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Adenosquamous Carcinoma in Buccal Mucosa-A Case Report

Gopal Chandra Halder, Jay Gopal Ray*, Santanu Patsa

Oral and Maxillofacial Pathology, Dr. R. Ahmed Dental College and Hospital, West Bengal University of Health Sciences, Kolkata

Abstract: Adenosquamous carcinoma is a rare malignant tumor with poor prognosis that is characterized histopathologically by the simultaneous presence of distinct areas of adenocarcinoma and squamous cell carcinoma. Until 2004, only 59 cases of adenosquamous carcinoma have been documented in English literatures in the head and neck region since the first one being reported in 1968. The present report is a case of adenosquamous carcinoma in a 75 years old man with a fast enlarging ulcerated lesion on right buccal mucosa. The patient developed trismus due to local infiltration of tumor mass within the muscle with fixation of the skin and a small perforation on the right side of the face. Adenosqamous carcinoma in the oral cavity is not seen frequently. All the reported cases are in the tongue, floor of the mouth and tonsillar palatine region in descending order. This presentation in the buccal mucosa with infiltration up to the skin is in itself significant and rare.

Keywords: Buccal mucosa, adenosquamous carcinoma, ulcerated lesion, infiltration

Received November 01, 2014; Revised November 25, 2014; Accepted November 30, 2014

Citation: Gopal Chandra Halder, Jay Gopal Ray, Santanu Patsa, Adenosquamous Carcinoma in Buccal Mucosa-A Case Report. International Journal of Dental Sciences and Research, 2 (6). 158-160. January 2014. doi:10.12691/ijdsr-2-6-9

1. Introduction

Adenosquamous carcinoma is a controversial neoplasm which has been defined by the World Health Organization as a malignant tumor with histopathological features of both adenocarcinoma and squamous cell carcinoma [1]. Adenosquamous carcinoma has been documented in numerous epithelial tissues all over the body including skin, colon, pancreas, thyroid gland, larynx etc. but it is rare in oral cavity [2].

Histogenesis of the tumor is not completely understood, but possibility of its origin by carcinomatous change of basal layer of surface epithelium and excretory ductal epithelium of minor salivary glands have been reported [3], [2]. The present report is of a highly aggressive adenosquamous carcinoma on the right buccal mucosa.

2. Case Report

A 75 years old man visited the out patient department of R. Ahmed Dental College & Hospital in May 2014 with complaints of pain, trismus and non-healing ulcer since last 15 days (Figure 2). The clinical examination revealed a small perforation on the right cheek with fixation of skin and the under lying structures adjoining the before said perforation (Figure 1). No cervical lymph node was palpable.

Intra oral examination revealed 2 cm X 1 cm ulcer with irregular border, rolled thickened margin and indurated base.

Orthopantomogram of jaws showed no bony involvement either in mandible or maxilla (Figure 3). Patient had the habits of smoking and betel quid chewing for long time. Medical history revealed no abnormality and the reports of routine blood examination were within normal limits. An incisional biopsy was done after taking informed consent from the patient.

Figure 1. A small perforation at right cheek

Histological examination with hematoxylin and eosin staining revealed variable proportions of the squamous and glandular components under an interrupted stratified squamous epithelium (Figure 4). Deeper in connective tissue dysplastic epithelial cells arranged in islands or sheets were present 9 Figure 5). Duct-like structures lined by a single or double layer of atypical columnar epithelial cells showing pleomorphism and hyperchromatism surrounding mucinous substance could be detected (Figure 6). Overall features were corroborative to adenosquamous carcinoma.

Figure 2. Ulcer with irregular border

Figure 3. Orthpantamogram showed no boney involvement

Figure 4. Neoplastic squamous island (right arrow) and neoplastic ductul component (left arrow)

Figure 5. dysplastic squamous cells in a sheet

Figure 6. Ductual structure with mucin pool

3. Discussion

A malignant salivary tumor described by Gerughty and his co-workers in 1968 had features of both squamous and glandular components [4]. It is a rare type of controversial malignant tumor because of its unknown histogenetic origin. Several theories have been postulated by various authors regarding the origin of the tumor. Some authors believe that it is a variant of salivary gland tumor. Some researchers opined that the origin of the tumor was from basal cells layer of squamous epithelium. An experimental model on adenosquamous carcinoma suggested that this tumor dose not originate from salivary or seromucous glands [5]. Hence this model provides a support in favor of basal cell origin. However, in 1991 WHO classification of salivary gland tumors did not include adenosquamous carcinoma as a salivary gland tumor [6]. In 1997, WHO defined adenosquamous carcinoma as a malignant tumor with features of both adenocarcinoma and squamous cells carcinoma in the same tumor mass in close proximity but generally distinct [7].

Larynx is most common place of origin of the tumor (44.8%) in the head-neck region. In oral cavity dominant sites are floor of the mouth and tongue. First reported case origin from buccal mucosa was published in 2009 [8]. The literatures revealed that adenosquamous carcinoma is characterized by local recurrence, cervical lymph node metastasis, distant metastasis and poor prognosis. Males are most commonly affected than females (3.5:1, male to female ratio) and the age range is 22 to 80 years. Patient reported with variable signs and symptoms but most common features is pain in maximum cases which may be due to the tendency of spreading by perineural invasion [9].

Microscopic features of the present case were confirmatory diagnosed as adenosquamous carcinoma and so we did not go for immunohistochemical analysis. However most literatures showed the glandular differentiated area was positive for pancytokeratin, epithelial membrane antigen, CK7/8 [10]. Squamous cell component was positive for high molecular weight cytokeratin (MNF116) & CEA and negative for CK7/8 & CK20.

In case of differential diagnosis for adenosquamous carcinoma, mucoepidermoid carcinoma and adenoid squamous cell carcinoma should be considered very carefully. Mucoepidermoid carcinoma and adenosquamous carcinoma, both manifest epidermoid and glandular features. However, separated and definitive areas of adenosquamous carcinoma and squamous cell carcinoma are not seen in mucoepidermoid carcinoma. High grade mucoepidermoid carcinoma consists of intermediate or epidermoid cells without keratin formation. Adenosquamous carcinoma shows the features of squamous cell carcinoma viz infiltrative pattern, nuclear pleomorphism, mitotic figures and keratin pearls formation without intermediate cells.

Adenosquamous carcinoma is highly aggressive, locally recurrent and highly metastatic lesion. In our case, there was no regional lymph node metastasis. Also, a similar type of case has been reported in 2009 without lymph node involvement and prognosis of the patient was greater than 5 years [8].

4. Conclusion

Adenosquamous carcinoma is a controversial neoplasm and the histogenesis of the tumor is not completely understood, but possibility of its origin by carcinomatous change of basal layer of surface epithelium remains [8]. The first reported case in the buccal mucosa is in 2009 [8]. We report this case because of its rarity.

Acknowledgement

This article could not have been possible without the help of the staffs of the Department of Oral and Maxillofacial Pathology, R. Ahmed Dental College & Hospital. I offer our sincere thanks to Assoc. Prof. (Dr.) Tushar Deb, Asst. Prof. (Dr.) Sila Datta, and Clinical Tutor (Dr.) Sandip Ghose of the same institution.

References

[1]   | Thackray AC, Sabin LH: “Histologic typing of salivary gland tumor”, Geneva, World Health Organization, 16, 1972.
[2]   | Gary L. Ellis, Aault L. Auclair, Douglas R. Gnepp, “surgical pathology of salivary gland”, volume-25, W.B Saunders company, page-455.
[3]   | Keelawat S, Liu CZ, Roehm PC, Barnes L. “Adenosquamous carcinoma of the upper aerodigestive tract: a clinicopathologic study of 12 cases and review of the literature”, Am J Otolaryngol, 23 (3), 160-8, May-Jun 2002.
[4]   | Gerughty RM, Hennigar GR, Brown FM, “Adenosquamous carcinoma of the nasal, oral, and laryngeal cavities: a clinicopathological survery of ten cases”, Cancer, 22, 1140-55, 1968.
[5]   | Cadesa A, Bombi JA, Pera M, Fernander PL, Campo E, Pera C, Mohr U, “Spectrum of glandular differentiation in experimental carcinoma of the esophagus induced by 2,6-dimethylintrosomorpholine under the influence of esophagojejuostomy” Exp Toxic Pathol, 46, 41-4, 1994.
[6]   | Seifert G, Sabin LH, “WHO histological typing of salivery gland tumors”, Springer-Vertag, Berlin, 1991, 2nd Ed.
[7]   | Pindborg JJ, Reichart PA, Smith CJ, Van der Wall I, “Histological typing of cancer and precancer of the oral mucosa. World Health Organization international histological classification of tumors”, Springer New York Heidelberg Berlin, 1997, 16, 2nd Ed.
[8]   | Kam-wing Leung, Kuo-Chung Yang, Chia-Jung Chen, “Adenosquamous carcinoma of buccal mucosa- A case report” Taiwan J Oral Maxillofac Surg, 20, 19-28, March 2009.
[9]   | Yoshimura Y, Mishima K, Obara S, Yoshimura H, Marayama R, “Clinical characteristic of oral adenosquamous carcinoma: report of a case and an analysis of the reported Japanese cases”, Oral Oncol, 39, 309-15, 2003.
[10]   | Sheahan P, Fitzgibbon J, Lee G, O’Leary G, “Adenosquamous carcinoma of the tongue in a 22 years old female: report of a case with immunohistochemistry”, Eur Arch Otorhinolaryngol, 206, 509-12, 2003.