This is the first reported case of medication-related osteonecrosis of the jaw (MRONJ) resulting from the use of romosozumab, a new anti-resorptive medication that functions through inhibition of sclerostin. It was approved by the FDA in 2019, and mentioned in the 2022 AAOMS Position Paper on MRONJ. Although initial evidence from animal studies and clinical trials showed a low risk of MRONJ from romosozumab, recent publications on adverse event monitoring demonstrate the need for continued observation and research. Little is known regarding the risk of MRONJ from romosozumab, however, oral surgeons will inevitably be first line clinicians to manage patients on this medication. The purpose of this extremely unusual case report is to improve knowledge and heighten awareness about the association between romosozumab and MRONJ.