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Basal Cell Adenoma of the Salivary Gland Original Article Case Report Arch Clin Exp Surg 2016;5:246-249 Archives of Clinical doi:10.5455/aces.20140304011652 Experimental Surgery Increased of Langerhans Cells in Smokeless Tobacco-Associated Oral Mucosal Lesions Basal cell adenoma of the salivary gland: Cribriform type, a rare case with review of literature Raghunath Prabhu1, Neha Kumar2, Sakshi Sadhu1, Rajgopal Shenoy1, Kuladeepa Ananda Vaidya3 Érica Dorigati de Ávila1, Rafael Scaf de Molon2, Melaine de Almeida Lawall1, Renata Bianco Consolaro1, Alberto Consolaro1 ABSTRACT Basal cell adenoma (BCA) of the salivary glands is a rare benign tumor resembling pleomorphic adenoma, but with a prominent basaloid cell layer. The majority of these tumors arise in the parotid glands and account for only 1% of all salivary gland epithelial tumors. We report one such case of a swelling in the foor of the mouth in a 55-year-old female where BCA 1 is the most likely diagnosis; however, histological variation does show a similarity to malignant adenoid cystic carcinoma, Bauru Dental School therebyAbstract making the diagnosis difcult. The incidence of malignancy is relatively higher in the submandibular, sublingual University of São Paulo and minor salivary glands. Approximately, 85% of sublingual gland tumors are malignant. Thus, we should be more careful Bauru–SP, Brazil whenObjective: making a diagnosis To evaluate in minor the salivary changes gland in tumors.the number of Langerhans Cells (LC) observed in the epithelium of 2 smokeless tobacco (SLT-induced) lesions. Araraquara Dental School Key words: Adenoid cystic carcinoma, adenoma, basal cell, clinicopathological feature, cribriform, excision, immunohisto- São Paulo State University chemistry, minor salivary gland, oral cavity Methods: Microscopic sections from biopsies carried out in the buccal mucosa of twenty patients, who were Araraquara-SP, Brazil chronic users of smokeless tobacco (SLT), were utilized. For the control group, twenty non-SLT users of SLT Received: February 05, 2012 Introductionwith normal mucosa were selected. Te sections weremorphic studied adenoma with routine and malignant coloring adenoidand were cystic immunostained car- Accepted: February 29, 2012 Basalfor S-100, cell adenomas CD1a, Ki-67 (BCAs) and arep63. rare T occurrencesese data were statisticallycinoma, should analyzed be present by the as Student’s these tumors t-test o tofen investigate have the Arch Clin Exp Surg 2012;X: X-X and dimostferences of them in usually the expression arise from of the immune parotid glandsmarkers inhistological normal mucosa similarities, and inespecially SLT-induced if relying leukoplakia on cytol- lesions. DOI: 10.5455/aces.20120229052919 (80%)[1]. Other sites, such as the lips, palate and na- ogy alone. Here, we present a case of one such diagnos- Results: Tere was a signifcant diference in the immunolabeling of all markers between normal mucosa sal septum have also been encountered. Tis condition tic uncertainty in a 55-year-old woman who presented Corresponding author and SLT-induced lesions (p<0.001). Te leukoplakia lesions in chronic SLT users demonstrated a signifcant usually afects people in the ffh decade of life, with a with an oral tumor, which was found to be a BCA, most Érica Dorigati de Avila Departamento de Estomatologia higherincrease prevalence in the in number women ofthan Langerhans men. Histologically, cells and in thelikely. absence of epithelial dysplasia. da Faculdade de Odontologia de a basaloidConclusion: cell layer Tsurroundede increase by in hyaline the number substance of these is cellsCase represents Report the initial stage of leukoplakia. Bauru noted.Key T words:e diferentiating Smokeless factor tobacco, for leukoplakicthis tumor, lesions,when cancer,A langerhans 55-year-old cells, woman chewing non-smoker, tobacco. presented to Universidade de São Paulo (USP) compared to other benign mixed tumors and salivary the surgical department with 2 years history of a pain- Avenida Alameda Octávio gland neoplasms is the absence of myoepithelial cells. less swelling in the foor of the mouth. Te swelling Pinheiro Brizola, 9-75, 17012-901 A strongIntroduction suspicion of other diagnoses, such as pleo- was slowlycontact growing with in size. the She oral reported mucosa no associated and creates a Bauru–SP, Brasil [email protected] Author afliations : 1Department of Surgery, Kasturba Medical College, Manipal University,more Manipal, Karnataka, alkaline India, environment, 2Foundation Year 1, University its Hospitalproducts of may AmongNorth tobacco Stafordshire users, NHS Trust, there London, isUnited a false Kingdom, be 3Department- of Pathology, Srinivasa Institute of Medical Sciences and Research Centre, Mukka, Mangalore, Karnataka, India Correspondence : Raghunath Prabhu, MD, Department of Surgery, Kasturba Medical College,even Manipal be University, more Manipal, aggressive Karnataka, India. to tissue [5]. Te lief that SLTe-mail: is [email protected] because it is not burned, Receivedwhich / Accepted leads : January many 14, 2014 people / March 04, to2014 quit cigaretes percentage of SLT users is lower compared and start using SLT [1]. However, SLT con- to cigarete users; however, usage is increasing tains higher concentrations of nicotine than among young individuals and it is therefore a cigaretes and, in addition, nearly 30 carci- signifcant and disturbing danger [6,7]. nogenic substances, such as tobacco-specifc Initial studies on the efects of SLT on the N-nitrosamines (TSNA), which is formed oral mucosa demonstrated the formation of during the aging process of the tobacco, [2-4] white lesions induced by chronic exposure to and which presents high carcinogenic poten- tobacco, characterized by epithelial thicken- tial. Moreover, because the tobacco has direct ing, increased vascularization, collagen altera- 247 Prabhu R et al. Tere was no evidence of infltration of the adjacent muscles or median raphe. No hemorrhage or calcifca- tion was observed within the lesion. Te right subman- dibular gland was normal in size and signal intensity. A few sub-centimeter lymph nodes were seen in the level 1 B region. An excision biopsy of this lesion was planned. In- Figure 1. An oval swelling measuring 4 cm × 3 cm was seen on the tra-operatively there was no infltration of the tumor to foor of the mouth on the right side, not crossing the midline. the surrounding muscles and a wide local excision with 0.5 cm margin was performed (Figure 3). She was then discharged post an uneventful recovery and is on regu- lar follow-up. Te histopathology (HPE) reported an encapsulat- ed tumor composed of small basaloid cells arranged in Figure 2. Magnetic resonance imaging of the head and neck showed a well-defned, lobulated heterogeneously enhancing T1 iso-hypointense and T2 hyperintense lesion measuring 3.5 cm × 2.9 cm × 3.2 cm in the right sublingual space. features such as xerostomia, sialorrhea, or pain. Tere was no difculty in the movement of the tongue. Her general well-being was otherwise unremarkable, and oral hygiene was good. Her background included hy- pertension and type 2 diabetes mellitus, as well as clini- cal depression. On examination, all her vitals were stable. An oval TXFMMJOHNFBTVSJOHଙDN¤ଙDNXBTTFFOPOUIFĚPPS of the mouth on the right side, not crossing the mid- line (Figure 1). Te mucosa over the swelling appeared Figure 3. Wide local excision of the tumor with 0.5 cm margin. normal, and the trans-illumination test was negative. Tere was no tenderness over the swelling and no local rise of temperature. It was frm to hard in consistency and was non-fuctuant, but was mobile in all directions. No local lymph nodes were palpable. Her blood results were all within normal limits (full blood count, urea, and electrolytes). Subsequently, she underwent an ultrasound scan of the neck, which showed a well-defned hypoechoic lobulated lesion with smooth margins in the right submandibular re- gion deep to the muscles. Magnetic resonance imaging of the head and neck showed a well-defned, lobulated heterogeneously enhancing T1 iso-hypointense and 5IZQFSJOUFOTFMFTJPONFBTVSJOHଙDN¤ଙDN¤ 3.2 cm in the right sublingual space (Figure 2). Non- Figure 4. Histopathology revealed an encapsulated tumor composed enhancing necrotic areas were seen within the tumor. of small basaloid cells arranged in tubules and a cribriform pattern. Archives of Clinical and Experimental Surgery Year 2016 | Volume 5 | Issue 4 | 246-249 Basal cell adenoma of the salivary gland 248 tubules and a cribriform patern. Te cells were found Te common minor salivary gland tumors such as the to have basophilic nuclei and scant cytoplasm, con- pleomorphic adenoma, BCA, adenoid cystic carcino- taining densely eosinophilic basement membrane like ma, and low grade adenocarcinoma present with simi- material within the tubules. Tese were surrounded by lar morphological and immunohistochemical features hyalonized stroma with thin walled blood vessels and to a large extent, thereby making a preoperative diagno- occasional lymphoplasmacytic infltrate. No perineu- sis difcult. An excision biopsy would be ideal to clinch ral invasion was seen (Figure 4). Te conclusion from the diagnosis [8]. Te presence of the cribriform pat- that report was of a high likelihood of the lesion being tern in this case makes it a rare case and relatively dif- a BCA of the salivary gland. Te absence of perineu- fcult to distinguish from the adenoid cystic carcinoma. ral invasion and good circumscription with a capsule Te cribriform type has a higher female predilection, ruled out adenoid cystic carcinoma in this case. How- afect the elderly, tend to be well-circumscribed and do ever, immunohistochemistry (IHC) staining with C- not recur afer a local excision [9]. IHC plays a key role kit favored a diagnosis of adenoid cystic carcinoma. in diferentiating these types of tumors from the ade- In view of this, a regular follow-up of the patient was noid cystic carcinoma. Tese types of tumors are nega- suggested. tive for MMP9, laminin, CD117, and myoepithelial Discussion marker (SMA, S100 and calponin) in the cells around Salivary gland tumors account for <3% of all the the cribriform spaces [9].
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