468 - Med costs in infants with trisomy 18 and 13: A population-based cohort study in Japan

Publication Number: 2456.468

Kana Fukui, National center for child health and development, Okura, Setagaya area,, Tokyo, Japan; Eri Hoshino, ​National Center For Child Health And Development, Tokyo, Tokyo, Japan; Tetsuya Isayama, National Center for Child Health and Development, Setagaya-ku, Tokyo, Japan

Background: More than 70% of children with trisomy 18 (T18) and trisomy 13 (T13) have congenital malformations, such as cardiovascular or gastrointestinal anomalies, that often require surgery. In recent years, advances in surgical and intensive care have improved long-term survival. However, information on the healthcare costs associated with these conditions remains limited.

Objective: To assess the medical costs for children with T18 and T13 and identify factors associated with higher costs.

Design/Methods: This retrospective cohort study included children with T18 and T13 born between 2010 and 2016 identified from the Japanese National Database (NDB). The NDB contains detailed nationwide medical claims data in Japan, including diagnoses and medical treatments such as prescriptions, examinations, and surgeries for both inpatients and outpatients, covering nearly the entire population. We excluded children who were not followed for more than one year. To identify factors associated with three-year medical costs (total inpatient, outpatient, and drug costs), we used a generalized linear regression model adjusted for sex, preterm birth, severe neonatal asphyxia, and malformations. Analyses were conducted from the healthcare payer's perspective.

Results: A total of 1,058 children with T18 and 294 children with T13 were included. The mean three-year medical costs were USD 104,244 for T18 and USD 117,417 for T13. Medical costs were higher among those with preterm birth, severe neonatal asphyxia, ventilator use, cardiac malformations, and surgery in T18, and among those with preterm birth, ventilator use, and surgery in T13 (Table 1). For both groups, costs were lower among children who died within 1 or 3 years. After adjustment for the above confounders, preterm birth, ventilator use, severe malformations, and surgery remained factors associated with higher medical costs in both T18 and T13 (Table 2). The adjusted mean cost differences between infants with and without surgical intervention were USD 126,469 for T18 and USD 127,083 for T13.

Conclusion(s): Surgical intervention, preterm birth, and ventilator use were significantly associated with higher medical costs. These findings are useful for policy makers to consider adequate financial support for families and hospitals caring for these medically complex infants, while balancing treatment decisions with considerations of survival, quality of life, and family preferences.

Table 1: Medical cost with trisomy 18 and trisomy 13 by patient characteristic
Abbreviations: SD, standard deviation; USD, US dollar.

Table 2: Predictors of medical cost with trisomy 18 and trisomy 13 using generalized liner regression models
Abbreviations: CI, confidence interval; USD, US dollar.
Adjusted mean cost differences was adjusted for potential confounders (sex, preterm birth, severe neonatal asphyxia, gastrointestinal or cardiac malformation).