An unusual case of recurrent oral cancer in a young woman HPV-negative, non-smoker and non-drinker

Abstract

Event Abstract Back to Event An unusual case of recurrent oral cancer in a young woman HPV-negative, non-smoker and non-drinker Pietro Tozzo1, Federica Canepa2, Khrystyna Zhurakivska3, Claudia Arena3*, Vera Panzarella2 and Olga Di Fede2 1 Azienda Ospedaliera Ospedali Riuniti Villa Sofia Cervello, Department of Stomatology, Italy 2 Università degli Studi di Palermo, Department of Surgical, Oncological and Oral Sciences, Italy 3 University of Foggia, Department of Clinical and Experimental Medicine, Italy Aim. Oral squamous cell carcinomas (OSCCs) represent the most frequent of all oral neoplasms and constitute over 90% of the different types of cancer that can develop in the oral and maxillofacial region. OSCCs usually develop from oral potentially malignant disorders (OPMDs); malignant transformation is a genetic process, which later makes a phenotyping change at the cellular level. Despite advances in the treatment of OSCC, the 5-year survival rate remains approximately 50% due to inability of early detection of OSCC and precursor lesions. Recurrence is an important prognostic factor in these patients; indeed, the incidence of second or multiple primary cancers is reported to range between 10 and 24%, and the 5-year survival rate of the patients who developed second or multiple primary cancers is significantly lower, as compared with the patients who do not develop these tumors. Although “field cancerization” was first described in OSCCs, only few studies have been concentrated on multifocal primary squamous cell carcinomas in the oral cavity. Synchronous carcinomas are defined as second neoplasms at the same time or within 6 months period of primary lesions. After this period, they are considered as metachronous neoplasms. Over the past 30 years, there is a growing incidence of oral and oropharynx cancer among young patients in particular in Western populations. The aim of this paper is to describe a case of a metachronous OSCC in a healthy and young patient with no risk factors (smoking, alcohol consumption and HPV infection). Materials and Methods. We report a case of a woman who has attended our sector of Oral Medicine (University Hospital “Policlinico Paolo Giaccone” Palermo, Italy) since 2008: during this period, she has undergone multiples biopsies with two diagnosis of primary OSCC in two different areas of the tongue. Results. A 45-year-old healthy female patient, with no relevant medical history, no smoker and no heavy drinker, was referred to our outpatient clinic with a chief complaint of non healing lesion in the tongue. An atrofic lesion (0.5 mm) associate with an erosive area with minor bleeding during the inspection with the gauze was observed; the lesion was located between the right margin of the tongue and the floor of the mouth. Brush cytology of the lesion was obtained and no atypical epithelial cells were found, therefore, conservative treatments were decided: chlorexidine gel topic application, the use of a night guard and diet without acid and hot foods for at least 40 days. The patient had undergone monthly follow up: the lesion was stable. After 6 months, due to the modification towards an ulcerative appearance, an incisional biopsy was performed. The histological examination confirmed a diagnosis of low grade dysplasia. The patient regularly attended follow up visits for the next three years and a half in which the lesion clinically improved; during this period oral rinse samples to detect for HPVs, autofluorescence, brush cytology and toluidine blue staining, used as adding tools, were negative. In early 2012, during a follow-up visit, the lesions changed aspect and acquired again an ulcerative feature with raised border: the patient was referred to our plastic surgery department for the complete surgical excision of the lesion. Histological examination showed a leukoplakia with evidence of low grade dysplasia. One year after, a new white lesion appear in the 2/3 anterior part of the tongue. It remained stable until late 2014, when the appearance changed into a non-homogenous lesion: subsequently an incisional biopsy was performed. The histological examination of severe atypia was confirmed and patient was referred again to our plastic surgery department for the complete surgical excision of the lesion. Histological examination confirmed the diagnosis of OSCC in situ. In June 2016, another lesion arose in the posterior part of the tongue, with a verrucous aspect and it was again biopsied. The histological analysis revealed a new OSCC lesion, and the patient received a further surgical therapy. In March 2017, a new ulcerated lesion was detected in the posterior part of the tongue: the histological examination revealed a low-grade dysplasia. Up to date, the patient is still under clinical follow up. Discussion. OPMDs are lesions from which malignancy is more likely to develop that from other tissues. Clinically, OPMDs have a varied clinical manifestation ranging from harmless appearing keratotic white patch to an aggressive red and white lesion with ulceration. The potential for malignant transformation of OPMDs is estimated by determining the degree of dysplastic changes in the epithelium: dysplasia grading is still considered the gold standard for diagnosing OPMDs. Chances of malignant transformation increases with increasing severity of dysplasia thus, influencing the clinical decision-making. Surveillance of oral epithelial dysplasia results in a number of newly diagnosed cases of OSCC, an advantage because of the early treatment of the lesion that usually enable simple and minor surgical intervention. However, over the past 30 years, increasing numbers of patients worldwide are being diagnosed with OSCC at a younger age: in particular tongue cancer in young white women with no risk factors (smoking, alcohol consumption, HPV infection) as the patient described in our study. Evidence suggests that young-onset OSCC patients have a clinically different phenotype compared to older patient; however, the true impact of young age on OSCC clinical behavior will remain difficult to determine unless multi-institutional databases will be combined. The rising incidence of young-onset OSCC mandates intensification of research into its etiology, clinical phenotype and optimal management. In the meanwhile, primary prevention campaigns which include healthy and young patients could be a chance for early detection of OPMDs or OSCC.