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Proliferative verrucous leukoplakia (PVL) is an aggressive and distinct oral disorder with a high potential for malignant transformation (MT). It presents as multifocal lesions that progress over time and frequently recur, often developing carcinomas. Accurately diagnosing PVL is crucial to distinguish it from other oral mucosa lesions that have a lower risk of cancer progression. However, due to the diverse histological features observed in PVL, identifying clinical criteria and histological patterns that can be applied by unfamiliar professionals is challenging. In this study, we present a case of PVL associated with dysplasia in a 53-year-old female patient. The patient exhibited macular and leukoplakic nonscrapable lesions disseminated throughout the oral cavity, with continuous growth. The diagnosis of PVL was established during an 18-month follow-up. This case highlights the difficulties faced by both clinicians and pathologists in diagnosing PVL, emphasizing the need for careful evaluation and accurate diagnosis, particularly in patients with unusual oral lesions, and highlighting the discrepancies observed in the application of available protocols to our particular case. Distinguishing PVL from similar conditions can be challenging due to overlapping clinical signs and indistinct histopathological features.