Identifying Barriers to Positive Health Outcomes in Youth with Osteogenesis Imperfecta: A Kids' Inpatient Database (KID) Study

Objective: To assess the impact of extra-skeletal manifestations of Osteogenesis Imperfecta (OI) during pediatric inpatient hospitalizations, and to identify potential social determinants of health, including racial demographics, healthcare costs, and geographic location.

Design: This study utilized the Kids’ Inpatient Database (KID) to investigate national trends in management and inpatient hospital outcomes in patients with OI from 1997-2016. Data regarding inpatient outcomes, cost, and demographics were collected and analyzed.

Setting: Over 4000 U.S. inpatient community hospitals: short-term, non-federal, general, and specialty hospitals.

Participants: Patients under 21 years old, with a diagnosis of OI, who underwent inpatient hospital stays.

Interventions: N/A

Main Outcome Measures: 1. Type and prevalence of extra-skeletal causes of admissions and inpatient complications, stratified by racial demographics. 2. Cost of admissions, stratified by patient’s residency in urban vs. rural settings.

Results: There were 7,291 admissions that listed OI as a diagnosis in the KID database from 1997-2016. The average age at admission was 7.34 ± 0.13 years (95% CI: 7.08-7.60). Of the 2,666 admissions without orthopedic primary diagnoses, single liveborn was the most common diagnosis (21%), followed by pneumonia (7.5%). Of patients with pulmonary hospitalization complications, Black, Latinx, and AAPI patients, were disproportionately affected (35-46%), as compared to white patients (15%), though only respiratory failure and infections met statistical significance (X²=22.88; p<.05). Of the admissions that resulted in death, 33% were due pulmonary issues, most commonly acute respiratory failure, dysplasia of the lung, and pneumonia. Total hospital stay costs did not significantly differ among patients living in urban versus rural areas. Total hospital charges, however, were significantly increased for patients treated at urban, academic centers (p=0.003).

Conclusions: Understanding the extra-skeletal causes, and inpatient complications, of hospitalizations in pediatric patients with OI informs the need for multi-system treatment management, and interdisciplinary care and rehabilitation teams, for the best possible outcomes. Further investigation of the socioeconomic determinants of health should be explored.