Case 1

A 70-year-old white male was referred to the otolaryngology department complaining of a 4 month history of a rapidly enlarging painful mass in the right submandibular region. He was positive for HIV and HCV, and his past medical history included a basal cell carcinoma on the dorsum of his nose that was diagnosed two years previously (T3N0M0) and successfully treated with surgery and radiotherapy. A physical examination of the submandibular region revealed a firm, fixed mass approximately 5 cm in diameter. The thyroid was normal and no neck masses were identified by palpation and image exams. A computed tomography (CT) scan revealed a heterogeneous mass measuring 5 cm × 7 cm, with lobular contours and nonspecific borders, in the submandibular region ( Fig. 1 A ). The chest/abdomen radiograph and magnetic resonance imaging (MRI) examinations were normal, and no supposedly primary tumour could be detected. A fine-needle aspiration biopsy (FNAB) revealed malignant cells of unknown origin ( Fig. 1 B and C). The patient underwent an incisional biopsy that confirmed the diagnosis of T3N0M0 SGSmCC (Stage III). Because of the patient’s age and his clinical status, tumour remission was achieved using a combination of radiotherapy and chemotherapy. He received 5940 cGy over 33 fractions at a dose of 180 cGy per fraction to his right submandibular region. The chemotherapy consisted of three monthly cycles of etoposide plus cisplatin. During therapy, the patient was twice admitted with febrile neutropenia, and a progressive weight loss was noted. Five months after treatment, the patient was admitted to hospital with a diagnosis of icterus and cholestasis. An ultrasonogram of the abdomen revealed heterogeneous masses in the pancreas (8 cm ³ ), liver (2 cm ³ ) and one para-aortic lymph node that were consistent with metastatic disease. The patient was not considered for therapy because of the systemic metastases. One month later (12 months after diagnosis), the patient died of complications related to metastatic disease.

(A) CT scans in an axial section with endovenous contrast. Heterogeneously mass in the region of the submandibular gland. (B) Papanicolau-stained aspirate smear 400×. Poor cohesive cell groups composed of cells with very scanty cytoplasm (case 1). (C) Papanicoloau-stained aspirate smear 1250×. Small to intermediate sized anaplastic cells with evident ovoid nucleous and a ‘salt and pepper’ chromatin pattern (case 1).

Case 2

A 69-year-old white male was referred to the otolaryngology department complaining of a 12-month history of a painless mass in the right upper neck near the mandible, nodules in the neck region and a progressive unintentional weight loss. His past medical history was not significant. A physical examination of the neck revealed an approximately 4 cm firm mass on the right parotid and a 2 cm diameter enlarged ipsilateral cervical lymph node. There were no masses on the left side of the neck, and the thyroid was normal. An FNAB of the right neck nodule was performed, resulting in a diagnosis of T2N1M0 metastatic disease (Stage III). A complete work up that included X-ray examinations of the chest and abdomen proved negative in detecting any other lesion. A total parotidectomy with a radical right neck dissection was performed. One day after surgery, the patient developed respiratory distress, and an MRI of the chest showed an idiopathic pleural effusion (hemothorax). 2 days after surgery, the patient died from respiratory failure. The family refused a necropsy examination.

Pathological findings

The two cases show similar cytological and histological patterns, and for this reason they are described together here. The Papanicolau-stained FNAB samples from both cases showed various clusters of small cells with scant cytoplasm and moderately pleomorphic round-to-ovoid nuclei with smooth or irregular contours containing dispersed thin, granular chromatin. No evidence of ductal formation or keratinization was found. The cells were dispersed among necrotic debris, erythrocytes and mononuclear inflammatory cells ( Fig. 1 B and C).

The hematoxylin–eosin stained histological slides from both cases showed malignant lesions formed by sheets, cords and irregular nests of small anaplastic cells containing nuclei that were two or three times larger than mature lymphocytes, were dispersed among dense fibrous stroma, and infiltrated the salivary gland tissue ( Fig. 2 A ). The tumour cells had scant cytoplasm and round-to-oval nuclei with absent or unremarkable nucleoli. Rosette-like and ductal structures, as well as squamous differentiation, were absent, while crush artifacts, mitotic figures and necrotic areas were found in the tumour ( Fig. 2 B). In both cases, focal areas of hemorrhage and discreet lymphocyte infiltration were noticed throughout the lesions.

(A) Hematoxylin–eosin (H–E) stain 200×. Irregular nests of small anaplastic cells infiltrating parotid gland tissue (case 2). (B) H–E stain 200×. A focal area of necrosis (case 2). (C) H–E stain 200×. Presence of crushing artifact and a perineural invasion (case 2). (D) H–E stain 1250×. Small to intermediate sized cells with scant cytoplasm, finely granular nuclear chromatin, and inconspicuous nucleoli (case 2).

Additionally in case 2, perineural invasion was clearly observed ( Fig. 2 C), and the surgical margins were compromised by the tumour. Small cells with more polygonal cytoplasm could also be seen ( Fig. 2 D). Neck dissection showed tumour in the submandibular gland and metastatic lesions in 19 lymph nodes.

Immunohistochemical evaluation of the tumours demonstrated positive immunoreactivity for epithelial and neuroendocrine markers. No positivity for lymphoid, neuronal or muscle markers was observed ( Table 2 ). In this way, the tumours were diagnosed as primary salivary gland small cell carcinomas.

Table 2

Lists of antibodies, clone, source, dilution and reactive for each case.

Antibodies to	Clone	Source	Dilution	Case I	Case II
Cytokeratin	AE1/AE3	Abcam	1:100	+	+
Human epithelial antigen	BER-EP4	Abcam	1:100	+	NS
Epithelial membrane antigen	E29	Milipore	1:400	+	+
Cytokeratin 20	Ks20.8	Milipore	1:200	−	−
Thyroid transcription factor-1	8G7G3/1	Abcam	1:300	+	+
Carcinoembryonic antigen	Policlonal	Dako	1:100	−	NS
Vimentin	V9	Dako	1:100	−	−
S100 protein	Policlonal	Dako	1:200	−	−
LCA (CD45)	PD7-26 e 2B11	Dako	1:100	−	−
MIC-2 (CD99)	12E7	Abcam	1:200	+	+
Neuron-specific enolase	BBS-NC-VI-H14	Abcam	1:100	+	+
Chromogranin A	DAK-A3	Dako	1:200	+ (focal)	+
Synaptophysin	SY38	Milipore	1:100	−	−
Neurofilament	2F11	Abcam	1:100	−	−
NCAM (CD56)	56C04	Lsbio	1:200	−	+
Leu-7 (CD57)	NK1	Milipore	1:500	+	+
Muscle-specific actin	1A4	Merck	1:400	−	−
Melanoma	HMB-45	Dako	1:200	−	−

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