Although the majority of children

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1 Long-stay children in intensive care: Long-term functional outcome and quality of life from a 20-yr institutional study Poongundran Namachivayam, MRCPCH, FCICM; Anna Taylor, BN, RN, Post Grad. Dip Women s Hlth; Terence Montague, FRCPCH; Karen Moran, BN, RN; Joanne Barrie, BN, RN; Carmel Delzoppo, BappSc; Warwick Butt, FRACP, FCICM Objective: Long-stay patients ( 28 days) in pediatric intensive care units consume a disproportionate amount of resources, and very few studies have reported their outcome. We determined the long-term outcome of these children admitted to intensive care over a 20-yr period (January 1, 1989 to December 31, 2008). Setting: Pediatric intensive care unit in a university-affiliated tertiary pediatric hospital in Melbourne, Australia Methods: Demographic and clinical characteristics were compared after dividing patients into four groups depending on year of admission ( , , , and ). Preadmission health status and long-term functional outcome were evaluated by a modified Glasgow outcome scale. Quality of life was assessed by using the Health Utilities Index Mark 1. Results: Over the 20-yr period, 233 long-stay patients had 269 long stay admission episodes to the pediatric intensive care unit, accounting for 1% (269 of 27,536) of all pediatric intensive care unit admissions and utilized 18.5% (15,740 of 85,032) of occupied bed days. Bed occupancy of long stay patients (as percentage of overall pediatric intensive care unit bed occupancy) increased from 8% in 1989 to 21% in 2008 (p =.001). Median age at admission was 4.2 months [interquartile range ] and median length of stay was 40 days [interquartile range 32 57]. One hundred sixteen of 233 (49.8%) patients had died at the time of follow-up. Children who died were younger compared to survivors (median 3.4 months [interquartile range vs. median 7.6 months, interquartile range , p =.026], had a higher proportion of comorbid illness (91% vs. 80%, p =.026), and 63% had a preexisting moderate or severe disability compared to 51% of survivors (p =.215). One hundred seventeen of 233 children survived and long-term functional outcome was favorable (normal, functionally normal, or mild disability) in 27% (63 of 233) and unfavorable (moderate or severe disability) for 17.2% (40 of 233). Outcome status was not known for 6% (14 of 233). Among survivors (n = 117), more than 50% (63 of 117) had favorable outcome. The quality of life in patients aged >2 yrs at follow up was good in 21% (40 of 222), moderate in 8% (16 of 222), poor quality in 68% (130 of 222, this includes deaths), and very poor in 3% (5 of 222). Conclusions: More than two-thirds of children who stay in intensive care for 28 days have an unfavorable outcome (moderate disability, severe disability, or death). Long-stay patients in pediatric intensive care utilized a large proportion of resources and this utilization has considerably increased with time. Service provision and policy making should expect worsening of these trends in the future; its effects on critical care bed availability and overall activity levels could be substantial. (Pediatr Crit Care Med 2012; 13: ) Key Words: children; long-stay; outcome; pediatric intensive care Although the majority of children admitted to pediatric intensive care units (PICUs) require a stay of only a few days and have a good outcome, a small proportion of patients require prolonged periods of admission and often utilize considerable amounts of intensive care resources. Long stay patients (length of stay 28 days) in From the Intensive Care Unit (PN, AT, KM, JB, CD, WB), The Royal Children s Hospital, Melbourne, Australia; Department of Anaesthesia (TM), Our Lady children s Hospital, Crumlin, Dublin, Ireland; and Department of Paediatrics (WB), University of Melbourne, Melbourne, Australia. The authors have not disclosed any potential conflicts of interest. For information regarding this article, siva. namachivayam@rch.org.au Copyright 2012 by the Society of Critical Care Medicine and the World Federation of Pediatric Intensive and Critical Care Societies DOI: /PCC.0b013e31824fb989 Australia and New Zealand account for <1.5% of intensive care admissions, but utilize >20 % of intensive care bed days (1). A number of previous studies have reported prevalence, identified risk factors, and predictors for long stay in intensive care (2 4) and have shown increased length of stay as a predictor of poor quality of life (5). Studies have also attempted to report the long-term outcome of these children (2, 6, 7). Pollack et al (2), using long stay as >13 days, reported that this subset of children were significantly younger, had higher prevalence of chronic illness, and had high PICU mortality (17.4%). At 1-yrfollow up 58 % had died or were severely disabled. More recently, Conlon et al (6), defining long stay as >28 days, concluded that the mortality rate is high among long stay patients but long-term health-related quality of life is normal for most. Investigators evaluating subgroups of infants post cardiac surgery have similarly identified risk factors for prolonged length of stay (8) and have also shown that long stay in intensive care is an independent predictor of impaired cognitive function (9). A similar finding of intensive care length of stay associated with abnormal neurodevelopmental outcome has also previously been reported in a diverse group of infants and children post cardiac surgery (10). Critical illness for long stay children might represent only a temporary state; recovery can be prolonged and outcomes vary over time and with illness severity. While technology has allowed us to support organ systems, they do not guarantee a recovery to health and a quality of life consistent with the family s or care provider s expectations. Evaluation of long-term outcome is important as this information will be helpful to expedite services within the intensive care unit, 520 Pediatr Crit Care Med 2012 Vol. 13, No. 5

2 assist in the counseling of families, and can also identify problems after intensive care that can be solved in partnership with other specialties. In two previous studies from Melbourne looking at cohorts of all consecutive admissions to intensive care in 1982 and 1995, we reported that the long-term outcome for 90% children at follow-up was favorable (11, 12) and these children were likely to lead an independent existence. More recently, in another study (13) comparing trends in intensive care outcomes over three decades, we noted an increase in the proportion of survivors with moderate or severe disability from 8.4% in 1982 to 17.9 % in The purpose of this paper is to 1) study the demographic and clinical profile of long-stay patients; and 2) evaluate the long-term functional outcome and quality of life among survivors. METHODS Subjects and Design The study population included all children who stayed 28 days between January 1, 1989, and December 31, 2008, in a tertiary PICU at Royal Children s Hospital in Melbourne, Australia. Children who stayed in intensive care for 28 days were classified as long stay patients in line with criteria set by the Australian and New Zealand Pediatric Intensive Care Registry (14). Potential patients were identified from the database maintained in the PICU. Data pertaining to the study and long-term outcome was collected in a prospective manner. The PICU in Royal Children s Hospital is a 19-bed multidisciplinary unit with a catchment population of about 5.5 million adults and children. The unit admits critically ill children from the states of Victoria, Tasmania, and southern New South Wales. Royal Children s Hospital, which has all major pediatric subspecialties, is the Australian national center for heart transplantation, and provides pediatric extracorporeal life support (ECLS) for children from Victoria and for children from other states. This study was originally started 10 yrs ago and was approved under the Royal Children s Hospital ethics committee rapid approval pathway process. In view of the length of the study, the human research and ethics committee recently confirmed that the review has been carried out according to the current principles governing human research. Data Collection Data for this study were collected from multiple sources, which included the pediatric intensive care database, medical records, and cardiology departmental database. The individual patient details in the intensive care database were recorded and coded according to guidelines produced by the Australian and New Zealand Pediatric Intensive Care Registry (15). The following data were recorded for the study: age at admission, length of stay, principal diagnosis, preadmission health status, reason for admission, comorbidities, treatment modalities used, number of other admissions to intensive care for each patient, and outcome (at discharge from PICU and long-term outcome). Subjects were divided into six major admission categories: cardiovascular, respiratory, neurological, accident/tuma, postoperative (non cardiac), and other medical.the presence of comorbid illness was also identified using International Classification of Diseases, Ninth Revision, Clinical Modification diagnostic codes and previously reported in literature 16. The nine major categories for comorbid illness are: cardiovascular, respiratory, gastrointestinal, renal, neuromuscular, hematology and immunodeficiency, metabolic, malignancy, and other congenital or genetic defect. For children who required more than one long stay admission, only details from the first admission were used in the study. For children who died, the following data were collected: timing of death in relation to the admission, place of death (PICU, ward, or home), and mode of death (elective withdrawal for poor prognosis, brain death, death despite full treatment, and other). Preadmission Health Status and Outcome Assessment Preadmission health status (before the onset of illness leading to PICU admission) and long-term outcome were assessed by research assistants (T.M., A.T., K.M., J.B.) trained in outcome evaluation. Preadmission health status and long-term functional outcome were evaluated using a standardized questionnaire to obtain a modified Glasgow Outcome Score (MGOS) (11). Outcome assessment was conducted by means of telephone interview with the child s parent or guardian. Children who reached adulthood at the time of evaluation were interviewed directly. The functional outcome obtained by the MGOS divides children into the following categories: normal, functionally normal (physically and intellectually normal) but requiring medication or medical supervision, mild disability but likely to lead an independent existence, moderate disability and dependent on care, severe disability and totally dependent on care (including persistent vegetative state), and death. Infants <1 month of age at the time of PICU admission were not included in preadmission health status assessment. Secondly, quality of life in our study was assessed by using the Health Utilities Index Mark 1 by Torrance et al (17). More extended versions of the Health Utilities Index (Mark 2 and Mark 3) have been developed, but were not used in our study as Mark 1 was the standard in the 1980s and 1990s. Four attributes in the Health Utilities Index: Mark 1 were defined (Appendix questionnaire) as follows: 1) mobility/physical activity; 2) self-care/role activity; 3) social/emotional function; and 4) health problems. The different levels within each category were assigned a numerical value and an overall health state utility value (HSUV) was calculated. The HSUV for all possible health states lies between 1.00 and 0.21, where 1.00 is healthy, 0.00 is dead, and negative values reflect a state worse than dead. Four outcome categories were assigned: good (HSUV 1 0.7); moderate (HSUV ); poor (HSUV ); and very poor (HSUV <0). Children <2 yrs of age at the time of follow-up were not assigned a HSUV score. Outcome evaluation was performed after at least a minimum of 6 months post discharge from intensive care. A child is considered lost to follow-up if they are unable to be contacted, refused subsequent consent for interview, are an overseas patient, or do not speak English. The relationship between age, diagnostic category, preadmission health status, and timing of outcome assessment with outcome was studied and presented in the Results section. Statistical Analysis We divided children into four groups (based on the year of admission) to compare demographic and clinical characteristics and mortality rate. Continuous variables are presented as medians with interquartile ranges (IQR) and categorical variables as percentages. Continuous variables were compared with Mann-Whitney U test (for two groups) or Kurskal-Wallis test (for >2 groups), and categorical variables were compared with chisquare test. Survival curves were estimated with the use of Kaplan-Meier product-limit method, and survival distributions between groups were compared with the log rank test. Data on subjects were censored at the time of last follow up or on January 1, Analysis was performed using SigmaPlot version 11 (Systat software, San Jose, CA). All reported p values are two-sided; those under.05 are considered to be statistically significant. RESULTS Characteristics of the Study Population During the study period, 233 patients stayed in intensive care for 28 days. The demographic and clinical characteristics during the four 5-yr periods of study ( , , , and ) are presented in Table 1. The median age at admission for all patients was 4.2 months [IQR months] and 60% (140 of 233) were male. Infants <1 month old at admission accounted for 34% (79 of 233) of study population. Pediatr Crit Care Med 2012 Vol. 13, No

3 Table 1. Demographic and clinical characteristics compared between the four 5-yr periods Variable All Patients (N = 233) (N = 34) (N = 39) (N = 66) (N = 94) p a Male sex n (%) 140 (60) 22 (65) 18 (46) 43 (65) 57 (60.5).24 Age months.99 Median Interquartile range ( ) ( ) ( ) ( ) ( ) Length of stay days Median Interquartile range (32 57) (31 60) ( ) ( ) ( ) No of long-stay admissions Average no of admissions for long-stay 4.1 (978 of (243 of (200 of (258 of (277 of 94 <.001 patients b (average/patient) patients) patients) patients) patients) patients) Diagnostic category no. (%).008 Cardiovascular 128 (55) Respiratory 43 (18) Neurological 21 (9) Other Medical 32 (14) Accident/trauma 7 (3) Postoperative (non-cardiac) 2 (1) 1 1 Long-term follow up, with deaths n (%) 219 (94) 32 (94) 38 (97) 63 (95) 86 (91) Prior moderate-severe disability c 86/150 (57%) 11/23 (48%) 22/30 (73%) 23/40 (57%) 30/57 (53%).21 Comorbid illness n (%) 200 (86) 27 (79) 37 (95) 58 (88) 78 (83).20 Intervention no. (%) <.001 Ventilated, no inotrope 35 (15) 9 (26.5) 12 (30.8) 8 (12.1) 6 (6.4) Ventilated, with inotrope 198 (85) 25 (73.5) 27 (69.2) 58 (87.9) 88 (93.6) Tracheostomy no. (%) 64 (28) Requirement for extracorporeal life 55 (24) support no. (%) Overall pediatric intensive care unit 472 of 7351 (6.4%) 396 of 6514 (6%) 383 of 6823 (5.6%) 256 of 6848 (3.7%) <.001 Mortality rate no. (%) Long-stay patient mortality rate n (%) 23 of 47 (49%) 21 of 46 (46%) 31 of 73 (42%) 41 of 103 (40%).27 a p values were calculated by Mann-Whitney U test, Kurskal-Wallis test, or chi-square analyses; b includes both long stay and non-long stay admissions; c infants <1 month old not assessed for pre-admission health status. Median length of stay in PICU for all patients was 40 days [IQR days] and there was no difference in the length of stay during the four time periods. Among children <1 month of age at admission, the median length of stay was much the same at 38 days [IQR days]. Long stay patients during each time period were prone to PICU readmission and subsequent further episodes of long stay. In , an average of 1.38 admissions (34 patients needing 47 admissions) was noted. This ratio dropped during subsequent years and in , an average of 1.09 admissions (94 patients needing 103 admissions) were needed. The total number of admissions per patient during each period (both long stay and non-long stay) was also estimated and there was a significant downward trend: in , 34 patients accounted for 243 admission episodes (7.1 admission episodes/patient) and in , 94 patients needed 277 admissions (2.9 admission episodes/ patient) to PICU (p.001). However, changes in PICU admission, discharge, and transfer criteria over time could have contributed to these differences. Bed occupancy (occupied bed days) is the length of stay computed in hours and converted into days after dividing by 24. The overall PICU bed occupancy increased from an average of 3,912 days per year in to 5198 days per year in (p.001). Similarly, the bed occupancy for long stay patients also showed a significant (p =.014) increase (515 bed occupancy days per year in and 1,036 in ). Long-stay patients utilized 8% of 4,363 PICU bed days in 1989 (Fig. 1), and in 2008 this increased to 21% of 5,263 PICU bed days, an increase of162 % (p.001). Also, the proportion of longstay admissions (expressed as a percent of all PICU admissions) increased from 0.7% of 1,522 admissions in 1989 to 1.5% of 1,199 admissions in 2008 (p =.048). The major diagnostic category was children with cardiovascular conditions that accounted for 55% (128 of 233) of patients. Children with respiratory illness accounted for 18% of admissions, neurological illness 9%, and other medical conditions accounted for 14% of patients. There was a trend toward an increase in admissions among children with cardiovascular illness from 44% (15 of 34) in to 59 % (56 of 94) in Long-term follow-up data (including deaths) for study patients were available for 94% of patients (219 of 233), and >90% of patients during each study period had outcome information. Among children in whom preadmission health status was recorded, 57% (86 of 150) had moderate or severe disability before the index admission to PICU. This estimate varied between 48% (11 of 23) in to 53% (30 of 57) in (p =.21). The therapeutic intervention needed was calculated based on the need for mechanical ventilation and inotrope support. Of the 233 study patients, 35 (15%) were ventilated (without needing inotropes) and 198 (85%) needed both mechanical ventilation and inotope infusion. In , 74% needed both ventilation and inotrope support and this increased to 94% in (p =.001). Similarly, the need for tracheostomy and requirement for ECLS was also studied. Overall, 28% (65 of 233) of longstay patients received a tracheostomy and there was no significant variation 522 Pediatr Crit Care Med 2012 Vol. 13, No. 5

4 of 233) subjects had comorbid illness and the proportion varied between 79% (27 of 34) in to 83% (78 of 94) in (p =.20).The PICU mortality rate decreased from 6.4% (472 of 7,351) in to 3.7% (256 of 6,848) in (p.001). The PICU mortality rate for long stay patients averaged 40% or more during all four study periods (p =.27). Functional Outcome and Quality of Life Figure 1. Trends in long stay patient bed occupancy and long stay admissions during the study period. PICU, pediatric intensive care unit. Figure 2. Relation between age and diagnostic groups. Long stay children with accident/trauma (n = 7) and noncardiac post operative patients (n = 2) not shown. (p =.55) in the percentage of children who had a tracheostomy over the four time periods of study. Children who were supported with ECLS accounted for 24% (55 of 233) of the study patients and there was a significant increase (p =.004) in the number of long stay patients supported with ECLS over the period of study 6 of 34 (18%) in and 33 of 94 (35%) in Overall, 86% (200 Long-term outcome status (including deaths) was available for 94% (219 of 233) of the study population. One hundred sixteen of 233 (49.8%) children died either during or following their long stay admission. Of the survivors, outcome status was favorable in 27% (63 of 233), who are likely to lead an independent existence: of which 5 (2.1%) were normal, 25 were functionally normal (10.7%), and 33 had mild disability (14.2%). 17.2% (40 of 233) children had an unfavorable outcome and were likely to survive dependant on care by others for activities of daily living, moderate disability in 20 (8.6%) and severe disability in 20 (8.6%). The outcome status for 14 (6%) survivors is not known. Among survivors (n = 117), >50% (63 of 117) had favorable outcome. Age at Admission, Diagnostic Category, and Outcome. The association between age at admission and diagnostic category is shown in Figure 2. Of the 80 infants the in 0 1 month age group, 70 (87.5%) had a cardiac diagnoses. With increasing age, the number of children with a cardiac diagnosis decreased and in children >5 yrs of age at admission it was 35% (16 of 45). Children in the other medical category contributed to 2.3% (2 of 80) in the 0 1-month age group and this increased to 38% (17 of 45) in children >5 yrs of age. The association between admission category and long-term outcome is presented in Table 2. More than 50% of children in each of the diagnostic categories died following their long stay admission, except those admitted with a neurological illness 24% (5 of 21) died. However, among children who survived with neurological illness 24% (5 of 21) had severe disability at long-term assessment and this proportion was higher than that of other diagnostic categories. Outcome as a Function of Preadmission Health Status. The relationship between preadmission health status (n = 143) and the long-term outcome of Pediatr Crit Care Med 2012 Vol. 13, No

5 Table 2. The relationship between reason for admission to pediatric intensive care unit and long-term functional outcome (n = 210) Admission Category Normal and Functionally Normal children evaluated by the MGOS is listed in Table 3. Infants <1 month of age on admission to PICU were not evaluated for preadmission health status. Fifty-six percent (14 of 25) of children with mild disability and 60% (41 of 68) of those with moderate disability prior to admission to PICU and who subsequently had a long stay admission died. Although only 28.5% (4 of 14) children admitted with prior severe disability died, the majority of children (57%, 8 of 14) in this group continued to have severe disability at long-term evaluation. Therapeutic Intervention and Outcome. Association between main mode of intensive care therapy (mechanical ventilation and vasoactive agents) is shown in Table 4. Among children in the ventilated, no inotrope group, 15.6% (5 of 32) had severe disability: these five children Mild Outcome, n (%) Moderate Severe Death Cardiovascular (n = 117) 17 (14.5) 16 (14) 13 (11) 6 (5) 65 (55.5) Respiratory (n = 41) 4 (10) 3 (7) 2 (5) 5 (12) 27 (66) Neurological (n = 21) 3 (14) 7 (33) 1 (5) 5 (24) 5 (24) Other Medical (n = 31) 7 (23) 2 (6) 3 (10) 2 (6) 17 (55) Total (n = 210) 31 (15) 28 (13.2) 19 (9) 18 (8.5) 114 (54.3) Table 3. The relationship between preadmission health status long-term functional outcome (n = 143) Preadmission Health Status Normal and Functionally Normal Mild Outcome, n (%) Moderate Severe Death Normal + functionally 7 (19.4) 8 (22.2) 3 (8.4) 6 (16.7) 12 (33.3) normal (n = 36) Mild disability (n = 25) 4 (16) 4 (16) 2 (8) 1 (4) 14 (56) Moderate disability (n = 68) 7 (10.3) 9 (13.2) 7 (10.3) 4 (5.9) 41 (60.3) Severe disability (n = 14) 1 (7.2) 1 (7.2) 8 (57.1) 4 (28.5) Total (n = 143) 19 (13.3) 22 (15.4) 12 (8.4) 19 (13.3) 71 (49.6) Table 4. The relationship between therapeutic intervention and long-term functional outcome (n = 217) Therapeutic Intervention Normal and Functionally Normal Mild Outcome, n (%) Moderate Severe Death Ventilated, no inotrope (n = 32) 5 (15.6) 9 (28.1) 3 (9.4) 5 (15.6) 10 (31.3) Ventilated, with inotrope (n = 185) 25 (13.5) 23 (12.4) 17 (9.2) 14 (7.6) 106 (57.3) had a combination of severe brain injury (3), cervical cord injury (3), and syndrome association (3). Three of these five children had severe disability on preadmission health evaluation. A higher proportion of children in the ventilated, with inotropes group died than those in the ventilated, no inotropes group (57.3% vs. 31.3%, p =.01). Functional outcome was known for 52 of 55 children who required ECLS during their long stay admission: 69.2% (36 of 52) died, 7.7% (4 of 52) had moderate disability, 9.6% (5 of 52) had mild disability, and 13.5% (7 of 52) were either normal or functionally normal. Outcome in Relation to Timing of Outcome Assessment. Outcome was evaluated at a median of 4 yrs (IQR ) after discharge from PICU. Longterm outcome was known for 103 of 117 survivors following their long-stay admission. Of these, 63% (65 of 103) survived with a favorable outcome (normal, functionally normal, or mild disability) and 37% (38 of 103) survived with unfavorable outcome (moderate or severe disability). The proportion of children with favorable and unfavorable outcome did not change (p =.995) in relation to the timing of outcome assessment (Fig. 3) after discharge from PICU. Thus the proportion of favorable and unfavorable outcome was similar whether outcome was assessed at 0 2, 2 5, 5 10 or yrs after PICU discharge. However, because each patient was only assessed once, individual outcome may have changed overtime. Quality of Life. The Health State Utilities Index was used to assess quality of life in children 2 yrs of age at the time of outcome assessment. Information was available for 191 of 222 (86%) eligible children who could be assessed. According to this measure, 40 (21%) had a good quality of life (HSUV, ), 16 (8%) had moderate quality (HSUV ), and 130 (68%) children (including deaths) had a poor quality (HSUV, ). Five children (3%) had very poor quality of life (HSUV, <0). The association between quality of life and functional outcome among 75 eligible children who had both measures ascertained is shown in Table 5. More than 85% of children who had poor quality of life also had severe functional disability and all subjects (n = 5) with very poor quality of life were severely disabled. Mortality Characteristics of Long- Stay Patients. One hundred sixteen children of 233 (49.8%) with long stay in intensive care died. Fifty-four of 116 (46.5%) were discharged from PICU after their initial long-stay admission but died subsequently. The median age at admission for children who died was 3.4 months (IQR, ). This was significantly lower (p =.026) than that of children who survived their long stay admission (7.6 months [IQR ]). Ninety-one percent (106 of 116) of children who died had a comorbid illness compared to 80% (94 of 117) who survived (p =.026). The median length of stay for children who died (40 days [IQR 31 57]) was similar to that of children who survived (40 days [IQR 33 58]). Among children in whom preadmission health status was available, 51% (39 of 76) of those with moderate or severe disability were alive and 63% (44 of 70) had died (p =.215). Of 116 children who died, cause of death was identified as 524 Pediatr Crit Care Med 2012 Vol. 13, No. 5

6 (9.1 months) and for the cardiology group was 63.7 months (1,911 days). DISCUSSION Figure 3. Relationship between timing of outcome assessment and outcome among survivors. Favorable outcome = normal, functionally normal, and mild disability. Unfavorable outcome = moderate and severe disability. Table 5. Relationship between quality of life and functional outcome among survivors (n = 75). Children < 2 yrs of age at time of follow-up were not assigned a quality of life score Quality of Life Normal and Functionally Normal follows: elective withdrawal for poor prognosis (n = 80), cardiovascular collapse (n = 23), respiratory arrest (n = 2), elective withdrawal for brain death (n = 3), and cause not known in eight (four died at home and four in other hospitals). Place of death: 93 children died in intensive care unit, seven died at home, four in the emergency department, four in Mild Outcome, n (%) Moderate Severe Good (n = 40) 22 (55) 17 (42.5) 1 (2.5) Moderate (n = 16) 1 (6.2) 7 (43.8) 7 (43.8) 1 (6.2) Poor (n = 14) 2 (14.3) 12 (85.7) Very poor (n = 5) 5 (100) the hospital ward, and four in a different hospital. The place of death of four children is not known. Figure 4 shows Kaplan Meier curves for survival for the different admission categories. Log rank test showed a significant survival difference (p =.036) among the different categories. The median (50th percentile) survival for the respiratory group was 275 days This study shows that more than two-thirds (67%) of children who stay in intensive care for 28 days have an unfavorable outcome (moderate disability, severe disability, or death). A very high proportion of long stay patients (86%) have pre existing comorbid illness, and the health care resource utilization for long stay patients in pediatric intensive care has increased considerably over the last two decades as reflected in their PICU bed occupancy--8% in 1989 to 21% in One of the strengths of our study is that it involves a large cohort of long stay patients in intensive care over a 20 yr period, with 94% outcome status known for the entire cohort and among survivors outcome ascertained for 88% (103 of 117). Although it is difficult to achieve high rates of outcome assessment (because of frequent address changes, privacy legislation, families maintaining anonymity), it is important to follow up every child in order to achieve high outcome assessment rates (18), as this enhances the reliability of results and minimizes uncertainty. Many of the outcome calls in our study were as a result of multiple attempts to contact families. Secondly, dividing our cohort into groups based on year of admission was helpful for comparing trends and the long-term follow-up has highlighted the very high mortality and morbidity in this group of children. This study evaluated the long-term functional outcome and quality of life of long stay patients admitted to our PICU over a 20-yr period. Two previous studies of long stay patients have given us varying results. A 2004 study by van der Heide et al (7) was a case control study of long stay patients involving only 19 subjects and reported no difference in mortality and functional outcome compared to controls. Conlon et al (6) looked at long stay children over an 8-yr period and reported high mortality rates while the quality of life for the majority of survivors was normal. However, the study population included preterm neonates and a 64.8% response rate to the survey. In our study, the inclusion of long stay patients over two decades and higher outcome assessment rates allowed for meaningful comparisons and potentially greater generalization to long stay patients in other units. Pediatr Crit Care Med 2012 Vol. 13, No

7 Figure 4. Kaplan-Meier curves for survival time among patients in the different diagnostic groups. Log-rank test showed statistical significance between the survival curves (p =.036). p =.01 between respiratory and neurology groups and p =.08 between cardiac and neurology groups for survival. A recent study looking at national estimates of comorbid illness in the United States showed that 41% of children admitted to critical care in 2006 had a comorbid condition. Presence of a comorbid condition was significantly associated with a longer stay, increased mortality, and greater hospital resource use (19). These children were also at increased risk of repeated admissions to intensive care (20). More than 85% of subjects in our study had a comorbid illness and the proportion was significantly higher among those who died. Successful weaning from mechanical ventilation and other intensive care therapies and even subsequent discharge from PICU for a long stay patient does not always guarantee long term survival, as most of these children have comorbid illness and many with ongoing organ dysfunction and are prone to recurrent hospitalizations. Almost half of all deaths in our study population (54 of 116) involved children who were discharged successfully following their long stay admission, but died subsequently. The overall PICU mortality has declined substantially over the last 20 yrs and during the same time we recorded a significant increase in the number of long stay patients. One possible explanation for this trend could be that patients with serious illness who would have been allowed to die in previous epochs are kept alive now and many of these children continued to have late mortality or experienced disability. Clearly an increase in technology (e.g., ECLS) and also family and society expectations correspond to these changes. Early identification of potential long stay patients would help patients and families understand the implications of disease and potential outcomes, it would allow for proper planning of therapies and also help to rationalize resource utilization. Length of stay is a marker of cost-efficient utilization in intensive care units and a main target for cost containment in health care (3). However, in spite of different predictors of long stay being identified (21), their usefulness in making management decisions is very limited. This possibly relates to multiple factors such as variations in case mix and illness severity, physician experience, prognostic uncertainty, and also the expectations of the family. The ideal study to determine predictors of poor outcome would be a prospective study from multiple intensive care units using a standard definition of long stay (22) Families of children with prolonged intensive care stays experience considerable burdens. Evidence from adult literature suggests that depression, financial hardships, decline in physical health, and high stress levels are very common among families of adults with chronic critical illness (23, 24). Although evidence from pediatric literature is limited, it is reasonable to suggest that similar patterns or more family burdens exist among parents and families of long stay pediatric intensive care patients. Parents of infants who received surgery for hypoplastic left heart syndrome have reported socioeconomic difficulties, traumatic stress, and posttraumatic stress disorder years after the admission (A. M. Cantwell-Bartl, personal communication, 2011). Traumatic stress and post-traumatic stress disorder is common among parents in PICU (25, 26) and may persist well after discharge. More studies looking at the impact on families of long stay patients and their physical and psychological outcome are needed. One of the major implications of our findings is the acceptance that long-stay patients are an integral part of intensive care and their numbers along with resource utilization will continue to increase in the future. As more resources are being spent on long-stay patients, our models of care have changed to accommodate this. When we plan strategy of care, we recognize 15% 20 % of our bed days will be occupied by long-stay patients. We also have long-stay patient coordinators who work closely with the different team members, families and other support services and this has been very instrumental in effective coordination of the care process. Study Limitations Firstly, our study is from a single institution and the genralizability of our findings to other institutions could be questioned. However, previous studies in literature have recorded similar findings (2, 6) in PICU outcomes among long stay patients. While wide variations in outcome may be noted among different institutions, we believe our observed trends would be a reasonable approximation of pediatric intensive care performance at least in the western society. Second, outcome was assessed at only a single time point and there were 526 Pediatr Crit Care Med 2012 Vol. 13, No. 5

8 variations on the timing of outcome assessment. Differences between time of PICU discharge and timing of outcome assessment could potentially result in variations in measured outcome. Improvement in quality of life overtime tends to occur as disease process changes and caregivers get more accustomed to patients needs (27, 28). Having more than one outcome measurement for every patient can give us information regarding temporal changes in functional status and quality of life with time. However, the process of locating a family and gaining an opportunity to communicate can often be very difficult, especially with privacy legislation, frequent address changes, and families willingness to maintain anonymity. Thirdly, availability and utilization of rehabilitation and social services can also influence outcome and quality of life overtime. In the small minority of children who need extensive rehabilitation services, individualized programs designed to achieve specific goals in areas like self care, mobility, and communication with active participation of families and caregivers can have significant impact on measured outcomes. Our institution has a rehabilitation service and this would have impacted on outcomes measured. Future studies looking at long-term outcome after discharge from intensive care should also sample the potential effects of these services on outcomes. Fourth, one could question the reliability of MGOS in assessing functional outcome in infants and children. In view of the diversity of age, generic tools with broad classifications were required to provide global assessments of functional outcome. MGOS is a practical and costeffective method of obtaining health information and also has wide recognition with high clinical relevance (29) along with a very high interrater reliability when used by experienced staff (30). CONCLUSION The number of children requiring prolonged intensive care admission and their resource utilization has increased considerably over the last two decades. More than two-thirds of children requiring 28 days stay in PICU have an unfavorable outcome (moderate or severe disability or death). This major finding has important implications both for the children and families and the healthcare provider. This important information must be considered when talking with families and also for future planning of critical care services. ACKNOWLEDGMENTS We thank Professor Trevor Duke for reading the manuscript and providing useful suggestions. REFERENCES 1. Report of the Australian and New Zealand Paediatric Intensive Care Registry 2009, Pollack MM, Wilkinson JD, Glass NL: Longstay pediatric intensive care unit patients: Outcome and resource utilization. Pediatrics 1987; 80: Marcin JP, Slonim AD, Pollack MM, et al: Long-stay patients in the pediatric intensive care unit. Crit Care Med 2001; 29: Fiser DH, Tilford JM, Roberson PK: Relationship of illness severity and length of stay to functional outcomes in the pediatric intensive care unit: A multi-institutional study. Crit Care Med 2000; 28: Morrison AL, Gillis J, O Connell AJ, et al: Quality of life of survivors of pediatric intensive care. Pediatr Crit Care Med 2002; 3: Conlon NP, Breatnach C, O Hare BP, et al: Health-related quality of life after prolonged pediatric intensive care unit stay. Pediatr Crit Care Med 2009; 10: van der Heide P, Hassing MB, Gemke RJ: Characteristics and outcome of long-stay patients in a paediatric intensive care unit: A case-control study. Acta Paediatr 2004; 93: Brown KL, Ridout DA, Goldman AP, et al: Risk factors for long intensive care unit stay after cardiopulmonary bypass in children. Crit Care Med 2003; 31: Newburger JW, Wypij D, Bellinger DC, et al: Length of stay after infant heart surgery is related to cognitive outcome at age 8 years. J Pediatr 2003; 143: Limperopoulos C, Majnemer A, Shevell MI, et al: Predictors of developmental disabilities after open heart surgery in young children with congenital heart defects. J Pediatr 2002; 141: Butt W, Shann F, Tibballs J, et al: Long-term outcome of children after intensive care. Crit Care Med 1990; 18: Taylor A, Butt W, Ciardulli M: The functional outcome and quality of life of children after admission to an intensive care unit. Intensive Care Med 2003; 29: Namachivayam P, Shann F, Shekerdemian L, et al: Three decades of pediatric intensive care: Who was admitted, what happened in intensive care, and what happened afterward. Pediatr Crit Care Med 2010; 11: Alexander J, Slater A: Australian and New Zealand Paediatric Intensive Care Registry Slater A, Shann F, McEniery J; ANICS Study Group: The ANZPIC registry diagnostic codes: A system for coding reasons for admitting children to intensive care. Intensive Care Med 2003; 29: Feudtner C, Christakis DA, Connell FA: Pediatric deaths attributable to complex chronic conditions: A population-based study of Washington State, Pediatrics 2000; 106: Torrance GW, Boyle MH, Horwood SP: Application of multi-attribute utility theory to measure social preferences for health states. Oper Res 1982; 30: Butt WW: Outcome assessment what outcome do we want? Pediatr Crit Care Med 2009; 10:133 4; discussion Odetola FO, Gebremariam A, Davis MM: Comorbid illnesses among critically ill hospitalized children: Impact on hospital resource use and mortality, Pediatr Crit Care Med 2010; 11: Dosa NP, Boeing NM, Ms N, et al: Excess risk of severe acute illness in children with chronic health conditions. Pediatrics 2001; 107: Slonim AD, Marcin JP, Pollack MM: Long-stay patients: Are there any long-term solutions? Crit Care Med 2003; 31: Carson SS, Bach PB: The epidemiology and costs of chronic critical illness. Crit Care Clin 2002; 18: Van Pelt DC, Milbrandt EB, Qin L, et al: Informal caregiver burden among survivors of prolonged mechanical ventilation. Am J Respir Crit Care Med 2007; 175: Douglas SL, Daly BJ: Caregivers of long-term ventilator patients: Physical and psychological outcomes. Chest 2003; 123: Balluffi A, Kassam-Adams N, Kazak A, et al: Traumatic stress in parents of children admitted to the pediatric intensive care unit. 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9 APPENDIX: OUTCOME ASSESSMENT QUESTIONNAIRE 1. Mobility/Physical Activity a. Does your child have any limitations in regard to walking/running/jumping? Yes/No. If yes, please specify. b. Does your child experience any unexpected breathlessness/tiredness when playing with other children or exercising? Yes/No. If yes, please specify. c. Is help required from other people, or from mechanical aids (wheelchair, frame), for your child to move around? Yes/No. If yes, please specify. d. Parent s perceptions of child s physical abilities in relation to other children of a similar age. 2. Self Care/Role Activity Please circle the most appropriate a. How much help does your child need to eat/dress/bathe/toilet (as age appropriate) 1. No help 2. A small amount of help 3. A moderate amount of help 4. Totally dependent upon help from another person b. Which of the following does your child attend? 1. Kindergarten 2. School Year 3. None c. Does your child need any of the following support? 1. Integration aide 2. Special needs school 3. Rehabilitation (including physiotherapy, occupational therapy, speech therapy) 4. None d. If your child attends school, how much school would your child miss throughout the year? 1. Less than 1 week 2. Between 1 and 2 weeks 3. Between 2 and 4 weeks 4. More than a month e. Parent s perception of child s ability to cope with school level. 3. Social/Emotional Function a. During an average day is your child generally 1. Happy 2. Anxious 3. Depressed 4. Aggressive b. Does your child have any problems in making and maintaining friendships? Yes/No c. Parents concerns with respect to child s behavior. Please specify. 4. Health problems a. Has your child developed any new health problems since your admission to hospital? Yes/No If yes, please specify. b. Has there been any change in the management, or the severity of those health problems that your child experienced prior to admission to hospital? Yes/No. If yes, please specify. c. Does your child require regular follow-up by a specialist doctor? Yes/ No. If yes, how often? d. Does your child require regular medication? Yes/No If yes, please specify e. Does your child experience any pain/ discomfort on a regular basis? Yes/ No. Specify location and frequency. f. Does your child have any vision problems? Yes/No. If yes, please specify. Does your child need to wear glasses? Yes/No g. Does your child have any hearing difficulties or do you have any concerns regarding your child s hearing (exclude selective deafness)? Yes/No Does your child require a technical aide (hearing aide, cochlear implant, other) to hear? Yes/No Did your child have any hearing difficulties prior to admission to hospital? Yes/No h. Does your child have any scars that have healed poorly, or have any other physical problems that cause your child, or you, concern? If yes, please specify. i. Additional comments: 528 Pediatr Crit Care Med 2012 Vol. 13, No. 5