The perinatal transmission of HIV is preventable through a regimen that includes testing of all pregnant individuals, antiretroviral treatment (ART) for the pregnant individual, prophylactic or preventative ART for the infant, and cesarean section delivery for mothers with HIV viremia at the time of delivery. Under this protocol, the United States has seen a significant decline in the perinatal transmission of HIV and achieved a perinatal HIV transmission rate of 0.9% in 2019. However, despite this progress nationally and after zero transmissions in 2021, Maryland recorded 6 cases of perinatal HIV diagnoses in 2022. Each of the 3 major referral centers for pediatric HIV patients in Maryland reported 2 new cases in 2022. A root cause analysis of the cases identified risk factors including delayed entry into perinatal and HIV care, premature birth, maternal adherence challenges in the setting of substance use and other adverse social determinants of health, and failure to diagnose maternal HIV infection in a timely way. All patients were successfully linked to care and initiated on ART. Multiple factors contributed to the 2022 increase in cases of perinatal HIV in Maryland. To achieve and then sustain the elimination of perinatal HIV transmission, the constancy of systems that eliminate barriers for all pregnant people to access testing, prevention, and treatment is critical.