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The influence of a pediatric palliative care program on health care utilization and costs Conte, Tania 2014

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THE	  INFLUENCE	  OF	  A	  PEDIATRIC	  PALLIATIVE	  CARE	  PROGRAM	  ON	  HEALTH	  CARE	  UTILIZATION	  AND	  COSTS	  by	  Tania	  Conte	  	  R.N.,	  Universidade	  Federal	  do	  Paraná	  (UFPR),	  2003	  	  A	  THESIS	  SUBMITTED	  IN	  PARTIAL	  FULFILLMENT	  OF	  THE	  REQUIREMENTS	  FOR	  THE	  DEGREE	  OF	  	  MASTER	  OF	  SCIENCE	  in	  THE	  FACULTY	  OF	  GRADUATE	  AND	  POSTDOCTORAL	  STUDIES	  	  (Population	  and	  Public	  Health)	  	  THE	  UNIVERSITY	  OF	  BRITISH	  COLUMBIA	  (Vancouver)	  	  	  November	  2014	  	  ©	  Tania	  Conte,	  2014	  	   	   ii Abstract	  	  Pediatric	  palliative	  care	  program	  (PPCP)	  is	  believed	  to	  increase	  system	  efficiency.	  British	  Columbia	  (BC)	  relies	  on	  a	  free-­‐standing	  hospice-­‐based	  PPCP,	  and	  its	  effects	  on	  health	  care	  utilization	  and	  costs	  remain	  an	  outstanding	  question.	  This	  study	  aimed	  to	  gather	  evidence	  in	  a	  combined	  analysis	  of	  data	  from	  literature	  and	  BC	  PPCP.	  	  A	  systematic	  review	  was	  conducted	  through	  an	  electronic	  search	  of	  Medline,	  Embase,	  CINAHL,	  LILACS,	  and	  grey	  literature.	  Comparative	  studies	  reporting	  admissions,	  length	  of	  stay,	  and	  health	  care	  costs	  between	  PPCP	  users	  and	  usual	  care	  were	  included.	  Additionally,	  a	  similar	  comparison	  was	  applied	  to	  the	  data	  from	  BC	  PPCP	  using	  a	  retrospective	  matched-­‐pairs	  cohort	  design	  (matched	  by	  ICD	  code	  and	  age	  at	  death)	  with	  a	  3-­‐year	  observational	  period	  prior	  to	  death.	  Data	  were	  obtained	  from	  Canuck	  Place	  Children’s	  Hospice	  and	  BC	  Children’s	  Hospital	  databases,	  and	  complemented	  by	  estimates	  from	  Canadian	  Institute	  for	  Health	  Information.	  A	  cost	  impact	  of	  the	  overall	  inpatient	  care	  provided	  by	  the	  hospice	  was	  presented.	  	  The	  review	  did	  not	  demonstrate	  a	  decrease	  in	  utilization	  by	  PPCP	  users	  yet	  suggested	  a	  shift	  to	  other	  health	  care	  settings,	  and	  potential	  cost	  saving	  in	  the	  Canadian	  context	  (1	  article).	  The	  cohort	  study	  (n=11	  pairs),	  suggests	  that	  children	  in	  both	  groups	  had	  similar	  upward	  trends	  in	  inpatient	  utilization	  and	  cost.	  However,	  PPCP	  users	  showed	  more	  inpatient	  care	  in	  the	  last	  year	  of	  life	  (especially	  critical	  care	  in	  the	  last	  2	  months),	  compared	  to	  their	  controls	  and	  to	  the	  period	  prior	  to	  referral.	  Post-­‐referral,	  a	  shift	  in	  health	  care	  setting	  utilization	  from	  hospital	  to	  hospice	  was	  observed,	  representing	  approximately	  50%	  of	  the	  costs.	  Without	  this	  shift	  PPCP	  users	  would	  have	  cost	  32%	  more	  with	  a	  median	  monthly	  increment	  of	  $7,163	  per	  child.	  All	  inpatient	  care	  provided	  by	  the	  hospice	  in	  the	  fiscal	  year	  2011-­‐2012	  represented	  a	  potential	  cost	  saving	  ranging	  from	  approximately	  $1.1M	  to	  $4.3M.	  The	  findings	  of	  this	  study	  suggest	  that	  PPCP	  users	  may	  present	  higher	  health	  care	  needs,	  and	  that	  the	  shift	  of	  inpatient	  care	  to	  the	  hospice	  optimized	  resource	  use,	  offering	  a	  more	  holistic	  approach	  to	  EOL	  care,	  relieving	  hospital	  resources	  to	  meet	  other	  demands.	  	  	  	   	   iii Preface	  	   I	  was	  involved	  in	  the	  conceptualization,	  study	  design,	  data	  collection,	  analysis,	  and	  interpretation	  of	  all	  three	  studies	  in	  this	  research.	  	  A	  version	  of	  Chapter	  2	  has	  been	  accepted	  for	  publication	  ["Effects	  of	  Pediatric	  Palliative	  Care	  Programs	  for	  Children	  with	  Life-­‐Threatening	  Conditions	  in	  Health	  care	  Resources	  Utilization	  and	  Costs:	  a	  Systematic	  Review	  of	  Comparative	  Studies",	  CMAJ	  Open	  2014,	  to	  be	  released]	  for	  which	  I	  was	  the	  corresponding	  author,	  responsible	  for	  all	  the	  major	  areas	  such	  as	  the	  systematic	  search	  and	  review,	  data	  synthesis	  and	  manuscript	  composition.	  Logan	  Trenaman	  and	  Dr.	  Harold	  Siden	  contributed	  as	  additional	  reviewers	  of	  the	  systematic	  review,	  and	  along	  with	  Negar	  Chavoshi	  and	  Dr.	  Craig	  Mitton	  contributed	  significantly	  to	  the	  analysis	  and	  interpretation	  of	  the	  results,	  and	  multiple	  editing	  to	  the	  manuscript.	  Abstracts	  of	  this	  systematic	  review	  were	  presented	  at	  Health	  Technology	  Assessment	  International	  Meeting	  (HTAi)	  2014	  and	  20th	  International	  Conference	  in	  Palliative	  Care.	  	  In	  Chapters	  3	  and	  4,	  I	  was	  involved	  in	  the	  design,	  data	  collection,	  analysis	  and	  interpretation	  of	  the	  cohort	  study	  results	  with	  support	  a	  multidisciplinary	  team.	  Dr.	  Craig	  Mitton	  was	  my	  supervisor	  involved	  from	  the	  early	  stages	  of	  conceptualization,	  interpretation	  of	  the	  results,	  and	  made	  substantial	  contributions	  to	  the	  structure	  of	  the	  research.	  Dr.	  Harold	  Siden	  and	  Negar	  Chavoshi	  participated	  significantly	  in	  the	  matching	  process,	  interpretation	  and	  editing	  of	  the	  findings,	  and	  Shanon	  Erderlyi	  contributed	  with	  the	  coding	  for	  the	  analysis	  and	  graphs.	  Cindy	  Stutzer	  and	  Rod	  Rassekh	  consulted	  with	  the	  cancer	  matching	  process.	  Tanice	  Miller	  and	  Joan	  Hill	  contributed	  with	  operating	  cost	  data	  from	  the	  hospice.	  Multiple	  members	  of	  PHSA	  Decision	  Support	  team	  participated	  retrieving	  and	  processing	  confidential	  data	  on	  utilization	  and	  cost	  for	  the	  hospital	  facility.	  This	  research	  was	  granted	  approval	  from	  the	  Research	  Ethics	  Board	  of	  Children’s	  and	  Women’s	  Health	  Centre	  of	  British	  Columbia	  (certificate	  numbers	  CW13-­‐0210	  /	  H13-­‐01162)	  and	  Canuck	  Place	  Research	  Review	  Committee.	  	   	   iv Table	  of	  Contents	  Abstract	  ........................................................................................................................................	  ii	  Preface	  ........................................................................................................................................	  iii	  Table	  of	  Contents	  ........................................................................................................................	  iv	  List	  of	  Tables	  ................................................................................................................................	  x	  List	  of	  Figures	  ............................................................................................................................	  xiii	  List	  of	  Abbreviations	  ..................................................................................................................	  xvi	  Acknowledgements	  ..................................................................................................................	  xviii	  Dedication	  ..................................................................................................................................	  xix	  Chapter	  1:	  Introduction	  ................................................................................................................	  1	  1.1	   Background	  ........................................................................................................................	  2	  1.1.1	   Conceptual	  Framework	  ............................................................................................	  2	  1.1.2	   Life-­‐threatening	  Conditions	  -­‐	  Definition	  ...................................................................	  3	  1.1.2.1	   Categorization	  of	  Life-­‐threatening	  Conditions	  .....................................................	  3	  1.1.3	   Pediatric	  Palliative	  Care	  Definition	  and	  Criteria	  .......................................................	  5	  1.1.4	   Estimates	  on	  Demand	  and	  Enrollment	  in	  Pediatric	  Palliative	  Care	  Programs	  ..........	  7	  1.1.5	   The	  Pediatric	  Palliative	  Care	  Program	  in	  British	  Columbia	  and	  Rationale	  for	  this	  Study	  ......................................................................................................................	  10	  1.2	   Objectives	  ........................................................................................................................	  13	  1.2.1	   Research	  Questions	  ................................................................................................	  13	  1.2.2	   Hypotheses	  .............................................................................................................	  14	  Chapter	  2:	  Effects	  of	  PPCP	  for	  Children	  with	  LTC’s	  in	  Health	  Care	  Resources	  Utilization	  and	  Costs:	  a	  Systematic	  Review	  of	  Comparative	  Studies	  ..............................................................................	  16	  	   	   v 2.1	   Objective	  .........................................................................................................................	  16	  2.2	   Methods	  ..........................................................................................................................	  16	  2.2.1	   Inclusion	  Criteria	  ....................................................................................................	  16	  2.2.2	   Search	  Methods	  for	  Identification	  of	  Studies	  .........................................................	  18	  2.2.3	   Data	  Collection,	  Analysis	  and	  Quality	  Assessment	  .................................................	  18	  2.3	   Results	  .............................................................................................................................	  19	  2.3.1	   Study	  Selection	  .......................................................................................................	  19	  2.3.2	   Study	  Characteristics	  ..............................................................................................	  21	  2.3.2.1	   Description	  of	  Studies	  .........................................................................................	  21	  2.3.2.2	   Risk	  of	  Bias	  and	  Quality	  Assessment	  ...................................................................	  22	  2.3.3	   Effects	  of	  Pediatric	  Palliative	  Care	  Program	  on	  Outcomes	  of	  Interest	  ...................	  25	  2.3.3.1	   Hospital	  Admissions	  ............................................................................................	  28	  2.3.3.2	   Length	  of	  Stay	  (LOS)	  ............................................................................................	  31	  2.3.3.3	   Health	  Care	  Costs	  ................................................................................................	  34	  2.3.3.4	   LOS	  in	  the	  Last	  Admission	  Before	  Death	  .............................................................	  38	  2.3.3.5	   Invasive	  Procedures	  to	  Prolong	  Life	  in	  the	  Last	  Admission	  .................................	  38	  2.3.3.6	   Resuscitation	  Attempts	  .......................................................................................	  38	  2.4	   Discussion	  ........................................................................................................................	  39	  2.4.1	   Summary	  of	  Main	  Results	  and	  Limitations	  .............................................................	  39	  2.4.2	   Overall	  Completeness	  and	  Applicability	  of	  Evidence	  .............................................	  42	  2.4.3	   Quality	  of	  the	  Evidence	  ..........................................................................................	  42	  2.4.4	   Potential	  Biases	  in	  the	  Review	  Process	  ..................................................................	  43	  2.4.5	   Agreements	  and	  Disagreements	  with	  Other	  Studies	  or	  Reviews	  ...........................	  43	  	   	   vi 2.5	   Conclusion	  .......................................................................................................................	  43	  2.5.1	   Implications	  for	  Practice	  .........................................................................................	  43	  2.5.2	   Implications	  for	  Research	  .......................................................................................	  44	  Chapter	  3:	  Health	  Services	  Utilization:	  a	  Matched-­‐Cohort	  Comparison	  .......................................	  45	  3.1	   Objective	  .........................................................................................................................	  45	  3.2	   Methods	  ..........................................................................................................................	  46	  3.2.1	   Study	  Design	  ...........................................................................................................	  46	  3.2.2	   Definitions	  ..............................................................................................................	  46	  3.2.3	   Assumptions	  ...........................................................................................................	  47	  3.2.4	   Exclusion	  Criteria	  ....................................................................................................	  48	  3.2.5	   Data	  Source	  ............................................................................................................	  48	  3.2.6	   Ethics	  Approval	  .......................................................................................................	  49	  3.2.7	   Sampling	  Procedures	  and	  Matching	  Process	  .........................................................	  49	  3.2.8	   Deviations	  from	  Original	  Matching	  Strategy	  ..........................................................	  50	  3.2.9	   Outcome	  Measures	  ................................................................................................	  51	  3.2.10	   Hypothesis	  ..............................................................................................................	  51	  3.2.11	   Sample	  Size	  ............................................................................................................	  52	  3.2.12	   Observational	  Period	  ..............................................................................................	  52	  3.2.13	   Statistical	  Analyses	  .................................................................................................	  53	  3.2.13.1	   Exploratory	  Analysis	  ...........................................................................................	  55	  3.3	   Results	  .............................................................................................................................	  56	  3.3.1	   Demographics	  ........................................................................................................	  56	  3.3.2	   Number	  of	  Admissions	  ...........................................................................................	  57	  	   	   vii 3.3.3	   LOS	  .........................................................................................................................	  64	  3.3.4	   Last	  Admission	  Before	  Death	  (EOL	  admission)	  .......................................................	  72	  3.3.4.1	   LOS	  ......................................................................................................................	  72	  3.3.4.2	   Invasive	  Procedures	  ............................................................................................	  72	  3.3.5	   Sample	  Size	  Calculation	  for	  Future	  Research	  .........................................................	  73	  3.4	   Discussion	  ........................................................................................................................	  75	  3.4.1	   Summary	  of	  Results	  ................................................................................................	  75	  3.4.2	   Limitations	  of	  this	  Study	  .........................................................................................	  78	  3.5	   Conclusions	  ......................................................................................................................	  82	  Chapter	  4:	  Cost	  Evaluation:	  a	  Matched-­‐Cohort	  Analysis	  and	  Budget	  Exercise	  ..............................	  85	  4.1	   Objectives	  ........................................................................................................................	  85	  4.2	   Methods	  ..........................................................................................................................	  86	  4.2.1	   Definitions	  ..............................................................................................................	  86	  4.2.2	   Study	  Design	  ...........................................................................................................	  88	  4.2.3	   Final	  Units	  of	  Costs	  .................................................................................................	  88	  4.2.4	   Characteristics	  of	  the	  Data,	  Source,	  Cost	  Components	  ..........................................	  89	  4.2.5	   Hypothesis	  ..............................................................................................................	  94	  4.2.6	   Sample	  Size	  ............................................................................................................	  94	  4.2.7	   Statistical	  Analyses	  –	  ‘Combined	  System’	  ..............................................................	  94	  4.2.8	   Simulation	  Exercise	  –	  ‘Single	  System’	  ....................................................................	  95	  4.3	   Results	  .............................................................................................................................	  96	  4.3.1	   Final	  Units	  of	  Cost	  ..................................................................................................	  96	  4.3.1.1	   Outpatient	  and	  Emergency	  Visits	  .......................................................................	  96	  	   	   viii 4.3.1.2	   Inpatient	  Admissions	  ..........................................................................................	  97	  4.3.2	   Cost	  Comparison	  –	  ‘Combined	  System’	  ...............................................................	  100	  4.3.3	   Sample	  Size	  Calculation	  for	  Future	  Research	  .......................................................	  109	  4.3.4	   Single	  System	  Simulation	  	  -­‐	  What	  if	  CPCH	  had	  not	  Provided	  Inpatient	  Care?	  ......	  112	  4.3.4.1	   Within	  the	  Matched-­‐Pairs	  Cohort	  Study	  ...........................................................	  112	  4.3.4.2	   Within	  the	  Entire	  Hospice	  Population	  ..............................................................	  117	  4.4	   Discussion	  ......................................................................................................................	  121	  4.4.1	   Summary	  of	  Results	  ..............................................................................................	  121	  4.4.2	   Limitations	  of	  this	  Study	  .......................................................................................	  126	  4.5	   Conclusions	  ....................................................................................................................	  127	  Chapter	  5:	  Final	  Discussion	  .......................................................................................................	  129	  5.1	   Summary	  of	  Results	  .......................................................................................................	  129	  5.2	   Contributions	  to	  Research	  .............................................................................................	  131	  5.3	   Limitations	  of	  this	  Study	  ................................................................................................	  133	  5.4	   Knowledge	  Translation	  ..................................................................................................	  136	  5.5	   Policy	  Implications	  .........................................................................................................	  137	  5.6	   Future	  Research	  ............................................................................................................	  137	  Bibliography	  .............................................................................................................................	  140	  Appendices	  ...............................................................................................................................	  144	  Appendix	  A	  .	  Search	  Strategy	  ....................................................................................................	  144	  A.1	   Medline	  Search	  Strategy	  ...............................................................................................	  144	  A.2	   EMBASE	  Search	  Strategy	  ...............................................................................................	  150	  A.3	   CINAHL	  Search	  Strategy	  .................................................................................................	  156	  	   	   ix A.4	   LILACS	  Search	  Strategy	  .....................................................................................................	  157	  A.5	   Grey	  Literature	  Search	  Strategy	  .......................................................................................	  157	  Appendix	  B	  .	  Study	  Characteristics	  –	  Full	  Details	  .......................................................................	  158	  Appendix	  C	  .	  Study	  Design	  Features	  for	  Non-­‐Observational	  Studies	  ..........................................	  162	  Appendix	  D	  .	  Newcastle-­‐Ottawa	  Scale	  ......................................................................................	  173	  Appendix	  E	  .	  Pairwise	  Comparison	  of	  Admissions	  and	  LOS	  ........................................................	  175	  Appendix	  F	  .	  CIHI	  Methodology	  for	  Calculation	  of	  Inpatient	  Ward	  and	  ICU	  Hospital	  Per	  Diem	  Rates	  Using	  the	  2011-­‐2012	  Canadian	  MIS	  Database59	  ...............................................................	  180	  Appendix	  G	  .	  Pairwise	  Comparison	  of	  Costs	  ..............................................................................	  208	  	  	  	   	   x List	  of	  Tables	  	  Table	  1:	  Categorization	  of	  LTCs	  ............................................................................................................	  4	  Table	  2.	  Characteristics	  of	  the	  Studies	  ...............................................................................................	  26	  Table	  3.	  Visual	  Summary	  of	  Results	  and	  NOS	  Quality	  Assessment	  ....................................................	  27	  Table	  4.	  Summary	  of	  Comparison	  of	  Hospital	  Admissions	  .................................................................	  30	  Table	  5.	  Summary	  of	  Comparison	  of	  LOS	  ...........................................................................................	  33	  Table	  6.	  Summary	  of	  Cost	  Comparison	  ..............................................................................................	  37	  Table	  7.	  Clinical	  Question	  and	  Outcomes	  ...........................................................................................	  45	  Table	  8.	  Demographics	  Table	  .............................................................................................................	  57	  Table	  9.	  Monthly	  Number	  of	  Admissions	  per	  Type,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Referral	  to	  PPCP	  Period§	  .......................................................................................................................................	  58	  Table	  10.	  Monthly	  Number	  of	  Admissions	  per	  Type,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Post-­‐Referral	  to	  PPCP	  Period	  ........................................................................................................................................	  59	  Table	  11.	  Changes	  in	  the	  Monthly	  Number	  of	  Admissions	  per	  Type,	  in	  the	  CPCH	  Arm	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ....................................................................................	  61	  Table	  12.	  Changes	  in	  the	  Monthly	  Number	  of	  Admissions	  per	  Type,	  in	  the	  BCCH	  Arm	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ....................................................................................	  62	  Table	  13.	  Monthly	  Number	  of	  Inpatient	  Admissions	  per	  Facility	  –	  Entire	  Study	  Period§	  ...................	  63	  Table	  14.	  Monthly	  LOS	  per	  Type,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Referral	  to	  PPCP	  Period§	  .......	  66	  Table	  15.	  Monthly	  LOS	  per	  Type,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Post-­‐Referral	  to	  PPCP	  Period	  ......	  68	  Table	  16.	  Changes	  in	  the	  Monthly	  LOS	  per	  Type,	  in	  the	  CPCH	  Arm	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................................................	  69	  	   	   xi Table	  17.	  Changes	  in	  the	  Monthly	  LOS	  per	  Type,	  in	  the	  BCCH	  Arm	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................................................	  70	  Table	  18.	  Monthly	  LOS	  per	  Facility	  –	  Entire	  Study	  Period§	  .................................................................	  71	  Table	  19.	  LOS	  in	  the	  Last	  Admission	  Before	  Death	  and	  Pairwise	  Test	  (Wilcoxon)	  .............................	  72	  Table	  20.	  Use	  of	  Invasive	  Procedures	  to	  Prolong	  Life	  in	  the	  EOL	  Admission	  and	  Pairwise	  Test	  (McNemar’s	  test)	  ................................................................................................................................	  72	  Table	  21.	  LOS	  for	  the	  EOL	  Admission	  by	  Pairs	  (in	  days)	  ......................................................................	  73	  Table	  22.	  Sample	  Mean	  LOS,	  Difference	  Between	  Groups	  and	  Variability	  ........................................	  74	  Table	  23.	  Parametric	  Test	  Sample	  Size	  Calculation	  ............................................................................	  74	  Table	  24.	  Term	  Definition	  for	  Cost	  Analysis	  .......................................................................................	  86	  Table	  25.	  Type	  of	  Admissions	  and	  Facility	  ..........................................................................................	  89	  Table	  26.	  Characteristics	  of	  the	  Cost	  Data	  per	  Type	  of	  Admission	  .....................................................	  90	  Table	  27.	  Cost	  Components	  Included	  in	  the	  Cost	  Allocation	  ..............................................................	  93	  Table	  28.	  Outpatient	  and	  Emergency	  Visits	  Final	  Units	  of	  Costs	  –	  Average	  Cost	  per	  Visit*	  ...............	  97	  Table	  29.	  Inpatient	  Admissions	  Final	  Units	  of	  Costs	  (Facility	  and	  Type	  of	  Admission)	  –	  Average	  Cost	  per	  Day	  per	  Bed*	  ................................................................................................................................	  98	  Table	  30.	  Canuck	  Place	  Cost	  Components	  and	  Cost	  Distribution	  –	  Direct	  Cost	  (assembling	  CIHI	  Methodology)	  .....................................................................................................................................	  99	  Table	  31.	  Monthly	  Cost	  per	  Type	  of	  Admission,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Referral	  ##	  .....	  103	  Table	  32.	  Monthly	  Cost	  per	  Type	  of	  Admission,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Post-­‐Referral.	  ......	  104	  Table	  33.	  BCCH	  Group:	  Changes	  in	  Monthly	  Cost	  per	  Type	  of	  Admission	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ..........................................................................................	  105	  	   	   xii Table	  34.	  CPCH	  Group:	  Changes	  in	  Monthly	  Cost	  per	  Type	  of	  Admission	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ..........................................................................................	  105	  Table	  35.	  Monthly	  Cost	  per	  Facility	  –	  Entire	  Study	  Period.	  §	  ............................................................	  106	  Table	  36.	  Difference	  Between	  Groups	  in	  Total	  Inpatient	  Cost	  Post-­‐Referral:	  Sample	  Mean	  and	  Variability.	  ........................................................................................................................................	  110	  Table	  37.	  Parametric	  Test’s	  Sample	  Size	  Calculation.	  .......................................................................	  110	  Table	  38.	  Monthly	  Cost	  per	  Type	  of	  Admission	  Simulating	  a	  Single	  System,	  and	  Pairwise	  Test	  (Wilcoxon)	  -­‐	  Post-­‐Referral	  Period.	  ....................................................................................................	  114	  Table	  39.	  CPCH	  Group,	  Changes	  in	  Monthly	  Cost	  per	  Type	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Simulating	  a	  Usual	  Care	  Scenario	  in	  the	  Post-­‐Referral	  Period	  ...........................................................................	  114	  Table	  40.	  CPCH	  Group,	  Absolute	  Number	  of	  Admissions	  to	  the	  Hospice	  and	  their	  Respective	  Costs	  of	  Similar	  Admissions	  at	  BCCH	  ..........................................................................................................	  115	  Table	  41.	  CPCH	  Group:	  Changes	  in	  Monthly	  Cost	  per	  Type	  of	  Admissions	  under	  a	  Single	  System	  Scenario,	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .............................................	  116	  Table	  42.	  CPCH	  Group,	  Changes	  in	  Monthly	  Cost	  per	  Type	  and	  Pairwise	  Test	  (Wilcoxon)	  –	  Simulating	  a	  Usual	  Care	  Scenario	  in	  the	  Entire	  Observational	  Period	  ...............................................................	  116	  Table	  43.	  Yearly	  Inpatient	  Care	  Provided	  at	  CPCH,	  Fiscal	  Year	  2011/2012	  (n=	  153	  children	  with	  LTC)	  ..........................................................................................................................................................	  119	  Table	  44.	  Sensitivity	  Analysis	  for	  the	  Uncertainty	  Around	  the	  Hospital	  Cost	  Units	  ..........................	  120	  	  	   	   xiii List	  of	  Figures	  	  Figure	  1.	  	  Macro	  Model	  of	  Health	  System	  ............................................................................................	  3	  Figure	  2.	  The	  Role	  of	  Pediatric	  Palliative	  Care	  ......................................................................................	  6	  Figure	  3.	  Flow	  Diagram	  of	  Study	  Selection	  .........................................................................................	  20	  Figure	  4.	  Monthly	  Number	  of	  Admissions	  per	  Type	  -­‐	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Referral	  to	  PPCP	  §	  ...............................................................................................................................	  58	  Figure	  5.	  Monthly	  Number	  of	  Admissions	  per	  Type	  -­‐	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Post-­‐Referral	  to	  PPCP	  .................................................................................................................................	  60	  Figure	  6.	  CPCH	  Arm	  -­‐	  Changes	  in	  the	  Monthly	  Number	  of	  Admissions	  per	  Type,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ...................................................................................	  61	  Figure	  7.	  BCCH	  Arm	  -­‐	  Changes	  in	  the	  Monthly	  Number	  of	  Admissions	  per	  Type,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  ...................................................................................	  63	  Figure	  8.	  LOS	  per	  Person-­‐Month	  at	  Risk	  Over	  Time,	  by	  Arm:	  Outpatient	  and	  Inpatient	  Admissions	  .	  65	  Figure	  9.	  LOS	  per	  Person-­‐Month	  at	  Risk	  Over	  Time	  Over	  Time	  by	  Arm:	  Critical	  and	  Non-­‐Critical	  Admissions..........................................................................................................................................	  65	  Figure	  10.	  Monthly	  LOS	  per	  Type	  -­‐	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Referral	  to	  PPCP§	  ....	  67	  Figure	  11.	  Monthly	  LOS	  per	  Type	  -­‐	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Post-­‐Referral	  to	  PPCP	  ...	  68	  Figure	  12.	  CPCH	  Arm	  -­‐	  Changes	  in	  the	  Monthly	  LOS	  per	  Type,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................................................	  70	  Figure	  13.	  BCCH	  Arm	  -­‐	  Changes	  in	  the	  Monthly	  LOS	  per	  Type,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................................................	  71	  Figure	  14.	  Sample	  Size	  Estimates	  for	  Different	  Effect	  Sizes	  -­‐	  LOS	  ......................................................	  75	  	   	   xiv Figure	  15.	  Average	  Monthly	  Number	  of	  Admissions:	  Summary	  of	  Comparison	  Between	  Groups	  and	  Changes	  within	  Groups	  from	  Pre-­‐	  to	  Post-­‐Referral	  Period	  **	  ............................................................	  76	  Figure	  16.	  Average	  Monthly	  LOS:	  Summary	  of	  Comparison	  Between	  Groups	  and	  Changes	  within	  Groups	  from	  Pre-­‐	  to	  Post-­‐Referral	  Period	  **	  .....................................................................................	  77	  Figure	  17.	  Total	  Cost	  per	  Person-­‐Month	  at	  Risk	  Over	  Time,	  by	  Group:	  Outpatient	  and	  Inpatient	  Admissions........................................................................................................................................	  101	  Figure	  18.	  Total	  Cost	  per	  Person-­‐Month	  at	  Risk	  Over	  Time	  Over	  Time	  by	  Group:	  Critical	  and	  Non-­‐Critical	  Admissions	  ...........................................................................................................................	  101	  Figure	  19.	  Monthly	  Cost	  per	  Type	  of	  Admission,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup,	  Pre-­‐Referral	  to	  PPCP.	  §	  ..........................................................................................................................................	  107	  Figure	  20.	  Monthly	  Cost	  per	  Type	  of	  Admission,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup,	  Post-­‐Referral	  to	  PPCP.	  §	  ..........................................................................................................................................	  107	  Figure	  21.	  CPCH	  Group	  -­‐	  Changes	  in	  the	  Monthly	  Cost	  per	  Type	  of	  Admission,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................	  108	  Figure	  22.	  BCCH	  Group	  -­‐	  Changes	  in	  the	  Monthly	  Cost	  per	  Type	  of	  Admission,	  by	  Cancer	  and	  Non-­‐cancer	  subgroup	  –	  Pre-­‐Post-­‐Referral	  to	  PPCP§	  .................................................................................	  108	  Figure	  23.	  Sample	  Size	  Estimates	  for	  Different	  Effect	  Sizes	  –	  Total	  Cost	  ..........................................	  111	  Figure	  24.	  	  Direct	  Cost	  per	  Person-­‐Month	  at	  Risk	  Over	  Time,	  by	  Group:	  Outpatient	  and	  Inpatient	  Admissions........................................................................................................................................	  112	  Figure	  25.	  Direct	  cost	  per	  Person-­‐Month	  at	  Risk	  Over	  Time	  Over	  Time	  by	  Group:	  Critical	  and	  Non-­‐Critical	  Admissions	  ...........................................................................................................................	  113	  Figure	  26.	  Average	  Monthly	  Cost:	  Summary	  of	  Comparison	  Between	  Groups	  and	  Changes	  within	  Groups	  from	  Pre-­‐	  to	  Post-­‐Referral	  Period	  **	  ...................................................................................	  122	  	   	   xv Figure	  27.	  Pairwise	  Comparison	  of	  Monthly	  Number	  of	  Admissions	  –	  Entire	  Observational	  Period.	  ..........................................................................................................................................................	  175	  Figure	  28.	  Pairwise	  Comparison	  of	  Monthly	  Number	  of	  Admissions	  –	  Pre-­‐Referral	  Period.	  ...........	  175	  Figure	  29.	  Pairwise	  Comparison	  of	  Monthly	  Number	  of	  Admissions	  –	  Post-­‐Referral	  Period.	  ..........	  176	  Figure	  30.	  CPCH	  Group	  –	  Changes	  in	  Monthly	  Number	  of	  Admissions	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  ..........................................................................................................................................................	  176	  Figure	  31.	  BCCH	  Group	  –	  Changes	  in	  Monthly	  Number	  of	  Admissions	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  ..........................................................................................................................................................	  177	  Figure	  32.	  Pairwise	  Comparison	  of	  Monthly	  LOS	  –	  Entire	  Observational	  Period.	  ............................	  177	  Figure	  33.	  Pairwise	  Comparison	  of	  Monthly	  LOS	  –	  Pre-­‐Referral	  Period.	  ..........................................	  178	  Figure	  34.	  Pairwise	  Comparison	  of	  Monthly	  LOS	  –	  Post-­‐Referral	  Period.	  ........................................	  178	  Figure	  35.	  CPCH	  Group	  –	  Changes	  in	  Monthly	  LOS	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  .........................	  179	  Figure	  36.	  BCCH	  Group	  –	  Changes	  in	  Monthly	  LOS	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  .........................	  179	  Figure	  37.	  Pairwise	  Comparison	  of	  Monthly	  Costs–	  Entire	  Observational	  Period	  ............................	  208	  Figure	  38.	  Pairwise	  Comparison	  of	  Monthly	  Costs	  –	  Pre-­‐Referral	  Period.	  .......................................	  208	  Figure	  39.	  Pairwise	  Comparison	  of	  Monthly	  Costs	  –	  Post-­‐Referral	  Period.	  ......................................	  209	  Figure	  40.	  CPCH	  Arm	  –	  Changes	  in	  Monthly	  Costs	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  ..........................	  209	  Figure	  41.	  BCCH	  Arm	  –	  Changes	  in	  Monthly	  Costs	  –	  Pre-­‐	  to	  Post-­‐Referral	  Period.	  ..........................	  210	  	  	   	   xvi List	  of	  Abbreviations	  ACT:	  Association	  of	  Children’s	  Palliative	  Care	  [Together	  for	  Short	  Lives]	  BC:	  British	  Columbia	  BCCH:	  British	  Columbia	  Children's	  Hospital	  CADTH:	  Canadian	  Agency	  for	  Drugs	  and	  Technologies	  in	  Health	  	  CMAJ:	  Canadian	  Medical	  Association	  Journal	  	  CHPCA:	  Canadian	  Hospice	  Palliative	  Care	  Association	  CIHI:	  Canadian	  Institute	  for	  Health	  Information	  	  CMDB:	  Canadian	  MIS	  Database	  CNPCC:	  Canadian	  Network	  of	  Palliative	  Care	  for	  Children	  CI:	  Confidence	  Interval	  CPCH:	  	  Canuck	  Place	  Children’s	  Hospice	  	  DNAR:	  Do	  Not	  Attempt	  Resuscitation	  EMR:	  Electronic	  Medical	  Records	  EOL:	  End-­‐of-­‐Life	  	  GPs:	  General	  Practitioners	  ICU:	  Intensive	  Care	  Unit	  ICD:	  International	  Statistical	  Classification	  of	  Diseases	  and	  Related	  Health	  Problems	  IT:	  Information	  Technologies	  	  LOD:	  Location	  of	  Death	  LOP:	  Length	  onto	  the	  Program	  LOS:	  Length	  of	  Stay	  LTCs:	  Life-­‐Threatening	  Conditions	  	  	   	   xvii MeSH:	  Medical	  Service	  Headings	  MNPR:	  Mandated	  Nurse-­‐Patient	  Ratios	  NICU:	  Neonatal	  Intensive	  Care	  Unit	  NOS:	  Newcastle-­‐Ottawa	  Scale	  PC:	  Palliative	  Care	  PedPalASCNET:	  Collaborative	  Research	  in	  Pediatric	  Palliative	  Care	  PHSA:	  Provincial	  Health	  Services	  Authority	  PICU:	  Pediatric	  Intensive	  Care	  Unit	  PPC:	  Pediatric	  Palliative	  Care	  PPCP:	  Pediatric	  Palliative	  Care	  Program	  QALY:	  Quality-­‐Adjusted	  Life	  Year	  	  RCT:	  Randomized	  Controlled	  Trials	  Sd:	  Standard	  Deviation	  UK:	  United	  Kingdom	  USA:	  United	  States	  of	  America	  	  	  	  	  	  	  	  	  	  	   	   xviii Acknowledgements	  	  This	  thesis	  could	  not	  have	  been	  possible	  without	  the	  immeasurable	  support	  from	  my	  loved	  ones,	  friends,	  colleagues	  and	  all	  the	  unfamiliar	  faces	  that	  crossed	  my	  pathway	  and	  offered	  a	  little	  help.	  	  	  I	  would	  like	  to	  thank	  my	  supervisor,	  Dr.	  Craig	  Mitton	  for	  the	  incredible	  support	  and	  humour	  he	  has	  provided	  throughout	  this	  experience.	  I	  thank	  Dr.	  Harold	  Siden	  for	  his	  brilliance	  and	  willingness	  to	  teach	  and	  Dr.	  Rollin	  Brant	  for	  his	  continued	  support	  and	  insight.	  	  	  A	  special	  thanks	  to	  Negar	  Chavoshi	  for	  the	  enjoyable	  guidance	  and	  sense	  of	  humour	  facing	  deadlines	  and	  reviews;	  to	  Shanon	  Ederly,	  statistician	  and	  friend,	  who	  supported	  the	  data	  analyses	  with	  brilliant	  insight,	  impressive	  skills,	  and	  an	  endless	  willingness	  to	  help;	  and	  to	  Beth	  Hensler	  for	  all	  her	  hard	  work	  behind	  the	  scenes	  to	  make	  things	  happen	  at	  SPPH.	  	  I	  would	  like	  to	  acknowledge	  the	  contribution	  of	  Madeleine	  Doyle-­‐Waters,	  librarian	  at	  the	  Centre	  for	  Clinical	  Epidemiology	  and	  Evaluation,	  who	  supported	  the	  construction	  and	  optimization	  of	  the	  search	  strategies.	  Peijun	  Sang,	  consultant	  from	  UBC	  Department	  of	  Statistics,	  who	  supported	  the	  statistical	  analyses	  planning.	  Karyn	  Epp	  for	  providing	  assistance	  from	  her	  PHSA	  team	  with	  data	  retrieval.	  Tanice	  Miller	  and	  Joan	  Hill	  for	  their	  commitment	  and	  openness	  to	  improve	  the	  quality	  of	  financial	  data	  in	  the	  hospice.	  	  	  I	  acknowledge	  and	  thank	  all	  of	  you	  for	  assisting	  me	  in	  this	  journey.	  	   	   xix Dedication	  	  I	  dedicate	  this	  work	  to...	  	  God	  first,	  for	  giving	  me	  the	  strength	  to	  pursue	  this	  hard	  journey.	  To	  my	  entire	  family	  with	  a	  special	  feeling	  of	  gratitude	  to	  my	  loving	  parents,	  Rosina	  and	  Francisco	  who	  dealt	  with	  my	  absence	  and	  physical	  distance	  throughout	  difficult	  moments	  of	  loss	  with	  understanding.	  	  To	  Ryan	  and	  the	  MacAulay’s	  for	  grounding	  me	  here	  during	  the	  hard	  times	  of	  grad	  school.	  To	  all	  my	  friends	  worldwide	  for	  making	  me	  feel	  close	  to	  their	  hearts	  even	  when	  I	  was	  far	  away	  from	  their	  eyes.	  	  This	  thesis	  is	  lovingly	  dedicated	  in	  memory	  of	  my	  grandmother,	  Detizia.	   	   1 Chapter	  1: Introduction	  The	  death	  of	  a	  child	  might	  be	  the	  most	  devastating	  tragedy	  a	  family	  can	  face.	  Care	  for	  severely	  sick	  children,	  and	  their	  families,	  is	  central	  to	  pediatrics;	  however,	  only	  recently	  palliative	  care	  (PC)	  for	  the	  pediatric	  population	  has	  been	  considered	  as	  a	  sub-­‐specialty	  within	  pediatrics.	  Consequently,	  the	  most	  suitable	  approaches	  to	  pediatric	  palliative	  care	  (PPC)	  are	  relatively	  in	  the	  early	  stages	  of	  development,	  and	  continuously	  evolving.1	   	  There	  is	  clinical	  interest	  in	  understanding	  how	  the	  different	  care	  settings	  where	  end-­‐of-­‐life	  (EOL)	  care	  is	  provided	  for	  children	  with	  life-­‐threatening	  conditions	  (LTCs)	  have	  a	  differential	  impact	  on	  patients	  and	  their	  families	  (e.g.	  hospital,	  home,	  hospice,	  etc.).	  Furthermore,	  it	  is	  not	  clear	  how	  different	  approaches	  to	  EOL	  and	  management	  of	  symptoms	  for	  LTCs	  across	  providers	  affect	  both	  costs	  and	  health	  care	  utilization.	  It	  is	  known	  that	  a	  disproportionate	  amount	  of	  health	  care	  funds	  are	  being	  spent	  on	  individuals	  in	  their	  final	  months	  of	  life	  2.	  Yet,	  evidence	  to	  informs	  how	  best	  to	  make	  these	  allocations	  are	  scarce.	  There	  is	  a	  prevailing	  belief	  that	  PPC	  programs	  (PPCP)	  can	  deliver	  services	  more	  efficiently	  by	  improving	  care	  offered	  in	  settings	  other	  than	  tertiary	  centres,	  more	  specifically	  in	  hospices	  or	  at	  home.	  Nonetheless,	  how	  to	  reallocate	  resources	  for	  each	  of	  these	  types	  of	  services	  and	  programs	  remains	  an	  outstanding	  question.	  British	  Columbia	  (BC)	  has	  a	  holistic	  PPCP	  carried	  out	  by	  a	  freestanding	  pediatric	  hospice	  that	  coordinates	  the	  care	  plan	  for	  referred	  children	  across	  the	  public	  funded	  health	  care	  settings	  thus	  offering	  a	  natural	  experiment	  to	  study	  such	  questions	  for	  hospice-­‐based	  programs.	  In	  order	  to	  provide	  evidence	  to	  support	  PPCP	  evaluation,	  planning,	  and	  funding,	  a	  combined	  analysis	  of	  data	  from	  the	  published	  literature	  and	  the	  BC	  PPCP	  performed	  to	  study	  the	  effects	  of	  PPCP	  on	  cost	  and	  utilization.	  Chapter	  2	  presents	  a	  systematic	  review	  of	  the	  literature	  investigating	  	   	   2 differences	  in	  patterns	  of	  utilization	  and	  cost	  among	  children	  with	  LTCs,	  who	  enrolled	  in	  PPCP	  compared	  to	  those	  who	  did	  not.	  It	  explores	  if	  and	  how	  research	  was	  conducted	  in	  this	  field,	  and	  compares	  the	  outcomes	  of	  PPCP	  according	  to	  different	  settings.	  Chapter	  3	  presents	  a	  comparison	  of	  the	  utilization	  patterns	  at	  EOL	  among	  children	  who	  died	  while	  enrolled	  in	  the	  provincial	  PPCP	  compared	  to	  those	  who	  were	  not.	  Chapter	  4	  presents	  a	  cost	  analysis	  of	  the	  utilization	  in	  this	  population,	  according	  to	  the	  different	  settings,	  and	  the	  impact	  on	  the	  health	  care	  system.	  	  1.1 Background	  	  1.1.1 Conceptual	  Framework	  Health	  systems	  are	  organized	  in	  such	  a	  manner	  that	  the	  process	  of	  health	  care	  delivery	  affects	  health	  system	  outcome	  and	  consequently	  population	  health	  outcomes	  (Figure	  1).	  Population	  needs,	  access	  to	  care,	  indirect	  effects	  and	  utilization	  of	  the	  health	  system	  affect	  these	  processes	  of	  delivery,	  which	  includes	  the	  organization	  of	  health	  care,	  or	  ways	  resources	  are	  deployed,	  and	  the	  actual	  process	  of	  care.3	  In	  the	  context	  of	  this	  research	  the	  place	  or	  program	  that	  is	  providing	  care	  (home,	  tertiary	  care,	  community	  hospital	  or	  hospice)	  to	  meet	  the	  needs	  of	  pediatric	  population	  with	  LTCs	  will	  affect	  health	  system	  performance.	  Being	  enrolled	  in	  any	  specific	  model	  of	  PPCP	  likely	  has	  an	  effect	  on	  differences	  on	  management	  approach	  (curative	  or	  palliative),	  health	  care	  utilization	  of	  other	  health	  care	  settings,	  and	  a	  variety	  of	  levels	  of	  care	  to	  meet	  patient	  needs.	  	  	   	   3 Figure	  1.	  	  Macro	  Model	  of	  Health	  System	  	  Image	  adapted	  with	  permission	  from	  Analyzing	  Health	  Systems:	  a	  modular	  approach	  @	  Oxford	  University	  Press,	  p.383	  	  	  	  	  1.1.2 Life-­‐threatening	  Conditions	  -­‐	  Definition	  	  According	  to	  the	  Canadian	  Network	  for	  Accessible,	  Sustainable	  and	  Collaborative	  Research	  in	  Pediatric	  Palliative	  Care	  (PedPalASCNET),	  LTCs	  are	  “conditions	  for	  which	  there	  is	  a	  likelihood	  of	  death	  before	  adulthood.	  It	  includes	  those	  for	  which	  curative	  treatments	  may	  be	  feasible	  but	  may	  fail,	  or	  those	  for	  which	  a	  cure	  is	  not	  possible	  and	  from	  which	  an	  affected	  child	  is	  expected	  to	  die.	  They	  are	  frequently	  chronic	  complex	  conditions	  with	  significant	  impact	  upon	  the	  lives	  of	  the	  child	  and	  family”.4	  The	  terminology	  may	  vary	  in	  other	  countries	  where	  they	  can	  also	  be	  defined	  as	  terminal	  or	  life-­‐shortening	  conditions	  (or	  diseases).5	  	  	  1.1.2.1 Categorization	  of	  Life-­‐threatening	  Conditions	  In	  order	  to	  help	  categorize	  these	  conditions,	  the	  advocacy	  organization	  Together	  for	  Short	  Lives	  (previously	  called	  Association	  of	  Children’s	  Palliative	  Care	  -­‐	  ACT),	  delineates	  4	  large	  groups	  of	  LTCs	  (see	  Table	  1	  below).	  This	  categorization	  relies	  exclusively	  on	  diagnosis;	  however,	  severity	  of	  	   	   4 disease,	  impact	  of	  the	  disease	  on	  child’s	  functionality	  and	  family	  dynamics,	  subsequent	  complications,	  and	  level	  of	  care	  required	  should	  also	  be	  considered.5	  	  The	  range	  of	  diagnoses	  under	  these	  categories	  is	  extremely	  wide	  (over	  300	  conditions),	  with	  a	  degree	  of	  overlap	  with	  severe	  disabilities	  and	  complex	  needs.	  	  Although	  cancer	  patients	  constitute	  a	  significant	  proportion	  of	  the	  children	  eligible	  for	  PPCP,	  the	  majority	  of	  the	  workload	  in	  this	  specialty	  is	  applied	  to	  inherited	  metabolic	  disease,	  neuromuscular	  diseases	  and	  acquired	  brain	  injury.	  Approximately	  15%	  of	  these	  children	  do	  not	  have	  a	  definitive	  primary	  and	  obvious	  diagnosis,	  and	  palliative	  care	  is	  usually	  delivered	  over	  a	  longer	  time	  frame	  compared	  to	  adult	  palliative	  care.6-­‐9	  	  	  	  Table	  1:	  Categorization	  of	  LTCs	  Category	  1	   Category	  2	  Life-­‐threatening	  conditions	  for	  which	  curative	  treatment	   may	   be	   feasible	   but	   can	   fail.	  Where	  access	   to	  palliative	  care	   services	  may	  be	  necessary	  when	   treatment	   fails	  or	  during	  an	  acute	  crisis,	  irrespective	  of	  the	  duration	  of	  that	   threat	   to	   life.	   On	   reaching	   long-­‐term	  remission	   or	   following	   successful	   curative	  treatment	   there	   is	   no	   longer	   a	   need	   for	  palliative	   care	   services.	   Examples:	   cancer,	  irreversible	   organ	   failures	   of	   heart,	   liver,	  kidney.	  	  Conditions	   when	   premature	   death	   is	   inevitable,	  where	   there	   may	   be	   long	   periods	   of	   intensive	  treatment	   aimed	   at	   prolonging	   life	   and	   allowing	  participation	  in	  normal	  activities.	  Examples:	  cystic	  fibrosis,	  Duchenne	  muscular	  dystrophy.	  	  Category	  3	   Category	  4	  Progressive	   conditions	   without	   curative	  treatment	   options,	   where	   treatment	   is	  exclusively	   palliative	   and	   may	   commonly	  extend	   over	   many	   years.	   Examples:	   Batten	  disease,	  mucopolysaccharidoses.	  	  Irreversible	   but	   non-­‐progressive	   conditions,	  causing	  severe	  disability,	   leading	  to	  susceptibility	  to	   health	   complications	   and	   likelihood	   of	  premature	   death.	   Examples:	   severe	   cerebral	  palsy,	  multiple	  disabilities	  such	  as	  following	  brain	  or	   spinal	   cord	   injury,	   complex	  health	   care	  needs	  and	   a	   high	   risk	   of	   an	   unpredictable	   life-­‐threatening	  event	  or	  episode.	  Adapted	  with	  permission	  from	  A	  Guide	  to	  the	  Development	  of	  Children’s	  Palliative	  Care	  Services	  ©	  Association	  for	  Children’s	  Palliative	  Care.	  5	  	  	  	   	   5 1.1.3 Pediatric	  Palliative	  Care	  Definition	  and	  Criteria	  The	  movement	  for	  PPCP	  in	  hospices	  started	  to	  be	  developed	  in	  the	  early	  80’s	  in	  the	  United	  Kingdom	  (UK),	  with	  the	  opening	  of	  Helen	  House	  hospice	  in	  Oxford,	  recognized	  as	  the	  pioneer	  in	  this	  field.	  10	  The	  UK	  recognized	  pediatric	  palliative	  care	  as	  a	  medical	  specialty	  in	  2009.	  The	  field	  has	  a	  later	  development	  in	  North	  America	  and	  a	  number	  of	  other	  countries	  in	  the	  mid	  90’s.	  In	  Canada,	  the	  first	  hospital-­‐based	  program	  started	  in	  1986,	  and	  the	  first-­‐hospice	  based	  program	  opened	  in	  1995.11	  PPCP	  provides	  services	  to	  individuals	  with	  LTC	  under	  the	  age	  of	  19.	  Individuals	  aged	  ≥	  19	  years	  are	  considered	  young	  adults.5	  However,	  there	  is	  no	  consistency	  in	  the	  age	  range,	  even	  within	  the	  PPCP	  literature,	  and	  some	  authors	  also	  include	  individuals	  under	  20	  years	  of	  age	  as	  children.12	  	  According	  to	  ACT,	  palliative	  care	  for	  children	  and	  young	  people	  is	  defined	  as	  “an	  active	  and	  total	  approach	  to	  care,	  from	  the	  point	  of	  diagnosis	  or	  recognition,	  throughout	  the	  child’s	  life,	  death	  and	  beyond.	  It	  embraces	  physical,	  emotional,	  social	  and	  spiritual	  elements	  and	  focuses	  on	  the	  enhancement	  of	  quality	  of	  life	  for	  the	  child	  and	  support	  for	  the	  family.	  It	  includes	  the	  management	  of	  distressing	  symptoms,	  provision	  of	  short	  breaks	  for	  the	  caregivers,	  and	  care	  through	  death	  and	  bereavement.	  It	  is	  distinct	  from	  disabled	  children’s	  care	  in	  a	  number	  of	  ways.	  Although	  many	  children	  with	  palliative	  care	  needs	  are	  disabled,	  the	  risk	  or	  certainty	  of	  death	  in	  childhood	  adds	  a	  degree	  of	  complexity	  and	  urgency	  to	  their	  care	  and	  the	  support	  that	  is	  needed	  for	  their	  family.	  And	  compared	  to	  disabled	  children,	  their	  need	  for	  services	  is	  more	  likely	  to	  fluctuate	  due	  to	  the	  particular	  nature	  of	  their	  illness	  trajectory,	  social,	  emotional	  and	  physical	  needs”	  (pg	  7).5	  	  Children	  with	  the	  same	  condition	  may	  require	  PPCP	  during	  different	  periods	  of	  life	  or	  progression	  of	  the	  disease,	  the	  rate	  and	  presentation	  of	  which	  can	  vary	  between	  them.	  In	  the	  same	  way,	  families’	  willingness	  to	  pursue	  treatments	  to	  significantly	  prolong	  life	  can	  range	  from	  	   	   6 supportive	  and	  relieving	  care,	  to	  aggressive	  and	  invasive	  treatments.	  Ideally,	  palliative	  care	  should	  be	  proposed	  from	  the	  moment	  of	  the	  diagnosis	  or	  from	  the	  point	  at	  which	  it	  is	  acknowledged	  that	  curative	  treatment	  is	  not	  available.	  PPCP	  is	  evolving	  and	  practitioners	  are	  increasingly	  recognizing	  that	  each	  child	  and	  their	  family	  will	  need	  an	  individualized	  range	  of	  support	  mechanisms.	  5	  	  Figure	  2	  projects	  the	  relative	  focus	  of	  care.	  The	  dashed	  line	  is	  differentiating	  between	  therapies	  intended	  to	  modify	  disease	  from	  those	  intended