Abstract

This paper aimed to identify the use of technology and to analyze the cost of hospital care for children and adolescents with medical complex chronic conditions at a public federal hospital specialized in high-complexity pediatric care, and was performed concomitantly with a prospective cohort study conducted over a one-year period. It included 146 patients with complex medical chronic conditions and 37 non-chronic patients. The analysis showed that most patients had, on average, two hospitalizations a year and were diagnosed with diseases related to at least two organic systems. Catheters, drains and gastrostomy were the most common technologies used. Median direct costs of patients with medically complex chronic conditions were higher than those of non-chronic patients when comparing the use of technology. The study shows high hospitalization cost to these patients. Technology use and hospitalization care costs documentation yields more data to support decision-makers in the planning, managing, and financing of pediatric health policies.

Key words
Child; Youth; Chronic disease; Cost and cost analysis

Introduction

In 2003, the World Health Organization addressed the need for reorganizing health systems care to people with chronic health conditions, considering the transformations in the epidemiological scenario¹1 World Health Organization (WHO). Innovative care for chronic conditions: building blocks for action: global report. Buildig blocks for action. WHO: Geneva; 2002.. Within this group, it is urgent to recognize the complex health care needs of children and adolescents with limited physical or mental functions, which require multi-professional care coordination among various health sectors, with particular emphasis on rehabilitation, dependence on multiple medications and feeding, breathing and excreting technology²2 McPherson M, Arango P, Fox H, Lauver C, McManus M, Newacheck PW, Perrin JM, Shonkoff JP, Strickland B. A new definition of children with special health care needs. Pediatrics 1998;102(1 Pt. 1):137-140.

3 Cohen E, Kuo DZ, Agrawal R, Berry JG, Bhagat SK, Simon TD, Srivastava R. Children with medical complexity: an emerging population for clinical and research initiatives. Pediatrics 2011;127(3):529-538.^-44 Newacheck PW, Kim SE. A national profile of health care utilization and expenditures for children with special health care needs. Arch Pediatr Adolesc Med 2005;159(1):10-17.. These groups are recognized in the literature as Children with Special Health Care Needs, Technology-Dependent Children, Children with Complex Medical Conditions²2 McPherson M, Arango P, Fox H, Lauver C, McManus M, Newacheck PW, Perrin JM, Shonkoff JP, Strickland B. A new definition of children with special health care needs. Pediatrics 1998;102(1 Pt. 1):137-140.^,44 Newacheck PW, Kim SE. A national profile of health care utilization and expenditures for children with special health care needs. Arch Pediatr Adolesc Med 2005;159(1):10-17.

5 Simon TD, Berry J, Feudtner C, Stone BL, Sheng X, Bratton SL, Srivastava R. Children with complex chronic conditions in inpatient hospital settings in the United States. Pediatrics 2010;126(4):647-655.^-66 Arrue AM, Neves ET, Magnago TS, Cabral IE, Gama SG, Hokerberg YH. Translation and adaptation of the Children with Special Health Care Needs Screener to Brazilian Portuguese. Cad Saude Publica 2016;32(6):e00130215.. In this paper, we use the term Chronic Complex Health Condition (CCC) to categorize children and adolescents, which allow the synthesis of situations triggered by life with chronicity: symptoms duration greater than twelve months, compromising one or more organic systems simultaneously and requiring specialized pediatric care in tertiary care centers.

CCCs are life-long and produce costly situations to patients and their families. They demand family and community adaptations, and an efficient health system³3 Cohen E, Kuo DZ, Agrawal R, Berry JG, Bhagat SK, Simon TD, Srivastava R. Children with medical complexity: an emerging population for clinical and research initiatives. Pediatrics 2011;127(3):529-538.^,44 Newacheck PW, Kim SE. A national profile of health care utilization and expenditures for children with special health care needs. Arch Pediatr Adolesc Med 2005;159(1):10-17.^,77 Burns KH, Casey PH, Lyle RE, Bird TM, Fussell JJ, Robbins JM. Increasing prevalence of medically complex children in US hospitals. Pediatrics 2010;126(4):638-646.

8 Burke RT, Alverson B. Impact of children with medically complex conditions. Pediatrics 2010;126(4):789-790.

9 Castro EK, Piccinini CA. Implicações da doença orgânica crônica na infância para as relações familiares: algumas questões teóricas. 2002. Psicol Reflex Crit 2002;15(3):625-635.^-1010 Leite MF, Gomes IP, Leite MF, Oliveira BRG, Rosin J, Collet N. Condição crônica na infância durante a hospitalização: sofrimento do cuidador familiar. Ciên Cuid Saude 2012;11(1):51-57.. The “chronic conditions” category includes a group of CCC children and adolescents whose prevalence is increasing due to reduced child mortality, access to new technologies, and improved social and health markers⁷7 Burns KH, Casey PH, Lyle RE, Bird TM, Fussell JJ, Robbins JM. Increasing prevalence of medically complex children in US hospitals. Pediatrics 2010;126(4):638-646.^,1111 Vieira MA, Lima RAG. Crianças e adolescentes com doença crônica: convivendo com mudanças. Rev Latinoam Enfermagem 2002;10(4):55-60.. In developed countries, it is estimated at approximately 16% in the population up to 18 years of age⁴4 Newacheck PW, Kim SE. A national profile of health care utilization and expenditures for children with special health care needs. Arch Pediatr Adolesc Med 2005;159(1):10-17.. Thirty years ago, children with these conditions would not survive¹²12 Hall DE. The care of children with medically complex chronic disease. J Pediatr 2011;159(2):178-180.. However, we can observe that they may be treated and indeed survive with medically fragile profiles and special health care needs³3 Cohen E, Kuo DZ, Agrawal R, Berry JG, Bhagat SK, Simon TD, Srivastava R. Children with medical complexity: an emerging population for clinical and research initiatives. Pediatrics 2011;127(3):529-538..

CCC children and adolescents are high users of technology and human resources through prolonged hospitalizations. The discharge processes involve many negotiations with stakeholders in other sectors of society - family, the Judiciary, primary health care, and social assistance services¹³13 Moreira MCN, Albernaz LV, Sá MRC, Correia RF, Tanabe RF. Recomendações para uma linha de cuidados para crianças e adolescentes com condições crônicas complexas de saúde. Cad Saude Publica 2017;33(11):1-13.. These patients are technology-dependent and with a fragile clinical situation during hospitalization, and subject to instability and deteriorated condition. Moreover, although they are a smaller portion of the population, this CCC group accounts for a significant proportion of hospital expenditure⁴4 Newacheck PW, Kim SE. A national profile of health care utilization and expenditures for children with special health care needs. Arch Pediatr Adolesc Med 2005;159(1):10-17.. Thus, estimating the cost of health care has been an object of analysis in developed countries¹⁴14 Berry JG, Hall M, Neff J, Goodman D, Cohen E, Agrawal R, Kuo D, Feudtner C. Children with medical complexity and Medicaid: spending and cost savings. Health Aff (Millwood) 2014;33(12):2199-2206.^,1515 Cohen E, Berry JG, Camacho X, Anderson G, Wodchis W, Guttmann A. Patterns and costs of health care use of children with medical complexity. Pediatrics 2012;130(6):e1463-1470..

The health emergency of the Zika virus epidemic in Brazil in 2015 highlights this discussion, considering that babies born with microcephaly or the various diseases caused by Congenital Zika Syndrome will be part of the group of children growing up with CCC¹⁶16 Brasil P, Calvet GA, Siqueira AM, Wakimoto M, de Sequeira PC, Nobre A, Quintana MS, Mendonça MC, Lupi O, Souza RV, Romero C, Zogbi H, Bressan CS, Alves SS, Lourenço-de-Oliveira R, Nogueira RM, Carvalho MS, Filippis AM, Jaenisch T. Zika Virus Outbreak in Rio de Janeiro, Brazil: Clinical Characterization, Epidemiological and Virological Aspects. PLoS Negl Trop Dis 2016;10(4):e0004636.. The Ministry of Health estimates that 29% of Brazilian municipalities have reported cases of microcephaly and nervous system changes¹⁷17 Brasil. Ministério da Saúde(MS). Centro de Operações de Emergências em Saúde Pública sobre Microcefalias. Monitoramento dos casos de microcefalias no Brasil. Informe Epidemiológico, 33 - Semana Epidemiológica de 26/2016 (26/06 a 02/07/2016). Brasília: MS; 2016..

This paper contributes to the systematization and analysis of data on the cost of healthcare for hospitalized CCC children and adolescents and the use of health technologies. These conditions demand a wide range of health services that are challenging to the Brazilian Unified Health System (SUS). These new and emerging health problems, which encompass multiple complexities, call for a definition of adequate resources for professional training, programming on life-support technology demands, networking, and adequate funding¹²12 Hall DE. The care of children with medically complex chronic disease. J Pediatr 2011;159(2):178-180..

The Brazilian literature systematizing patterns of technology use and medical costs is still scarce. Discussing these patterns in the group of CCC children and adolescents becomes an important issue given the relevance of their participation in the health sector budget in both national and international settings¹⁴14 Berry JG, Hall M, Neff J, Goodman D, Cohen E, Agrawal R, Kuo D, Feudtner C. Children with medical complexity and Medicaid: spending and cost savings. Health Aff (Millwood) 2014;33(12):2199-2206.^,1818 Ananth P, Melvin P, Feudtner C, Wolfe J, Berry JG. Hospital Use in the Last Year of Life for Children With Life-Threatening Complex Chronic Conditions. Pediatrics 2015;136(5):938-946.. This study is part of broader research that systematized the hospital morbidity profile of admitted children and adolescents, and aimed to calculate the direct cost of medical care of a cohort of hospitalized CCC patients.

Methods

This is a retrospective analysis of a cohort of CCC patients conducted between May 2014 and April 2015. All patients aged 0 to 18 years hospitalized for 12 months at the Fernandes Figueira National Institute for Women, Child and Adolescent Health (IFF), a federal public hospital specialized in pediatric care, located in and covering the city of Rio de Janeiro, were included. The hospital is a reference center for children and adolescents with rare diseases in Brazil. These diseases can be clinically identified as CCC, which means that research conducted in this setting may reflect its national demands. The institution’s Research Ethics Committee approved the study.

The chronic or non-chronic condition of the child was defined on admission. Any non-chronic patient undergoing technology-assistance procedures, such as tracheostomy and gastrostomy, would be included in the CCC group. Patients were identified from the classification proposed by Feudtner et al.¹⁹19 Feudtner C, Hays RM, Haynes G, Geyer JR, Neff JM, Koepsell TD. Deaths attributed to pediatric complex chronic conditions: national trends and implications for supportive care services. Pediatrics 2001;107(6):E99. and updated by the International Classification of Diseases - 10^th Revision (ICD-10). A new version of that classification was published in 2014, adapting CCC diagnosis to ICD-10. Three new categories were added, namely, diagnoses of origin in the neonatal period, transplants, and technology dependence²⁰20 Feudtner C, Feinstein JA, Zhong W, Hall M, Dai D. Pediatric complex chronic conditions classification system version 2: updated for ICD-10 and complex medical technology dependence and transplantation. BMC Pediatr 2014;14:199.. This update was confirmed in this study by consulting pediatric experts with experience in medical care for children and adolescents diagnosed with CCC.

Patients readmitted and transferred from other sectors of the hospital - general pediatrics outpatient clinic, specialized outpatient clinics, pediatric neonatal and intensive care units, pediatric surgery and infectious diseases wards, and semi-intensive and inpatient units - were included in the period of cohort follow-up. Thus, CCC patients were divided into the following groups, as per the ICD-10: respiratory, metabolic, neuromuscular, cardiovascular, renal, gastrointestinal, hematological diseases, immunodeficiencies, psychiatric disorders, congenital or genetic disabilities, and cancer.

Then, from the clinical profile, the study population was subdivided into: i. Patients using technologies indicative of complexity: tracheostomy, gastrostomy, use of drains (chest, peritoneal or subgaleal) and catheters (peritoneal ventricle shunt, deep venous or peripheral insertion), use of oxygen therapy, invasive mechanical ventilation, special infant formulas, and antibiotic therapy; ii. Number of diagnoses as per the ICD-10 groups associated with CCC; iii. Origin of patients readmitted or transferred from other sectors of the hospital; and iv. Clinical outcomes: discharge with and without technology, hospital stay until the end of the study, and hospital death.

We estimated the direct medical cost from the SUS perspective. We applied the cost per patient method, which considers each patient path through different services. The absorption costing per hospital cost center system allowed the valuation of health resources. The total direct cost per patient was calculated, considering the daily hospital cost of each service used by the patient. The cost items included tests (laboratory and imaging), materials, medicines, special milk formulas, human resources, use of oxygen therapy and overhead costs (electricity, telephone, water, cleaning, safety, collection of chemical and environmental waste and laundry). The time horizon was one year and did not consider the period before and after the beginning and end of the cohort. No discounts or inflationary adjustments were applied due to the short time span. The cost refers to 2016 Brazilian Reais (R$).

CCC patients and non-chronic patients’ costs were compared and included, based on the use of the following medical technologies prescribed for both groups: special dairy formulas, antibiotic therapy, oxygen therapy, noninvasive ventilation, and imaging tests (x-ray, tomography, ultrasound, and echocardiography). We hypothesized that CCC patients would have higher health resource consumption than non-chronic patients and, therefore, higher costs.

The total direct cost per patient was aggregated and shown, in absolute values, as the total direct median cost of care of CCC patients and non-chronic patients. Costs were displayed as median, minimum and maximum values, whereas categorical variables were expressed as absolute frequencies and percentages. We carried out regression analyses using the Generalized Estimating Equations (GEE) model to verify the impact of comorbidities, patient origin, length of stay, and procedures on the total hospitalization cost. We apply the logarithmic transformation to fit the above model as the response variable, in this case, the total direct cost of the CCC patients did not have a Normal distribution. The Kolmogorov-Smirnov test was used to assess the normality assumption. The SPSS 18 (SPSS Inc. Release VCSI. SPSS Statistics for Windows, version 18, IL, USA) and R 3.2.2. (R Core Team. Foundation for Statistical Computing V, Austria, version 3.2.2) software were used for data analyses with a significance level of 5%.

Results

A total of 183 patients were included during the study period, of which 146 (79.8%) had CCC. Non-chronic patients totaled 37 individuals (20.2%), with 241 hospitalizations. The CCC group had a median age of one year and a median hospital stay of 11 days (mean: 35 days; standard deviation: 72 days). Table 1 shows that of the total CCC patients, 35.7% had diseases involving at least two organic systems, 62.4% three systems, and approximately 92% had two yearly hospitalizations. The most frequent hospitalizations were recorded among patients diagnosed with genetic or congenital defects (78.1%), respiratory (64.4%), and neuromuscular (50%) diseases. The use of drain or catheter (39.7%) and gastrostomy (15.8%) were the most commonly employed technologies during hospitalization.

Table 2 shows the comparison between CCC patients and non-chronic patients, and the total median cost of the first group was higher, ranging from 60% for hospitalization for the diagnosis of respiratory diseases, to 657% for tomography. The cost of procedures performed during treatment ranged from 133% to 339% between the two groups.

Regarding CCC patients, the regression model results showed that the length of stay (LOS), the number of systems affected, the patient’s origin service, the outcomes and the need for technologies at hospital discharge significantly increased the cost. The total median cost was directly proportional to the length of hospital stay (Figure 1a) and highly sensitive to the number of diseases/organic systems affected (Figure 1b), and the highest cost was for CCC patients diagnosed with respiratory and metabolic diseases (Figures 1c and 1d). The other categories of diseases classified by ICD-10 (neuromuscular, cardiovascular, renal, gastrointestinal, hematological or immune deficiency, psychiatric disorders, and genetic or congenital disabilities and cancer) were not statistically significant for the total median cost.

The cost of CCC patients from two pediatric inpatient units - semi-intensive and inpatient - was higher than estimated for the other hospital sectors. The lower cost was associated with patients from specialized outpatient clinics when compared to those from inpatient units (Figure 2a). Patients who remained hospitalized at the end of the study period recorded the highest total median cost among the outcomes studied, followed by those who died and those who were discharged (Figure 2c). This is probably due to the median LOS of 77.5 days, 21.5 days, and 10 days for hospitalization, death, and discharge, respectively. The total median cost of patients requiring technologies at hospital discharge was also high (Figure 2c).

Discussion

The median age of one year in the sample reflected the predominance of congenital conditions and pointed to the complexity of the cases, as there was a significant burden associated with CCC, evidenced by the presence of three or more diagnoses, a scenario also observed in the American program Medicaid¹⁴14 Berry JG, Hall M, Neff J, Goodman D, Cohen E, Agrawal R, Kuo D, Feudtner C. Children with medical complexity and Medicaid: spending and cost savings. Health Aff (Millwood) 2014;33(12):2199-2206.. It is noteworthy that almost all patients (97%) recorded up to three hospitalizations during the study period, and the literature shows that one of the leading predictive variables for pediatric hospitalizations, readmissions, and hospital expenses are admissions with two or more CCCs⁷7 Burns KH, Casey PH, Lyle RE, Bird TM, Fussell JJ, Robbins JM. Increasing prevalence of medically complex children in US hospitals. Pediatrics 2010;126(4):638-646.^,1515 Cohen E, Berry JG, Camacho X, Anderson G, Wodchis W, Guttmann A. Patterns and costs of health care use of children with medical complexity. Pediatrics 2012;130(6):e1463-1470..

The cost of medical care for CCC patients was higher than non-chronic patients cost, and proportionally increased concerning the LOS, as expected. Respiratory and metabolic diseases were the CCC groups responsible for the highest cost. In our study, cystic fibrosis, a progressive multisystemic disease, accounted for the largest group of metabolic diseases, which could explain the higher proportion of costs as observed in other countries²¹21 van Gool K, Norman R, Delatycki MB, Hall J, Massie J. Understanding the costs of care for cystic fibrosis: an analysis by age and health state. Value Health 2013;16(2):345-355.. Cystic fibrosis patients and other complex conditions, including those with technology dependence, survive longer than in the past and have special health care needs requiring significant financial, hospital and community resources¹²12 Hall DE. The care of children with medically complex chronic disease. J Pediatr 2011;159(2):178-180.^,2121 van Gool K, Norman R, Delatycki MB, Hall J, Massie J. Understanding the costs of care for cystic fibrosis: an analysis by age and health state. Value Health 2013;16(2):345-355..

In this study, the total median cost of patients who died was higher than the discharged, suggesting different patterns of chronicity and complexity and more intensive use of health resources. The study by Miller et al.²²22 Miller RL, Gebremariam A, Odetola FO. Pediatric high-impact conditions in the United States: retrospective analysis of hospitalizations and associated resource use. BMC Pediatr 2012;12:61. corroborates this result by showing that patients’ LOS was inversely proportional to clinical severity when compared to extremely severe and moderately severe patients, resulting in a higher total cost for the former. While we did not aim to estimate the “cost of death”, it is worth reflecting on the challenges and constraints imposed by clinical complexity and the resources required to address them in the daily scenario. The sentence “death has a cost” points to two issues: the costs to families concerning the demands of comprehensive and permanent care, and the therapeutic obstinacy of health professionals supported by technological and therapeutic advances. The prolonged lifetime may require measures that are sometimes necessary to meet the needs of some patients, but they could be extraordinary for others²³23 Diniz D. Quando a morte é um ato de cuidado: obstinação terapêutica em crianças. Cad Saude Publica 2006;22(8):1741-1748..

Regarding the result of the higher total median cost among patients with CCC from semi-intensive care units and inpatients, future studies would benefit from discussing the path that includes birth in adverse situations leading to technological dependence and the realization of surgical procedures, extending hospital stays over the years. This is reflected in this study when we compare the patients’ origin, understood as their entry point in the research, with the result of the total cost of medical care.

The hospital under study is equipped with a semi-intensive unit working as a prolonged mechanical ventilation weaning unit, which prevents pediatric intensive care beds from being blocked by mechanical ventilation-dependent CCC patients. Consequently, we believed that the cost could be even higher had they remained in intensive care, potentially avoiding access to acute and unstable patients²⁴24 Traiber C, Piva JP, Fritsher CC, Garcia PC, Lago PM, Trotta EA, Ricachinevsky CP, Bueno FU, Baecker V, Lisboa BD. Profile and consequences of children requiring prolonged mechanical ventilation in three Brazilian pediatric intensive care units. Pediatr Crit Care Med 2009;10(3):375-380. who would benefit most from this specialized care. This issue highlights the need for the organization of services to care for this group of mechanical ventilation-dependent CCC patients. It also demands the articulation and organization of the in-hospital, outpatient and emergency health services network, which is confronted with the health needs of the pediatric population in the state of Rio de Janeiro²⁵25 Duarte JG, Gomes Júnior SC, Pinto MT, Gomes MAS. Perfil dos pacientes internados em serviços de pediatria no município do Rio de Janeiro: mudamos? Physis 2012; 22(1):199-214..

This study has some limitations. It was performed in a single tertiary hospital, which hinders generalization of the results and restricts their comparison with other lower complexity hospitals. However, it has been argued⁷7 Burns KH, Casey PH, Lyle RE, Bird TM, Fussell JJ, Robbins JM. Increasing prevalence of medically complex children in US hospitals. Pediatrics 2010;126(4):638-646. that the hospital is the best locus to observe and analyze the increasing number of CCC children and adolescents since this group has frequent hospital readmissions, and CCCs can be developed during their illness. A second limitation refers to the time horizon since the total median cost was calculated for one year. Some cohort patients were already hospitalized before the start of the study, and prior use of resources was not considered. Patients also demand lifelong resources and outpatient visits, and the cost to families was not included. This component should be incorporated into future research as it is higher for technology-dependent children¹⁵15 Cohen E, Berry JG, Camacho X, Anderson G, Wodchis W, Guttmann A. Patterns and costs of health care use of children with medical complexity. Pediatrics 2012;130(6):e1463-1470.^,2626 Benneyworth BD, Gebremariam A, Clark SJ, Shanley TP, Davis MM. Inpatient health care utilization for children dependent on long-term mechanical ventilation. Pediatrics 2011;127(6):e1533-1541.

27 Costa MT, Gomes MA, Pinto M. Chronic dependence on mechanical pulmonary ventilation in pediatric care: a necessary debate for Brazil's Unified Health System. Ciên Saude Colet 2011;16(10):4147-4159.^-2828 Anderson D, Dumont S, Jacobs P, Azzaria L. The personal costs of caring for a child with a disability: a review of the literature. Public Health Rep 2007;122(1):3-16.. It is also noteworthy that the total median cost increased with the number of diseases and systems affected. We did not aim to compare this cost by discriminating which systems were affected, but instead to understand how complexity could affect technology use, LOS, and cost. As a result, the outcomes may be underestimated, and we suggest that future research in this field be expanded in Brazil in order to identify the cost from the perspective of society.

It is worth pointing out the relevance of planning dehospitalization actions in the care line for these patients, considering the cost generated by technological dependence and repercussions on the health system and families. Other demands are also added, such as access to social security benefits, transportation services for continuous, out-of-home treatment, and home adaptations¹³13 Moreira MCN, Albernaz LV, Sá MRC, Correia RF, Tanabe RF. Recomendações para uma linha de cuidados para crianças e adolescentes com condições crônicas complexas de saúde. Cad Saude Publica 2017;33(11):1-13.. Dehospitalization programming can be a complex process including multidisciplinary approaches, involving different health care levels to ensure that each patient remains healthy, develops, and receives support for ongoing care at home²⁹29 Elias ER, Murphy NA. Council on Children with D. Home care of children and youth with complex health care needs and technology dependencies. Pediatric 2012;129(5):996-1005.. In the discharge planning, the emotional burden imposed on families when taking on the daily home care of CCC patients should also be considered.

In Brazil, a change in the pediatric hospital profile has been observed over the last decades²⁵25 Duarte JG, Gomes Júnior SC, Pinto MT, Gomes MAS. Perfil dos pacientes internados em serviços de pediatria no município do Rio de Janeiro: mudamos? Physis 2012; 22(1):199-214.^,2727 Costa MT, Gomes MA, Pinto M. Chronic dependence on mechanical pulmonary ventilation in pediatric care: a necessary debate for Brazil's Unified Health System. Ciên Saude Colet 2011;16(10):4147-4159.. A transition to “new pediatrics”³⁰30 Moreira ME, Goldani MZ. Child is the father of man: new challenges for child health. Ciên Saude Colet 2010;15(2):321-327. faces health professionals to new challenges. As the prevalence of CCC increases globally, it is important to document health resource utilization and cost, even at the local level, and to understand the obstacles to plan the health system for CCC children and adolescents effectively.

References

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