Abstract
There has been a significant increase in cases of oral squamous cell carcinoma (OSCC) in young adults, unrelated to smoking, alcohol consumption, or high-risk human papilloma virus (HPV). This article presents the case of a 39-year-old woman who denied habits such as smoking and alcohol consumption. She presented with a red plaque with ulcerated areas, located on the left border of the tongue, measuring 3 cm at its largest diameter, with an 8-year evolution characterized by periods of exacerbation and remission, accompanied by a burning sensation in the area. Under the hypothesis of oral erythroplakia (OE), an incisional biopsy was performed, and histopathological analysis revealed severe epithelial dysplasia, disruption of the basement membrane, and discrete epithelial islands on the surface of the adjacent connective tissue, which tested positive for pancytokeratin in immunohistochemical analysis. The diagnosis was microinvasive OSCC, and the patient underwent surgical treatment, with the diagnosis confirmed in the surgical specimen, and is currently disease-free. The importance of early diagnosis and histopathological examination in OE is highlighted to prevent progression to OSCC, an entity increasingly present in young adults without associated risk factors. We reiterate that further studies, including genetic studies, may clarify and explain cases of OSCC and potentially malignant lesions in young adults, such as the one reported here.
References
Sung H, Ferlay J, Siegel RL, Laversanne M, Soerjomataram I, Jemal A, Bray F. Global Cancer Statistics 2020: GLOBOCAN Estimates of Incidence and Mortality Worldwide for 36 Cancers in 185 Countries. CA Cancer J Clin. 2021;71(3):209-49. doi:10.3322/caac.21660
Maroun CA, Zhu G, Fakhry C, Gourin CG, Seiwert TY, Vosler PS, et al. An immunogenomic investigation of oral cavity squamous cell carcinoma in patients aged 45 years and younger. Laryngoscope. 2021;131(2):304-11. doi:10.1002/lary.28674
Louredo BV, Vargas PA, Pérez-de-Oliveira ME, Lopes MA, Kowalski LP, Curado MP. Epidemiology and survival outcomes of lip, oral cavity, and oropharyngeal squamous cell carcinoma in a southeast Brazilian population. Med Oral Patol Oral Cir Bucal. 2022;27(3):e274-e284. doi:10.4317/medoral.25147
Toporcov TN, Znaor A, Zhang ZF, Yu GP, Winn DM, Wei Q, et al. Risk factors for head and neck cancer in young adults: a pooled analysis in the INHANCE consortium. Int J Epidemiol. 2015;44(1):169-85. doi:10.1093/ije/dyu255
Ng JH, Iyer NG, Tan MH, Edgren G. Changing epidemiology of oral squamous cell carcinoma of the tongue: A global study. Head Neck. 2017;39(2):297-304. doi:10.1002/hed.24589
Muller S, Tilakaratne WM. Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Tumours of the Oral Cavity and Mobile Tongue. Head Neck Pathol. 2022;16(1):54-62. doi:10.1007/s12105-021-01402-9
Zafereo ME, Xu L, Dahlstrom KR, Viamonte CA, El-Naggar AK, Wei Q, et al. Squamous cell carcinoma of the oral cavity often overexpresses p16 but is rarely driven by human papillomavirus. Oral Oncol. 2016;56:47-53. doi:10.1016/j.oraloncology.2016.03.003
Warnakulasuriya S, Ariyawardana A. Malignant transformation of oral leukoplakia: A systematic review of observational studies. J Oral Pathol Med. 2016;45(3):155-66.
Aguirre-Urizar JM, Lafuente-Ibáñez de Mendoza I, Warnakulasuriya S. Malignant transformation of oral leukoplakia: Systematic review and meta-analysis of the last 5 years. Oral Dis. 2021;27(8):1881-95.
de Freitas Silva BS, Batista DCR, de Souza Roriz CF, Silva LR, Normando AGC, Dos Santos Silva AR, et al. Binary and WHO dysplasia grading systems for the prediction of malignant transformation of oral leukoplakia and erythroplakia: a systematic review and meta-analysis. Clin Oral Investig. 2021;25(7):4329-40. doi:10.1007/s00784-021-04008-1
Dutra MJ, Alves Santos K, Adorno-Farias D, Anbinder AL, Kaminagakura E. Proliferative verrucous leukoplakia mimicking oral lichen planus: case report and literature review. Odontoestomatologia. 2023;25(42):e419. doi: 10.22592/ode2023n42e419
van der Waal I. Potentially malignant disorders of the oral and oropharyngeal mucosa; terminology, classification and present concepts of management. Oral Oncol. 2009 Apr-May;45(4-5):317-23. doi: 10.1016/j.oraloncology.2008.05.016.
Dutra MJ, Malta IS, de Almeida Lança ML, de Vasconcellos LMR, Adorno-Farias D, Jara JA, et al. Effects of artemisinin and cisplatin on the malignant progression of oral leukoplakia. In vitro and in vivo study. J Cancer Res Clin Oncol. 2024 Aug 18;150(8):390. doi: 10.1007/s00432-024-05924-x.
Roza ALOC, Kowalski LP, William WN Jr, de Castro G Jr, Chaves ALF, Araújo ALD, et al. Oral leukoplakia and erythroplakia in young patients: a systematic review. Oral Surg Oral Med Oral Pathol Oral Radiol. 2021;131(1):73-84. doi:10.1016/j.oooo.2020.09.002
Warnakulasuriya S, Johnson NW, van der Waal I. Nomenclature and classification of potentially malignant disorders of the oral mucosa. J Oral Pathol Med. 2007;36(10):575-80. doi:10.1111/j.1600-0714.2007.00582.x
Warnakulasuriya S. Clinical features and presentation of oral potentially malignant disorders. Oral Surg Oral Med Oral Pathol Oral Radiol. 2018;125(6):582-90. doi:10.1016/j.oooo.2018.03.011
Allen K, Farah CS. Patient perspectives of diagnostic delay for suspicious oral mucosal lesions. Aust Dent J. 2015;60(3):397-403. doi:10.1111/adj.12246
Patel MA, Blackford AL, Rettig EM, Richmon JD, Eisele DW, Fakhry C. Rising population of survivors of oral squamous cell cancer in the United States. Cancer. 2016;122(9):1380-7. doi:10.1002/cncr.29921
Costa V, Kowalski LP, Coutinho-Camillo CM, Begnami MD, Calsavara VF, Neves JI, et al. EGFR amplification and expression in oral squamous cell carcinoma in young adults. Int J Oral Maxillofac Surg. 2018;47(7):817-23. doi:10.1016/j.ijom.2018.01.002
Adorno-Farias D, Morales-Pisón S, Gischkow-Rucatti G, Margarit S, Fernández-Ramires R. Genetic and epigenetic landscape of early-onset oral squamous cell carcinoma: Insights of genomic underserved and underrepresented populations. Genet Mol Biol. 2024;47 Suppl 1:e20240036. doi:10.1590/1678-4685-GMB-2024-0036
Kaminagakura E, Tango RN, Cruz-Perez D, Bonan R, Yamamoto de Almeida L, de Almeida Lança ML, et al. Oral squamous cell carcinoma outcome in adolescent/young adult: Systematic review and meta-analysis. Head Neck. 2022;44(2):548-61. doi:10.1002/hed.26940
Cury SS, Miranda PM, Marchi FA, Canto LMD, Chulam TC, Petersen AH, et al. Germline variants in DNA repair genes are associated with young-onset head and neck cancer. Oral Oncol. 2021;122:105545. doi:10.1016/j.oraloncology.2021.105545
Dutra MJ, Schuch LF, Loureiro FJA, Silveira FM, Lopes MA, Martins MD, et al. Germline Variations in Patients With Oral Squamous Cell Carcinoma-Systematic Review. Oral Dis. 2025;20. doi: 10.1111/odi.70040.
Sridharan G, Alex S, Bhandare PR, Patankar SR. Microinvasive oral squamous cell carcinoma—A clinicopathological study. Med Res Arch. 2017;5:1-11.
Saldivia-Siracusa C, Araújo AL, González-Arriagada WA, Nava FJ, Hunter KD, Lopes MA, et al. Histopathological parameters reported in microinvasive oral squamous cell carcinoma: a systematic review. Med Oral Patol Oral Cir Bucal. 2022;27:e25675. doi:10.4317/medoral.25675
Pentenero M, Navone R, Motta F, Marino R, Gassino L, Broccoletti R, et al. Clinical features of microinvasive stage I oral carcinoma. Oral Dis. 2011;17(3):298-303. doi:10.1111/j.1601-0825.2010.01740.x
Gerardo G, Eduardo P, René P. Oral epithelial reactive atypia/dysplasia: An underestimated true atypia/dysplasia? Med Hypotheses. 2020;144:110217. doi:10.1016/j.mehy.2020.110217
Ferreira E Costa R, Leão MLB, Sant'Ana MSP, Mesquita RA, Gomez RS, Santos-Silva AR, et al. Oral squamous cell carcinoma frequency in young patients from referral centers around the world. Head Neck Pathol. 2022;16(3):755-62. doi:10.1007/s12105-022-01441-w
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
Copyright (c) 2025 Mateus José Dutra, Gina Pennacchiotti Vidal, Marcela Hernández, Juan Pablo Aitken-Saavedra, Daniela Adorno-Farias