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human ENS stem cells

2.1     Introduction

 

The   isolation   and   culture   of   ENS   stem   cells   from   embryonic   mice   is   well   established   and   there   are   several   different   techniques   available   to   do   this,   although  all  of  them  take  the  dissection  and  dissociation  of  the  embryonic  bowel   into  a  single  cell  suspension  as  the  first  step.    ENSC  are  then  enriched  and  grown   in  culture  using  either  FACS  or  magnetic  beads  to  select  for  the  stem  cell  markers   p75(Pomeranz   et   al.,   1993),   p75   and   α4   integrin(Bixby   et   al.,   2002)   and   RET(Lo   and   Anderson,   1995),   or   by   culturing   in   the   form   of   neurospheres   with   no   immunoselection(Almond   et   al.,   2003,   Bondurand   et   al.,   2003,   Schafer   et   al.,   2003).  

 

These   techniques   have   been   modified   to   allow   for   the   isolation   and   culture   of   postnatal   rodent   ENSC(Kruger   et   al.,   2002,   Bondurand   et   al.,   2003,   Suarez-­‐ Rodriguez   and   Belkind-­‐Gerson,   2004).     Although   there   are   obvious   differences   between  mouse  and  human  colon,  most  obviously  in  the  size  of  the  tissue  and  the   relative   maturity   of   human   colon   in   respect   to   rat   and   mouse   colon(Karen   Walthall,   2005),   it   seems   logical   to   attempt   to   adapt   these   techniques   to   the   isolation  and  culture  of  human  postnatal  ENSC.  

 

2.1.1    Identification  of  stem  cells  

 

As   defined   in   Chapter   1,   a   stem   cell   has   the   capacity   to   self-­‐renew   (make   more   stem  cells  by  cell  division)  as  well  as  to  differentiate  into  mature,  specialized  cells.     The  identification  of  ENSC  as  genuine  stem  cells  is  problematic.      

There  are  various  cellular  markers  that  have  been  used  to  identify  ENSC,  including   Sox10,  RET,  p75  and  nestin  (see(Young  and  Newgreen,  2001)  for  a  review).    RET,   all  undifferentiated  neural  crest  cells  within  the  gut  wall  express  p75  and  Phox2b   as  they  begin  to  colonise  the  hindgut(Young  and  Newgreen,  2001).    RET,  p75  and   nestin   have   all   been   used   in   previous   studies   to   identify   ENSC   (Natarajan   et   al.,   1999,  Sidebotham  et  al.,  2001,  Schafer  et  al.,  2003,  Belkind-­‐Gerson  et  al.,  2006).     The  problem  with  such  markers  is  that  they  are  not  necessarily  exclusive  to  ENSC.     RET   and   Phox2b   expression   is   maintained   in   cells   differentiating   into   a   neural   lineage  and  p75  expression  is  maintained  in  cells  differentiating  into  a  glial  lineage   (Young  et  al.,  1999).    Nestin  has  been  advocated  a  good  marker  for  ENSC(Belkind-­‐ Gerson  et  al.,  2006,  Rauch  et  al.,  2006)  but  although  its  expression  in  glial  cells  in   the  CNS  is  very  low,  it  is  expressed  postnatally  in  ENS  glia  and  ICC(Vanderwinden   et  al.,  2002).    It  can  therefore  be  concluded  that  there  is,  at  the  present  time,  no   specific  marker  for  ENSC.  

 

Another  method  of  identifying  neural  stem  cells  is  utilising  their  capacity  to  form   neurospheres(Reynolds  and  Weiss,  1996).    This  capacity  has  led  to  the  concept  of   the  neurosphere  assay  –  the  assumption  that  under  specific  conditions,  only  such   stem  cells  will  form  free  floating  spherical  cellular  aggregates(Pacey  et  al.,  2006).     However,   other   cell   types   will   also   form   spherical   aggregates   in   culture   and   therefore  this  assay  is  not  specific  enough  to  positively  identify  ENSC.  

 

A  further  method  of  identifying  ENSC  is  by  their  behaviour:    a  true  stem  cell  is  able   to  undergo  asymmetric  division  to  produce  an  identical  daughter  cell  and  a  more   differentiated  precursor  cell  which  undergoes  subsequent  differentiation,  in  this   case   to   either   neurons   or   glia.     ENSPC   can   therefore   be   demonstrated   by   identifying  a  single  cell  that  goes  on  to  produce  both  neurons  glia  within  the  same   colony   –   a   demonstration   of   clonality(Bondurand   et   al.,   2003).     Alternatively,   it   could  also  be  demonstrated  that  a  neurosphere  is  capable  of  generating  multiple   daughter  cells  when  disaggregated.      If  these  daughter  cells  have  characteristics   identical  to  those  of  the  original  neurosphere,  and  can  form  further  neurospheres  

Combinations   of   the   above   techniques   are   therefore   employed   in   the   identification  ENSC  during  the  course  of  this  thesis.  

   

2.1.2  Summary  of  work  done  prior  to  the  commencement  of  

this  thesis  

 

Work  done  by  Almond  on  the  isolation  and  characterisation  of  embryonic  mouse   ENSC  has  been  previously  described(Almond  et  al.,  2003)  and  the  techniques  used   in   this   thesis   build   on   this   work.     In   summary   Almond   developed   a   method   of   producing  ENSC  from  embryonic  mouse  caeca  at  harvested  at  E11.5  and  culturing   them  in  the  form  of  neurospheres.    These  neurospheres  were  shown  to  contain   differentiated   neurons   and   glia   and   contained   single   cells   with   the   capacity   to   form  both  neurons  and  glia.    Furthermore  it  was  possible  to  clonally  expand  the   neurospheres   from   a   single   cell,   proving   the   existence   of   true   ENSC   within   the   neurospheres.  

2.2    Aims  

 

The   aim   of   this   study   was   to   develop   a   method   of   isolating   postnatal   human   ENSPC.     Initially,   competency   in   growing   embryonic   mouse   neurospheres   previously   shown   to   contain   ENSPC   was   achieved.     The   method   for   growing   mouse   ENSC   was   then   adapted   to   produce   human   ENSC   in   the   form   of   neurospheres.    ENSC  were  identified  by  immunofluorescence  for  the  markers  p75   and   nestin,   the   ability   to   produce   both   neurons   and   glia   and   the   ability   to   self-­‐ renew  by  the  generation  of  secondary  and  tertiary  neurospheres.    ENSC  were  also   identified   by   their   ability   to   produce   neurons   and   glia   once   transplanted   into   aganglionic,  embryonic  mouse  hindgut.  

 

The   following   sections   describe   groups   of   experiments;   for   each   group   the   methods   and   results   will   be   described,   followed   by   a   discussion   of   significant   results   and   issues   related   to   the   experimental   design   before   moving   on   to   the   next  section.  

2.3     Generation   of   embryonic   mouse   neurospheres  

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