A peculiar case of multiple cancerizations in a patient with oral potentially malignant disorder showing overlapping features (proliferative verrucous leukoplakia / oral lichen planus) during 18-year follow-up
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1
University of Parma, Department of Medicine and Surgery, Italy
Aim. Oral lichen planus (OLP) is a chronic inflammatory disease of oral mucosa characterized by the occurrence of multiple and symmetrical lesions. The classic form (reticular form) is often asymptomatic. The erosive form of OLP is characterized by mucosal erosions and pain OLP is classified as potentially malignant disorder on the basis of the sporadic development of dysplasia and/or carcinoma within the context or in the proximity of the mucosal lesion. In particular, erythematous lesions are somewhat more associated with cancerization mostly in patients with Hepatitis C virus (HCV) infection. It can sometimes be difficult to distinguish the clinical and/or histological lesions of OLP from oral lichenoid lesions (OLL).
Proliferative verrucous leukoplakia (PVL) is a rare potentially malignant disorder with a high rate of cancerization. PVL shows multiple white lesions which tend to spread o the adjacent apparently normal oral mucosa areas. OLP (especially plaque-type) and PVL may rarely coexist or, one of these diseases seem in the years to evolve in the other. Therefore, in such peculiar cases, differential diagnosis can sometimes be very difficult. The case reported here highlights the importance of periodic close clinic control for cases displaying overlapping features in order to perform very early diagnosis of possible malignant lesions.
Materials and Methods. A 90-year-old man affected by arterial hypertension, at anamnesis reporting seropositivity for HBV and HCV, has been followed since October 2000 in several centers (Bologna, Milan, Modena and Parma) for the presence of multiple erythro-leukoplakic oral lesions. The patient did not smoke and/or drink alcohol. The first diagnosis of OLP was rendered at San Paolo Hospital in Milan (October 2000). The first carcinoma was surgically removed in June 2002. During 18-year follow-up, 12 different oral carcinomas were diagnosed. Such malignant lesions developed in different areas of the oral cavity such as left and right floor of the mouth, lower alveolar crests, right cheek mucosa, left and right lateral border of the tongue. The patient underwent several biopsies to monitor the histopathological nature of lesions in order to highlight as soon as possible the presence of dysplasia.
During follow-up some of these lesions underwent malignant transformation and had been subsequently surgically removed. Of all the malignant lesions some were followed by one or more recurrences. During follow-up, cervical lymph node metastases were detected both in the right and left sides (2002 and 2004), leading to bilateral neck dissection.
Results. Taking into account the multiple localizations of the leukoplakic lesions, it is reasonable to make a retrospective diagnosis of PVL. Such a pathology can easily lead to the development of multiple malignant lesions. Eight of twelve carcinomas were histologically characterized as G1 (non-infiltrating); 3 as G2 and 1 G3. All the malignant lesions were squamous cell carcinoma. Particularly, 3 lesions were localized on the floor of the mouth and lower alveolar crest, 3 on the right cheek mucosa, 5 on the lateral borders of the tongue (3 on the right, 2 on the left). The last developed on the base of the tongue. Radical excision of malignancies was achieved in all cases.
Discussion. The clinicopathologic findings of the present case are remarkable. The patient did not have known etiologic factors, and PVL is usually a disease affecting female patients. It is essential to highlight here the importance of frequent, close, period follow-up visit in order to achieve a diagnosis of malignant transformation as soon as possible. Through such a scheme, it is perhaps possible to obtain an improvement in the quality of life as well as a longer survival.
References
1. Pentenero M, Meleti M, Vescovi P, Gandolfo S. “Oral proliferative verrucous leucoplakia: are there particular features for such an ambiguous entity? A systematic review”. Br J Dermatol. 2014 May;170(5):1039-47.
2. Mester A, Lucaciu O, Ciobanu L, Apostu D, Ilea A, Campian RS. “Clinical features and management of oral lichen planus (OLP) with emphasis on the management of hepatitis C virus (HCV)-related OLP”. Bosn J Basic Med Sci. 2018 Aug 1;18(3):217-223.
3. Aghbari SMH1, Abushouk AI2, Attia A3, Elmaraezy A3, Menshawy A3, Ahmed MS4, Elsaadany BA5, Ahmed EM6. “Malignant transformation of oral lichen planus and oral lichenoid lesions: A meta-analysis of 20095 patient data”. Oral Oncol. 2017 May;68:92-102.
Keywords:
Oral diseases,
Oral lichen planus (OLP),
Leukoplakia,
Proliferative verrucous leukoplakia,
Squamous cell carcinoma,
verrucous carcinoma
Conference:
5th National and 1st International Symposium of Italian Society of Oral Pathology and Medicine., Ancona, Italy, 19 Oct - 20 Oct, 2018.
Presentation Type:
Poster Presentation
Topic:
Oral Diseases
Citation:
Frontera
M,
Ghidini
G,
Meleti
M,
Manfredi
M and
Vescovi
P
(2019). A peculiar case of multiple cancerizations in a patient with oral potentially malignant disorder showing overlapping features (proliferative verrucous leukoplakia / oral lichen planus) during 18-year follow-up.
Front. Physiol.
Conference Abstract:
5th National and 1st International Symposium of Italian Society of Oral Pathology and Medicine..
doi: 10.3389/conf.fphys.2019.27.00049
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Received:
02 Nov 2018;
Published Online:
09 Dec 2019.
*
Correspondence:
Dr. Marco Frontera, University of Parma, Department of Medicine and Surgery, Parma, Emilia-Romagna, 43121, Italy, marco.frontera@studenti.unipr.it