Hepatic metastasis as an initial manifestation of salivary adenoid cystic carcinoma: Cytologic diagnosis

Abstract

Adenoid cystic carcinoma is a malignant tumor of the salivary glands. It is slow growing and is characterized by the delayed development of distant metastasis, which may develop even a decade or more after initial treatment of the primary tumor. We present the case of a 68-year-old male with complaints and radiological findings suggestive of primary hepatocellular carcinoma. However, fine-needle aspiration findings showed metastasis of adenoid cystic carcinoma. A retrospective clinical and ultrasound examination of the patient revealed a small asymptomatic enlargement of the submandibular salivary gland that was aspirated and showed features of adenoid cystic carcinoma. Diagn. Cytopathol. 2009;37:45-47. ' 2008 Wiley-Liss, Inc.Key Words: hepatic metastasis; initial; adenoid cystic carcinoma; primary; retrospective Adenoid cystic carcinoma (ACC) is a rare neoplasm and accounts for less than 1% of all head and neck malignancies and~10% of salivary neoplasms.1 It is however the most common malignant tumor of the submandibular, sublingual, and minor salivary glands. It is slow growing and shows multiple local recurrences. Distant metastasis is more common than regional lymph node metastasis. 2 Case ReportsA 68-year-old male patient presented with complaints of weight loss, an upper abdominal lump, and itching all over the body of 3 months duration. On examination, he had hepatomegaly, which was hard, nodular, and nontender, extending up to the right iliac fossa. An ultrasound (USG) and CT revealed multiple hypoechoic lesions surrounded by a hyperechoic rim involving both the lobes (Fig. 1), suggestive of hepatocellular carcinoma. Chest radiograph did not reveal any significant findings. USG-guided fine-needle aspiration (FNA) was carried out using a 22-gauge needle from multiple sites. Alcohol-fixed smears were stained by Papanicolaou (Pap) stain and hematoxylin and eosin stain. Air-dried smears were stained by May Grü nwald Giemsa (MGG) stain.FNA smears revealed basaloid tumor cells arranged in multilayered sheets and dispersed as single cells with high nucleo-cytoplasmic ratio. Nuclei were uniform in size, hyperchromatic, with coarsely granular chromatin and small nucleoli. Hyaline spherical globules of varying size with adherent tumor cells were seen, staining pale gray-blue with Pap ( Fig. 2) and bright magenta with MGG stain (Fig. 3). Reactive and normal hepatocytes were also present (Fig. 4). A diagnosis of ACC was made. The patient was then examined for the possible site of origin, and was found to have an asymptomatic left submandibular swelling of size 1 3 1 cm. USG-guided FNA was then carried out from this submandibular mass that showed cytological features of ACC. DiscussionACC is a malignant tumor that is characterized by slow growth, multiple local recurrences, and a prolonged clinical course often with delayed development of distant metastasis. The organs involved in decreasing order of frequency are lung, bone, brain, and liver.3 Other rare metastatic sites include stomach, choroids, kidney, and skin....

Adenoid cystic carcinoma of hard palate with coincidental metastases to lung and liver

“…In another study Spiers et al 12 presented two cases of liver metastasis in both of which liver metastasis occurred subsequent to pulmonary involvement. Deshpande et al 13 introduced a case presented with hepatomegaly and fine-needle aspiration revealed ACC and further investigations showed the tumour originating from submandibular gland however they did not explain the treatment and the patient's survival. Harish et al reported a case of liver metastasis 3 years after primary parotid surgery without lung and bone involvement.…”

Recurrent adenoid cystic carcinoma in the liver: a repeated laparoscopic surgical approach

“…We were able to find in literature five clinical report of liver metastases from cancer of the salivary glands (four ACC and one Mucoepidermoid Carcinoma) and three cytopathology diagnosis case-report of liver DM from ACC [16,[27][28][29][30][31][32][33] (See Table 1). Zeidan [34] instead, reports an extended right hemihepatectomy for isolated DM from ACC of the lacrimal gland 20 years after resection of the primary tumor.…”

Adenoid Cystic Carcinoma of the Submandibular Gland, Locoregional Recurrence, and a Solitary Liver Metastasis More Than 30 Years Since Primary Diagnosis

“…Though rare, there is an increasing frequency of reported cases of ACCs either first presenting with hepatic metastatic disease [7–9] or of distant metastases that have arisen at variable times since primary diagnosis [10–12]. There is no consensus regarding optimal treatment.…”