Yonago Acta medica 2009;52:73–76

Large-Cell Undifferentiated Carcinoma ofthe Submandibular GlandKazunori Fujiwara, Katsuyuki Kawamoto and Hiroya Kitano
Division of Otolaryngology, Head and Neck Surgery, Department of Medicine of Sensory andMotor Organs, School of Medicine, Tottori University Faculty of Medicine, Yonago 683-8504
Japan

Occurrence of large-cell undifferentiated carcinoma in the submandubular gland is uncommon. A 52-year-old man with a 30-mm tumor underwent radical resection of the left
submandibular gland together with selective dissection of enlarged ipsilateral cervicalnodes followed by postoperative radiotherapy. After histologic examination identified thesubmandibular gland mass as large-cell undifferentiated carcinoma, selective dissectionof enlarged contralateral cervical nodes was performed. The patient showed no evidence
or of distant metastasis at 24 months after initial treatment.
Key words: large-cell type; submandibular gland; undifferentiated carcinoma

Large-cell undifferentiated carcinoma of major
salivary glands is extremely rare accounting forless than 3% of epithelial cancers involving theseorgans. Most large-cell undifferentiated carcinomas of the salivary gland occur in the parotid. A
review of the literature disclosed only a few cases
where large-cell undifferentiated carcinoma originated from the submandibular gland. We reportsuch a tumor diagnosed by histology and immunohistochemistry.

(MRI) demonstrated a tumor with indistinct margins and contrasted enhancement slightly greaterthan in the submandibular gland, measuring 30mm in greatest dimension and located in the lower
part of the gland. Multiple cervical lymph nodes
on the left side showed contrast enhancement (Fig.
1). A malignant neoplasm was strongly suspected. Fine-needle aspiration cytologic examination
resulted in a provisional diagnosis of adenocarcinoma versus pleomorphic adenocarcinoma.
The patient underwent radical left submandibular gland resection including wide local excision and selective neck dissection in July 2006.
Based on these findings, the tumor was diagnosed
as large-cell undifferentiated carcinoma. CT andMRI demonstrated no distant metastasis in thechest or abdomen.
Postoperative radiotherapy was performed
with a total dose of 60 Gy over a wide left submandibular and cervical field. After radiotherapy,
contralateral cervical lymph nodes began to enlarge gradually. Cytologic malignant metastasis
to these nodes was suspected. Accordingly, thePatient Report
A 52-year-old Japanese man noticed a painless left
submandibular mass in May 2006. He consulted
a clinic where he was diagnosed with a submandibular gland neoplasm, and was referred to ourhospital in June 2006. A firm fixed mass with notenderness was noted in the left submandibularregion. Cervical lymphadenopathy was evident in
the upper left portion of the neck. Computed tomography (CT) and magnetic resonance imaging

Abbreviations: CT, computed tomography; MRI, magnetic resonance imaging

73K. Fujiwara et al.

patient underwent selective node dissection on the right side of the neckin November 2006. Malignant cells
were not found in resected lymph
nodes. The patient has shown no signsof recurrence or metastasis over the 24months following this node dissection.
Pathological Findings
Most of the left submandibular gland
had been replaced by a firm tumor
measuring 30 mm. The resected tumor was yellow-gray and rubbery (Fig.
2). No necrotic or cystic areas werefound. The tumor showed no micro- Fig. 1. By CT (a) and MRI (b), a tumor with indistinct margins shows
scopic invasion of surrounding tissue. contrast enhancement slightly exceeding that in submandibular gland.
The tumor measures 30 mm in maximal dimension and is located in
Keratinization was not apparent in rou- the inferior portion of the gland.
tinely stained sections (Fig. 3). Tumor
cells were arranged in palisades or sheets, and hadlarge, atypical nuclei (Fig. 4). ImmunohistochemiDiscussion
cally, tumor cells showed positive reactions for
keratin AE1to3, keratin 7 and epithelial membrane Undifferentiated carcinoma of the major salivary
antigen, while showing negative reactions for keraglands is uncommon, and reported to account for
tin 20, carbohydrate antigen 19-9, S-100, carcino1% to 4.1% of epithelial salivary gland neoplasms
embryonic antigen and vimentin. Based on these(Nagao et al., 1982; Wang et al., 2004). Most
findings, the tumor was diagnosed as large-cell
undifferentiated salivary gland carcinomas haveundifferentiated carcinoma. Metastasis was foundoccurred in the parotid. Even so, undifferentiin 2 lymph nodes from the upper neck.
ated carcinoma has been reported to account for
only 1% to 5.5% of all malignant parotid glandneoplasms (Eneroth, 1964; Patey et al., 1965;
Seifert and Donath, 1976; Wang et al., 2004).
Little is known about undifferentiated carcinomaof the submandibular gland, and only a few
cases have been reported (Wahlberg et al., 2002;
Uemaetomari and Ito, 2005; Lin et al., 2006).
Undifferentiated carcinoma of salivary
glands is classified as either small- or large-cell
type. Small-cell undifferentiated carcinoma apparently is 2 to 3 times more frequent than largecell undifferentiated carcinoma (Nagao et al.,
1982; Hui et al., 1990). The present patient wasdiagnosed with large-cell undifferentiated carcinoma of the submandibular gland.
Fig. 2. The resected specimen included a yellow-gray,firm and rubbery tumor.
The differential diagnosis of large-cell undif-ab74