Abstract
Heparan sulfate (HS) proteoglycans modulate the biological activity of a number of growth factors in development, homeostasis, and cancer. Specific modifications of HS chains by HS biosynthetic enzymes have been implicated in growth factor signaling in multiple aspects of organogenesis. Although the role of HS 6-O-sulfotransferases has been described in processes such as trachea formation in Drosophila and vasculogenesis in zebrafish, little is known about how HS 6-O-sulfotransferases (Hs6st1-3 in mice) influence mouse development. To address this issue, we generated a conditionally mutant Hs6st1 mouse line and then generated mice with systemic inactivation of Hs6st1. Hs6st1-null pups were viable and grossly normal at birth. The lack of obvious abnormalities in lung, liver, and kidney, which express high levels of Hs6st1 during development, suggests that at least during embryonic life, the loss of Hs6st1 function may be compensated for by mechanisms involving other HS modifying enzymes. During early adulthood, however, Hs6st1-null mice failed to thrive and exhibited growth retardation, body weight loss, enlargement of airspaces in the lung and, in some cases, lethality. Our results suggest a potentially critical role for HS 6-O...Continue Reading
References
Jul 18, 1997·The Journal of Biological Chemistry·R P MechamR M Senior
Sep 26, 1997·Science·R D HautamakiS D Shapiro
Feb 7, 1998·Proceedings of the National Academy of Sciences of the United States of America·S O'GormanY Marchuk
Jun 24, 1998·Genes & Development·S L BullockV A Wilson
Jan 25, 2000·The Journal of Biological Chemistry·H HabuchiK Kimata
Feb 2, 2000·BioEssays : News and Reviews in Molecular, Cellular and Developmental Biology·D M Ornitz
Feb 9, 2000·FEBS Letters·G FanG Hu
Mar 21, 2000·Genesis : the Journal of Genetics and Development·W N de VriesB B Knowles
Jun 15, 2000·The Journal of Biological Chemistry·M RingvallE Forsberg
Jun 22, 2000·The Journal of Biological Chemistry·V NurcombeH Hondermarck
Aug 30, 2000·Matrix Biology : Journal of the International Society for Matrix Biology·X Lin, N Perrimon
Mar 30, 2001·The Journal of Biological Chemistry·K KamimuraH Nakato
Jun 5, 2002·Annual Review of Biochemistry·Jeffrey D Esko, Scott B Selleck
Mar 6, 2003·Genome Research·Pentao LiuNeal G Copeland
Jun 5, 2003·Developmental Biology·Konstantin I IzvolskyWellington V Cardoso
Jun 6, 2003·The Journal of Biological Chemistry·Jin-Ping LiUlf Lindahl
Jun 24, 2003·American Journal of Physiology. Lung Cellular and Molecular Physiology·Konstantin I IzvolskyWellington V Cardoso
Jul 16, 2003·The Journal of Cell Biology·Xingbin AiCharles P Emerson
Dec 9, 2003·The Journal of Biological Chemistry·Ken NogamiKoji Kimata
Feb 11, 2004·FASEB Journal : Official Publication of the Federation of American Societies for Experimental Biology·Eyal ZchariaIsrael Vlodavsky
Mar 31, 2004·Proceedings of the National Academy of Sciences of the United States of America·Shouwen WangCharles P Emerson
Oct 23, 2004·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Jeff SeditaWellington V Cardoso
Nov 3, 2004·The Journal of Clinical Investigation·April D StraderRandy J Seeley
Jul 13, 2005·Developmental Biology·Eleanor ChenStephen C Ekker
Jul 14, 2005·Chemical Reviews·John M Whitelock, Renato V Iozzo
Jul 16, 2005·Development·Kay GrobeJeffrey D Esko
Feb 14, 2006·The Journal of Clinical Investigation·A McGarry HoughtonSteven D Shapiro
Jun 30, 2006·The Journal of Neuroscience : the Official Journal of the Society for Neuroscience·Thomas PrattJohn O Mason
Jul 27, 2006·Biochemistry·Jean L SpencerMatthew A Nugent
Aug 12, 2006·The Biochemical Journal·William C LamannaCatherine L R Merry
Sep 13, 2006·The Journal of Cell Biology·Keisuke KamimuraHiroshi Nakato
Nov 23, 2006·Molecular and Cellular Biology·David H LumZena Werb
Jan 4, 2007·The Journal of Clinical Investigation·Jennifer M MacArthurJeffrey D Esko
Apr 5, 2007·The Journal of Biological Chemistry·Hiroko HabuchiKoji Kimata
Citations
Oct 27, 2010·The Journal of Biological Chemistry·Ding XuJeffrey D Esko
Mar 2, 2011·The Journal of Biological Chemistry·Xiuxia QuXin Zhang
Dec 12, 2012·The Journal of Biological Chemistry·Md Ferdous Anower-E-KhudaKoji Kimata
Oct 13, 2012·The Journal of Biological Chemistry·Kevin T BushSanjay K Nigam
Jun 22, 2011·Cold Spring Harbor Perspectives in Biology·Stephane SarrazinJeffrey D Esko
Jun 30, 2012·Development·Xiuxia QuXin Zhang
Apr 23, 2014·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Zhigang CaiXin Zhang
Nov 1, 2018·Nature Methods·Hong QiuLianchun Wang
Mar 20, 2010·Birth Defects Research. Part C, Embryo Today : Reviews·Sophie M ThompsonDavid G Fernig
May 27, 2010·The Anatomical Record : Advances in Integrative Anatomy and Evolutionary Biology·Nicole C SmitsToin H van Kuppevelt
Sep 1, 2017·PLoS Genetics·Hua HeXinhua Lin
Nov 6, 2009·The Journal of Biological Chemistry·Kristin I StanfordJeffrey D Esko
Jul 30, 2019·Frontiers in Cell and Developmental Biology·Hyun-Ju ChoSusan Wray
Oct 18, 2012·Developmental Dynamics : an Official Publication of the American Association of Anatomists·Rita A Buresh-StiemkePaul C Marker
Jul 8, 2016·International Journal of Experimental Pathology·Vivien Jane Coulson-Thomas
Jan 30, 2021·Frontiers in Cell and Developmental Biology·Matthew R JonesSaverio Bellusci
Oct 22, 2015·Development·Fabienne E Poulain, H Joseph Yost
May 19, 2011·Developmental Biology·Omai B GarnerSanjay K Nigam