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Reports of liver metastasis as the initial clinical manifestation of salivary gland adenoid cystic carcinoma. Four cases, including this one have been published
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Background:
Adenoid cystic carcinoma (ACC) is a common malignant tumor of salivary gland. The lung and liver are frequent sites of distant metastasis. Liver metastasis as the initial clinical manifestation of sublingual gland ACC is very rare.
Case summary:
A 51-year-old Chinese woman presented with a painless mass in the right lobe of liver. Th...
Context in source publication
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Citations
... Prior studies have reported liver metastasis of solid-type ACC, but no case described a clinical course similar to the one seen in this patient, with initial misdiagnosis as nasopharyngeal carcinoma. [6][7][8][9] The patient initially received six days of radiation therapy, which seemed to mitigate her pain with no changes in her vision loss. Despite this improvement, her course again became complicated with unusual findings on a CT of the chest/abdomen/ pelvis. ...
Adenoid cystic carcinoma (ACC) is a malignancy of the secretory epithelial cells of the salivary glands and constitutes less than 1% of all head and neck tumors. Metastasis occurs frequently and most commonly affects the lungs at a rate of 35 to 50%. In this case report, we present a rare case of nasopharyngeal ACC with distant metastasis to the liver. Our patient initially presented to the hospital with dental and sinus pain with initial imaging suggesting nasopharyngeal carcinoma invading the temporal lobe laterally and the cavernous sinus and clivus medially. The foramen ovale and the optic nerve were also involved, leading to loss of vision bilaterally. Immunohistochemical staining of the biopsy eventually led to the correct diagnosis of high-grade ACC, solid type. The patient’s hospital course was complicated, with pulmonary thrombosis eventually leading to hypoxic respiratory failure and death. Although this patient was initially diagnosed with nasopharyngeal carcinoma, thorough pathologic investigations allowed for a clearer understanding of the disease, primarily ACC’s eventual distal metastasis in the patient. In the future, providers should continue to keep ACC in their differential diagnosis list when evaluating patients with head and neck tumors, with the goal of maintaining locoregional control of the tumor.Keywords: adenoid cystic carcinoma, liver metastasis, nasopharyngeal salivary glands, head and neck cancer
... It is considered a slow-growing tumor with perineural spread (4) and a high incidence of local recurrence. Regional lymph node metastases are rare (5,6,7), while distant metastases occur in 19%-24% of AdCC cases with a mean presentation time of 7.6 years, although delayed recurrences can manifest even after 12-15 years (8), typically have an hematogenous spread involving the lungs, bones, brain, and liver (4,9,10) , . The disease has an unfavorable long term prognosis with survival rate at 5-10-20 years around 68%-52%-28% (11). ...
... Metastases are generally metachronous, can arise even decades after the primary tumor removal (16), Coupland et al. reported a liver metastasis occurred more the 30 year (17), otherwise if synchronous they can represent the initial presentation of the disease (10,18). ...
... Some case reports and small series suggest that surgery could improve survival (3,10,12,16); re-operation appears to be a feasible option in the case of a single hepatic recurrence, with an improvement in disease-free survival (19). Li XH et al. (10) recommended resections for isolated AdCC liver metastases due to the poor sensitivity to chemotherapy. ...
Adenoid cystic carcinoma (AdCC) is a rare tumor that typically develops in the salivary glands and less frequently in other sites of the head and neck region. Only a few cases of resected metachronous liver metastases have been reported. Minimally invasive surgery is currently the gold standard of care for liver resections; furthermore, the use of Indocyanine Green (ICG) is continuously increasing in surgical practice, especially in cases of primary liver tumors and colorectal liver metastases, due to its capacity to enhance liver nodules. We report the case of a 54-year-old male with a single liver metastasis of AdCC, located in SIII, who presented in our center 9 months after resection of a primary tumor of the laryngotracheal junction and adjuvant proton therapy. A 25-mg injection of ICG (0.3 mg/kg) was administered 48 h before surgery in order to highlight the tumor and perform an ICG-guided resection. The lesion was clearly visible during surgery, and, given its position and the proximity to the main lobar vessels of the left lobe, we opted for a left lateral sectionectomy. The outcome was unremarkable, with no major postoperative complications. The administration of ICG 48 h before surgery seems to be a valid tool even in cases of AdCC liver metastases, providing surgeons with better visualization of the lesion and improving the precision of the resection.
Key Clinical Message
The case report describes an adenoid cystic carcinoma occurring at a rare location and the diagnostic pathway. Swellings of the floor of the mouth, whether painful or without subjective symptoms and regardless of consistency, should be taken seriously.
Abstract
Adenoid cystic carcinoma (ADCC) of the sublingual salivary gland only accounts for 2% of all ADCCs. In this study, we report a rare case of ADCC with sublingual salivary gland origin in a 35‐year‐old man and a comprehensive review of articles published over the past 62 years.
