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A4133 - Diffuse Pleural Metastases as a Manifestation of Recurrence in Adenoid Cystic Carcinoma
Author Block: B. Birnbaum, M. Oron, B. A. Mina; Lenox Hill Hospital, New York, NY, United States.
INTRODUCTION:
Adenoid cystic carcinoma (ACC) is a rare salivary gland malignancy with a propensity for recurrence as distant metastases in lung parenchyma or liver. Here we present an unusual case of recurrent ACC manifesting as diffuse pleural metastases, but without any involvement of lung parenchyma or liver.
CASE REPORT:
A 49 year old woman never smoker with a history of ACC of the right tongue base status-post resection and stereotactic body radiation therapy (SBRT) 20 years ago, presented with vague severe right upper quadrant abdominal pain. Imaging with ultrasound and positron emission tomography-computed tomography (PET-CT) revealed submental lymphadenopathy, extensive left-sided pleural nodularity including a 1.9 x 1.7cm nodular opacity at the left lung base, and multiple hepatic lesions concerning for metastases. Abdominal ultrasound revealed multiple solid hypoechoic hepatic masses consistent with metastasis, and two large complex cystic hepatic masses indeterminate for metastases versus hematomas or abscesses. Laparoscopic resection of segment III of the liver was subsequently performed, with pathology results notable only for hepatocellular adenoma; no cystic lesions were seen. CT-guided left pleural core biopsy and fine-needle aspiration of her pleural nodules revealed metastatic ACC. She additionally underwent laparotomy and right hepatectomy with pathology also negative for ACC. Her case was presented at multidisciplinary tumor board and she was recommended to undergo repeat imaging with contrast-enhanced CT chest as well as PET-CT to better evaluate biopsy approach and to obtain more tissue for cytometric testing, which would aid in tailoring adjuvant therapy.
DISCUSSION:
Though rare overall, ACC is a well-recognized salivary gland malignancy probably owing to its unusual pattern of slow-growth, aggression, resistance to treatment, and distant metastases manifesting as recurrence many years after initial diagnosis and treatment. What is unusual about our case is the extensive metastatic disease found in the left lung pleura and the notable absence of parenchymal lung or liver metastases, findings that have not been described in previous reports. While these findings certainly pose new prognostic and treatment considerations for our patient, it ultimately reminds us of the importance of long-term follow-up and surveillance in ACC.