Introduction: Familial hypercholesterolemia (FH) is a common genetic disorder, but fewer than 10% are appropriately diagnosed. Use of electronic health records (EHRs) can potentially improve diagnosis and treatment.Systemwide strategies using the EHR can identify patients who may have FH with the goal of improving communication and preventive treatment.Methods: Our analysis included patients identified as having possible FH utilizing an LDL-C ≥190 mg/dL screen in the EHR. Patients were followed for 6 months, and lipid levels were measured at baseline, 6 weeks, 4 months, and 6 months. Changes in lipid levels were analyzed using paired sample t-tests for normalized data or Wilcoxon Rank Sum Test for skewed data. Our previously reported qualitative data were collected from 5 focus groups, 34 semi-structured key informant interviews, and open-ended survey items from patients and primary care clinicians before and after EHR messages about FH were sent.Results: In 12 patients, the LDL-C (25.3 ± 39.4 mg/dL, 18.7 ± 29.1%; p = 0.029) and total cholesterol/HDL-C (3.8 ± 1.0, 3.2 ± 0.9; p = 0.040) decreased at 4 months after lipid lower therapy initiation. In our previous work, which provides context to these findings, key themes identified were that patients trusted FH resources from the EHR and sought outside resources to educate themselves and their family, empower themselves, and improve their health. Yet, patients did not feel empowered due to the perceived lack of educational resources. Patients concluded that the physician-patient relationship via an EHR facilitates sustainable behavioral changes and treatment adherence. They also found that using the EHR to communicate with clinicians and to get laboratory results was helpful.Leveraging the EHR to identify patients with FH can lead to better lipid management. Patients and clinicians can identify methods to improve diagnostic processes and communication channels in the EHR.