Primary Intestinal-Type Adenocarcinoma of Tongue: A Case Report with Immunohistochemical and Molecular Profiles and Review of the Literature

Abstract

Primary lingual intestinal-type adenocarcinomas are extremely rare with only a few cases described. A case with immunohistochemical expression of Androgen Receptor (AR) which was treated solely by chemo-radiotherapy is reported herein. A 54-year-old male was referred with symptoms of fullness in his tongue. Clinical examination showed an asymmetry of the tongue with a hard mass palpable within the middle of the tongue. Biopsy showed intestinal-type adenocarcinoma. The tumour showed positive staining with cytokeratin 7, cytokeratin 20, CDX2, AR, b-catenin and was mismatch repair proteins (MMR) proficient. The molecular analysis did not show mutations in the KRAS, NRAS, BRAF and PIK3CA genes. The patient was treated with radiochemotherapy and is in remission 3.5 years after the diagnosis. This is the first case of intestinal-type tongue adenocarcinoma which showed AR expression and was treated solely with radical chemoradiotherapy.

Primary intestinal‐type adenocarcinoma of the oral tongue: Case report and review of histologic origin and oncologic management

“…Expression of both CK20 and CDX2 have been consistently positive within the 3 previously reported cases of ITAC of the oral cavity. Expression of CK7 has been varied; Rahimi et al and McDaniel et al report positive staining of CK7, whereas our case and Smith et al had negative staining of CK7.…”

“…Within the head and neck region, ITAC most commonly occurs in the sinonasal tract . Six reports have described primary lingual ITAC, 3 in the base of the tongue and 3 in the oral tongue . We report an additional case of oral tongue ITAC and, we believe for the first time in the literature, provide histologic evidence of metaplasia within the salivary gland epithelium in the oral cavity, thus differentiating the tumor from a distant metastasis.…”

“…Given the histologic findings in our case of multiple positive nodes, perineural invasion, lymphovascular invasion, and extranodal extension of the tumor, adjuvant chemotherapy and radiotherapy was recommended and given . McDaniel et al and Rahimi et al utilized IMRT at 62 and 65 Gy in 31 and 30 fractions, respectively. Smith et al treated with external beam radiation but did not report the exact regimen.…”

“…There are only 6 cases in which primary ITAC has been described outside the sinonasal tract within the head and neck, 3 of which originated in the oral tongue (Table ) . Of these 3 cases, 1 presented with cervical metastases, and 2 went on to develop metastases to the lungs …”

“…Treatment of ITAC consists of surgical resection with adjuvant radiation for advanced tumors, and may or may not include chemotherapy depending upon postoperative characteristics of the tumor . The cases reported by McDaniel et al and Smith et al were both managed with surgical resection in the form of partial glossectomy with adjuvant chemoradiation and radiation respectively, whereas Rahimi et al chose to treat solely with chemotherapy and radiation.…”