Asian Adenoid J Oral Cystic Maxillofac Carcinoma Surg. 2007;19:58-61.
Adenoid Cystic Carcinoma of Minor Anterior Lingual Salivary Gland: Case Report and Review of the Literature Masato Yamaguma,1 Hideo Kurokawa,2 Kuniyuki Matsubara,1 Shoji Kanda,3 Hiroyuki Takahashi,4 Min Zhang,5 Sumio Sakoda,2 Hiroshi Fukuyama5 1 Department of Dentistry and Oral Surgery, Japanese Seamen’s Relief Association Moji Hospital, Kitakyushu, 2Division of Medicine of Sensory and Motor Organs, Department of Oral and Maxillofacial Surgery, Miyazaki Medical College, University of Miyazaki, Miyazaki, 3 Dental Office of Kanda, Kitakyushu, 4Department of Radiology, Japanese Seamen’s Relief Association Moji Hospital, Kitakyushu, and 5Division of Oral Pathology, Department of Biosciences, Kyushu Dental College, Kitakyushu, Japan
Abstract Adenoid cystic carcinoma arising from the anterior third of the tongue, especially from an anterior lingual salivary gland, is extremely rare. An 80-year-old Japanese woman with an adenoid cystic carcinoma arising from an anterior lingual gland is reported. Key words: Adenoid cystic carcinoma, Anterior lingual gland, Minor salivary glands, Salivary gland neoplasms, Tongue neoplasms
Introduction An adenoid cystic carcinoma (ACC) is a slow-growing, but highly invasive, malignant tumour with a high recurrence rate, high rate of distant metastases, and high mortality.1,2 The salivary glands most commonly affected by this tumour are the parotid, submaxillary, and the accessory glands in the palate. Of the malignant tumours originating from minor salivary glands of the tongue, more than 85% involve the base of the tongue.3 However, ACC arising from an anterior lingual salivary gland is extremely rare.1-3 In this report, we describe a case of ACC that originated from an anterior lingual gland. In addition, the available English literature on ACC of the tongue is reviewed.
On extraoral evaluation, there was no enlargement of the submandibular or submental lymph nodes. Oral examination revealed a firm, submucosal, elastic, soft mass covered with normal mucosa that occupied the right anterior tongue to within 15 mm of the tip (Figure 1). Intraoral ultrasonography revealed a well-defined sonolucent mass, 15 mm in size (Figure 2a). On enhanced T2weighted magnetic resonance imaging, the mass appeared as a well-circumscribed, submucosal, nodular lesion that occupied the apical portion of the tongue (Figure 2b). The
Case Report An 80-year-old Japanese woman was referred to the outpatient clinic of the Department of Dentistry and Oral Surgery, Japanese Seamen’s Relief Association Moji Hospital, on 25 June 2004 with a chief complaint of a painless swelling in the right anterior part of the tongue. She had first noticed the swelling about 2 months earlier. Her past medical history was non-contributory. Physical examination showed a welldeveloped, well-nourished woman in no apparent distress. Correspondence: Hideo Kurokawa, Division of Medicine of Sensory and Motor Organs, Department of Oral and Maxillofacial Surgery, Miyazaki Medical College, University of Miyazaki, Kiyotake-Cho, Kihara 5200, Miyazaki 889 1692, Japan. Tel: (81 98) 585 3786; Fax: (81 98) 585 7190; E-mail: [email protected]
Figure 1. Clinical view of adenoid cystic carcinoma in the anterior tongue. © 2007 AsianAsian Association J Oral Maxillofac of Oral andSurg. Maxillofacial Vol 19, No Surgeons. 1, 2007
Yamaguma, Kurokawa, Matsubara, et al
Figure 2. (a) Intraoral ultrasonography showing a well-defined sonolucent mass. (b) Magnetic resonance imaging scan showing a highintensity signal in the right anterior tongue.
biopsy specimen showed a typical ACC of the cribriform type. Under general anaesthesia, a partial glossectomy was performed, and the hypoglossus, genioglossus, and geniohyoid muscles were removed together with the sublingual gland. The cut surface of the tumour showed a firm, yellowish-white mass, 15 × 15 mm in size, involving the apical portion. Microscopic examination of the lingual mass revealed the non-encapsulated infiltrating tumour to be composed of solid nests of columnar epithelial cells showing glandular formations surrounded by hyaline demoplastic stroma. Some of these glandular formations were filled with an eosinophilic or hyalinised substance. Tumour tissue was distinct from the sublingual gland and was associated with anterior lingual glands. The tumour cells had large round or ovoid nuclei with occasional hyperchromatic nucleoli. Mitoses were seen infrequently. No intravascular invasion was noted. A small tumour island was seen in the proximity of a peripheral nerve. Immunohistochemical staining was performed on the formalin-fixed paraffin-embedded sections of the surgical specimen by the streptavidin-biotin-peroxidase complex labelling method. The reaction for S-100 protein (antibodies for S-100 protein, 1:100; Dako, Glostrup, Denmark) was positive in the nuclei and cytoplasm of the tumour. The tumour was diagnosed as ACC, predominantly of the cribriform type, originating from an anterior lingual gland (Figure 3). No evidence of recurrence or metastasis was noted in the 18 months follow-up so far.
Discussion An ACC consists of small, deeply staining uniform cells resembling basal cells that are commonly arranged in Asian J Oral Maxillofac Surg. Vol 19, No 1, 2007
anastomosing cords or a duct-like pattern, the central portion of which may contain mucoid material.1,4 The tumour grows slowly, with uncommon mitotic figures, but it has a propensity to invade perineural space. 5 This tumour is classified into 3 growth patterns according to the predominant histological features1,6: cribriform (glandular), tubular, and solid types. Furthermore, the tumour cells are of 2 types, duct-lining and myoepithelial type cells. In the present case, the tumour was predominantly of the cribriform type, with rare mitotic figures. Moreover, there was no intravascular or perineural invasion. In immunohistochemical studies, positive staining for S-100 protein has been shown in the normal myoepithelial cells and was focally present in the ACC.3 In the present case, the existence of S-100 protein in the tumour cells indicates that some tumour cells differentiated toward myoepithelial cells. The minor salivary glands in the tongue are divided into 3 types according to their distribution: serous type posterior lingual glands, mixed type lateral lingual glands, and mucous type anterior lingual glands (Blandin-Nuhn glands).7 Salivary gland tumours of the tongue frequently arise in the posterior tongue but are uncommon in the anterior tongue.1-3,8 In our case, the tumour was located in the apical portion of the tongue and was histologically distant from the lateral lingual glands. Moreover, normal anterior lingual glands were observed around the tumour. From the location and histopathologic findings, the tumour was considered an ACC originating from an anterior lingual gland. A review of the literature revealed 6 cases of anterior lingual ACC, but produced only 3 well-documented cases of primary ACC arising in the anterior tongue. The clinical features of these cases along with those of the present case are summarised in Table 1.3,5,9-11 Patient age ranged from 61 to 80 years, and averaged 70.8 years. The lesions showed no 59
Adenoid Cystic Carcinoma
Figure 3. (a) Histopathology of the tumour lesion showing border area between minor salivary gland and tumour tissue (haematoxylin and eosin; original magnification, × 2.1). (b and c) The tumour nests have a cribriform pattern (haematoxylin and eosin; original magnification, × 10 [b], × 40 [c]). (d) Immunoreaction for S-100 protein is positive in the nuclei and cytoplasm of the tumour (original magnification, × 25). Table 1. A review of the literature of adenoid cystic carcinoma in the anterior tongue. Reference
Survival after diagnosis
Luna et al5
Potdar and Paymaster9
Potdar and Paymaster9
Ishikawa et al3
20 × 20 × 15
NED (30 months)
Moles et al10
30 × 30
Partial glossectomy + MRND
NED9 (64 months)
Takubo et al11
20 × 20
NED (36 months)
15 × 15
NED (18 months)
Abbreviations: NS = not specified; MRND = modified radical neck dissection; NED = no evidence of disease.
gender predilection, and ranged from 15 to 30 mm, with an average size of 21.3 mm. The initial clinical finding was a painful or painless enlarging mass. The duration of symptoms before treatment ranged from 1 month to 2 years. Regional 60
lymphadenopathy was absent in all cases. All patients underwent surgical excision of the tongue lesion; 1 patient underwent modified radical neck dissection.10 Histologically, 2 tumours were classified as the cribriform type and 2 as Asian J Oral Maxillofac Surg. Vol 19, No 1, 2007
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the tubular type. The follow-up period in this series ranged from 16 to 64 months, with an average of 35.5 months. One patient was found to have lung metastasis at a 15-month follow-up.3 However, all patients were alive and free from disease. The literature suggests that ACCs are histologically aggressive and give rise to metastases many years after excision of the primary tumour. To date, the local tumour in this case is controlled, and there is no evidence of metastasis. However, long-term follow-up will be continued.
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