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We report a rare case involving a patient with C3 vertebral body metastasis secondary to adenoid cystic carcinoma of the tongue.
Five years after local resection of the primary tumor, magnetic resonance imaging showed a metastasis located in the left posterior border of the C3 vertebral body. Additionally, multiple pulmonary metastases were identif...
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Citations
... For this reason, there are few data in the literature as only isolated cases or very small series of four to seven cases being published [15,[17][18][19][20][21]. ...
... Spinal metastases originating in the oral cavity cancer can be located in the cervical, thoracic and lumbar regions [15,17,18], although some authors emphasize that they metastasize mainly in the lumbar region and are most frequently represented by moderately differentiated or poorly differentiated squamous cell carcinoma and much less often by an adenoid cystic carcinoma of the tongue [17][18][19][20][21], aspects that are similar to those identified in our series. ...
Spinal metastases from gastrointestinal (GI) cancer are rare and as a result there are only case reports or small series in the literature. The aim of our work was to identify the demographic aspects, the location, and the histopathological aspects of spinal metastases from GI cancers diagnosed and treated in a reference Hospital in Romania over a period of nine years, and comparing the data obtained with those from the recent literature. This is a retrospective case series study on spinal metastases from GI cancers, developed in patients older than 18 years that were surgically treated between January 2013 and December 2021 within three Neurosurgery Clinics from Prof. Dr. Nicolae Oblu Emergency Clinical Hospital, Iaşi, Romania, which is a tertiary Hospital in Romania regarding the surgical treatment of spinal metastases. We included in our study the patient's demographic data (age and gender), clinical data (location of spinal metastases), radiological investigations and pathological features of the lesions. Regarding the immunohistochemical stainings, the following antibodies were used: anti-cytokeratin (CK)7, anti-CK20, anti-CK19, anti-caudal-type homeobox 2 (CDX2), anti-human epidermal growth factor receptor 2 (HER2), and anti-Ki67. Our series included 40 adult patients (≥18 years old) with a male:female ratio of 3:1, in favor of male patients. The mean age of all patients was 66.42 years. The primary sites of spinal metastases from GI cancers were from all segments of the GI system: the most frequent, however, was from the colorectal level (40%) and the least from the oral cavity level (2.5%). The most common site of spinal metastases from GI cancer was predominantly lumbar region (47.5%), and the most frequent histological type was adenocarcinoma (57.5%), followed by hepatocellular carcinoma (27.5%), pancreatic ductal carcinoma (5%) and squamous cell carcinoma (2.5%). Our results have important clinical implications because they suggest that there are certain subsets of patients with certain types of GI cancers that cause metastases in certain regions of the spine.
... 16,17 The lesion is typically irregular in shape, containing bone fragments that can be seen by CT and MRI. 10 Among the treatment options available for ADCC, including radical surgery, radiotherapy, chemotherapy, and concurrent chemotherapy with radiotherapy, the most common modality is radical surgery in combination with adjuvant radiotherapy. 9,10,12,18 The effect of adjuvant radiotherapy on the survival rate of patients with ADCC is highly debated. 11 It is now clear that ADCC is still a very complex disease to treat. ...
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.
... [2] Lee et al. [3] reported two cases of lumbar vertebral metastasis from the SCC of the tongue. One case of delayed metastasis to the C3 vertebral body from primary adenoid cystic carcinoma of the tongue was reported by Feng et al. [4] Metastasis to cervical vertebrae from oral SCC has also been reported by Carlson and Ord in 4 patients (0.7%) of 597 patients in their series. [2] Although metastasis to the spine is usually extradural, intramedullary metastasis has also been reported by Törnwall et al. [5] in one case. ...
... Spine metastases are extremely rare, and often occur locally (cervical), though perineural invasion in surgical resection with positive margins has been reported [3,4,7,12]; perineural infiltration, hematogenous dissemination and frequent positive local margins [12] make surgical eradication difficult. For all these reasons, later relapse is common [17][18][19]. It is unclear whether tumor metastases result from hematogenous dissemination (via the venous drainage into Batson's venous plexus and then into the arterial arcade) or perineural invasion from the primary tumor. ...
... Although the lungs are the most common site of ACC metastasis, distant vertebral involvement seems to predict poorer median survival (20-20.6 months) [7,20]. As shown in Table 1, 16 cases of spine metastases of ACC were reported in the literature, with mean time to metastasis of 4.1 years (range, 0.2-35 years) [1,16,[18][19][20][21][22][23]. Trilling et al. identified 3 cases of ACC spine metastases, with a mean interval of 9.4 years from primary cancer to metastasis [24]. ...
Background:
Adenoid cystic carcinoma (ACC), or cylindroma, is a rare malignancy believed to arise from epithelial cells of salivary glands. It is a slow-growing but aggressive tumor with a propensity for perineural invasion. Metastases are common to the lung, but rare to the spine. The natural history of ACC spine metastases is unknown and progression is unpredictable.
Methods:
1) A case report was described for a patient diagnosed with spine ACC metastasis of the T6/T7 vertebrae. 2) A literature search was conducted on Medline via PubMed and the Cochrane databases according to the PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines to identify articles from 1973 to March 2019 using following keywords: adenoid cystic carcinoma; cylindroma; metastasis; salivary glands. Results- Case report We report an unusual case of spine ACC metastasis of the T6/T7 vertebrae in a patient suffering from progressive radiculopathy and thoracic pain. Based on these findings, the patient underwent T6/T7 laminectomy. The diagnosis was confirmed by the pathology results. The primary parotid tumor had been resected 35 years before spine metastasis. A review of literature encompassing 40 years identified 16 patients treated for ACC spine metastases. Mean time to spine metastases was 4.1 years after primary treatment of ACC. Spine metastases were isolated, without local recurrence at the neck. Conclusion This case highlights the critical importance of long follow up of ACC patients, due to late spine metastases. Diagnosis spinal metastasis of salivary carcinoma should be considered, to guide management, especially in clinical follow-up.
... Lee et al. [10] reported two cases of lumbar vertebral metastasis from SCC of tongue. One case of delayed metastasis to C3 vertebral body from primary adenoid cystic carcinoma of tongue was reported by Feng et al. [3] Metastasis to cervical vertebrae from oral SCC has also been reported by Carlson et al. in 4 patients (0.7%) out of 597 patients in their series. [2] Although metastasis to spine is usually extradural, intramedullary metastasis has also been reported by Tornwall et al. in one case. ...
Background:
Carcinoma of tongue is a common site of oral cancer. It usually occurs at mean age of 61.1 years and is more common in males when compared with females. It commonly spreads directly and through lymphatics to the surrounding structures. It has a low incidence of hematogenous metastasis. Lung is the usual distant metastasis site for carcinoma of tongue and other head and neck cancers. Metastases to vertebrae are rare and very few cases have been reported.
Case description:
We report a rare case of carcinoma of tongue spreading to lumbar vertebrae causing destruction of the body and thecal sac compression. A patient underwent posterior transpedicular approach, tumor decompression, and titanium cage placement. The patient had good relief of symptoms and could be mobilized on first postoperative day.
Conclusion:
Hematogenous spread to the spine is a rare phenomenon but should be kept in mind particularly in advanced stage of oral cancers.
... DiscussionAdenoid cystic carcinoma most commonly arises in the parotid, submandibular and minor salivary glands [1]. However, it can also occur in other sites, including the tongue which accounts for 4.4% of cases [2]. This therefore establishes the uncommon nature of the left-sided ACC tongue mass in this case. ...
Adenoid cystic carcinoma (ACC) is a rare epithelial malignancy arising from secretory glands, particularly the salivary glands. It tends to invade nerves and has a high potential for distant hematogenous metastasis, especially to the lungs, bone, liver and brain. The breast and hypophysis are not common sites of ACC metastatic disease. Herein, we report a case of ACC of the head and neck region with two unusual sites of metastases, the hypophysis and breast.
Adenoid cystic carcinoma (ACC) of the sublingual gland is an extremely rare neoplasm. The clinicopathological characteristics of ACC are slow-growing swelling with or without ulceration, perineural spread, local recurrence, and distant metastasis. This report describes a 58-year-old male who had a slowly growing swelling without ulceration on the right side of the mouth floor that had been present for 1 month. In a radiological examination, the mass showed multilocular cystic features and no bony or tongue muscle invasion. No enlarged cervical lymph nodes were detected. Excisional biopsy and histological analysis showed that the lesion was ACC. In addition to reporting a rare case of ACC, this report also discusses the differential diagnosis and treatment of ACC with a review of the relevant literature.
