduce and delay an evoked secretory response by causing the fusion of vesicles that have been previously emptied by repeated cycles of transient fusion. The exocytotic phenotype that we describe here may be a cause of diseases like platelet storage pool deficiency and prolonged bleeding times for which the ruby-eye mouse serves as an animal model. Furthermore, the identification of the gene causing the fusion pore phenotype reported here will illuminate the molecular mechanisms regulat...Continue Reading

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Citations

Aug 23, 2005·Brain Research. Brain Research Reviews·Carlos Fernández-PeruchenaGuillermo Alvarez de Toledo
Feb 13, 2001·Journal of Neuroscience Methods·T L ColliverA G Ewing
Oct 20, 2001·Trends in Cell Biology·R D BurgoyneM E Graham
May 19, 1998·Pigment Cell Research·R T SwankL Feng
Jun 12, 2008·Annual Review of Genomics and Human Genetics·Marjan HuizingWilliam A Gahl
Apr 29, 1998·Annual Review of Physiology·W J Betz, J K Angleson
Nov 28, 2002·Pigment Cell Research·Marjan HuizingWilliam A Gahl
Oct 9, 2014·Frontiers in Immunology·Ulrich BlankClaudia González-Espinosa
Mar 21, 1998·Neuroscience·H C Palfrey, C R Artalejo
Jun 2, 2004·BioEssays : News and Reviews in Molecular, Cellular and Developmental Biology·Wei LiRichard T Swank
Jun 10, 2018·The Journal of Immunology : Official Journal of the American Association of Immunologists·Na-Ryum BinShuzo Sugita
Mar 31, 2018·The Journal of Biological Chemistry·Berenice A GutierrezRoberto Adachi
Dec 12, 2001·Endocrine Reviews·N Ben-Jonathan, R Hnasko
Mar 16, 2000·Reviews in the Neurosciences·D Sulzer, E N Pothos
Nov 17, 2017·The Journal of Biological Chemistry·Elsa M RodarteRoberto Adachi
Nov 7, 1999·The Journal of Biological Chemistry·R Fernández-ChacónT C Südhof
Mar 29, 2003·Physiological Reviews·Robert D Burgoyne, Alan Morgan
Nov 29, 2001·British Journal of Pharmacology·F ToreP Aubineau
May 28, 2013·The Journal of Biological Chemistry·Jose M CabezaEva Alés
Aug 29, 2001·The Journal of Biological Chemistry·L TabaresG Alvarez de Toledo

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