This case explores clinicopathologic correlation in a rare presentation of odontogenic myxoma (OM), emphasizing the association between clinical manifestations and pathological features. A 25-year-old male presented with lower left jaw pain. Clinical examination, imaging, and histopathological analysis led to the diagnosis. Findings revealed a unique correlation between clinical, radiographic, and histopathological features, underscoring the importance of a multidisciplinary approach. Increased alkaline phosphatase activity was noted. The case enriches the literature on OM, enhancing understanding of its etiologies.