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Case Report Open Access

Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report

  • Authors:
    • Tianyi Li
    • Tingyao Ma
    • Yue Zhao
    • Shujing Zhang
    • Guoliang Yang
    • Xuelian Wang
    • Xudong Wang
    • Mingzhu Wang
    • Xi Zhao
    • Hongfei Liu
    • Jun Wu
    • Xiaoli Zhao
    • Xiaohong Chen
  • View Affiliations / Copyright

    Affiliations: Department of Otolaryngology‑Head and Neck Surgery, Beijing Tongren Hospital, Capital Medical University, Beijing 100730, P.R. China, Department of Pathology, Beijing Tongren Hospital, Capital Medical University, Beijing 100730, P.R. China
    Copyright: © Li et al. This is an open access article distributed under the terms of Creative Commons Attribution License.
  • Article Number: 596
    |
    Published online on: October 15, 2025
       https://doi.org/10.3892/ol.2025.15342
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Abstract

Adenoid cystic carcinoma (ACC) is a rare and aggressive malignancy characterized by a high incidence of distant metastasis. However, cases of ACC with liver and lymph node metastases are rarely reported and lack a standardized treatment consensus. The present report describes the case of a submandibular gland ACC with liver metastasis manifesting as the initial symptom. PET/CT revealed masses in the left submandibular gland and the liver. The patient underwent right tri‑lobe hepatectomy and resection of the submandibular gland tumor. Histological examination of the tumor revealed a cribriform type with both lymphovascular invasion and perineural invasion. New lymph node metastases were observed 21 months after surgery of the submandibular gland tumor, and no further treatment was administered to date. The patient has survived for 28 months since the diagnosis of liver metastasis and currently exhibits stable vital signs. In conclusion, the present case illustrates a rare, high‑risk variant of rapidly progressing submandibular gland ACC, with liver metastasis serving as the initial presenting symptom. A multidisciplinary approach is essential for the effective management of complex metastatic ACC.
View Figures

Figure 1

Abdominal CT image showing multiple
liver lesions.

Figure 2

Histological image of liver metastasis
in adenoid cystic carcinoma showing tumor cells arranged in tubular
and cribriform patterns (H&E stain; magnification, ×200).

Figure 3

Cervical MRI. Nodular lesion in the
(A) horizontal and (B) coronal plane with slightly elongated T1 and
(C) long T2 signals in the left submandibular gland region, with
(D) high signal on diffusion-weighted imaging.

Figure 4

Preoperative color Doppler ultrasound
of the salivary glands and draining lymph nodes. (A) Irregular
hypoechoic mass in the left submandibular area, with (B) sparse
blood flow signals around and within the lesion.

Figure 5

Histological image of submandibular
gland adenoid cystic carcinoma (hematoxylin-eosin stain). (A)
Predominantly composed of cribriform structures (80%), with tubular
(15%) and solid (5%) patterns, demonstrating high differentiation
without high-grade transformation (magnification, ×100). (B)
Perineural invasion and (C) lymphovascular invasion were observed
(magnification, ×200). (D) Lymph node exhibited metastasis exhibit
extranodal extension (magnification, ×40).

Figure 6

Immunohistochemical images of
submandibular gland adenoid cystic carcinoma, revealing positive
expression of Calponin, CD117, CK, CK7, CK8/18, P63, S-100, SOX-10,
Vimentin, β-catenin, C-Myc, EGFR, VEGF, and Ki-67 index in hot-spot
(10–15%), alongside negative expression of Her-2, GFAP, SOX-2, P53
and PD-L1 (magnification, ×100). CK, creatine kinase; CK7,
cytokeratin 7; CK8/18, cytokeratin 8/18; SOX, SRY-box transcription
factor; GFAP, glial fibrillary acidic protein; PD-L1, programmed
death-ligand 1.

Figure 7

MYB gene rearrangement was not
detected by fluorescence in situ hybridization in the
primary adenoid cystic carcinoma (magnification, ×1,000).

Figure 8

Cervical MRI enhancement in the (A)
coronal and (B) horizontal plane showing a heterogeneous and
markedly enhanced mass in the left cervical level Ib and II
regions.
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Copy and paste a formatted citation
Spandidos Publications style
Li T, Ma T, Zhao Y, Zhang S, Yang G, Wang X, Wang X, Wang M, Zhao X, Liu H, Liu H, et al: Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report. Oncol Lett 30: 596, 2025.
APA
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X. ... Chen, X. (2025). Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report. Oncology Letters, 30, 596. https://doi.org/10.3892/ol.2025.15342
MLA
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X., Wang, X., Wang, M., Zhao, X., Liu, H., Wu, J., Zhao, X., Chen, X."Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report". Oncology Letters 30.6 (2025): 596.
Chicago
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X., Wang, X., Wang, M., Zhao, X., Liu, H., Wu, J., Zhao, X., Chen, X."Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report". Oncology Letters 30, no. 6 (2025): 596. https://doi.org/10.3892/ol.2025.15342
Copy and paste a formatted citation
x
Spandidos Publications style
Li T, Ma T, Zhao Y, Zhang S, Yang G, Wang X, Wang X, Wang M, Zhao X, Liu H, Liu H, et al: Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report. Oncol Lett 30: 596, 2025.
APA
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X. ... Chen, X. (2025). Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report. Oncology Letters, 30, 596. https://doi.org/10.3892/ol.2025.15342
MLA
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X., Wang, X., Wang, M., Zhao, X., Liu, H., Wu, J., Zhao, X., Chen, X."Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report". Oncology Letters 30.6 (2025): 596.
Chicago
Li, T., Ma, T., Zhao, Y., Zhang, S., Yang, G., Wang, X., Wang, X., Wang, M., Zhao, X., Liu, H., Wu, J., Zhao, X., Chen, X."Submandibular adenoid cystic carcinoma presenting with liver metastasis as the initial symptom: A case report". Oncology Letters 30, no. 6 (2025): 596. https://doi.org/10.3892/ol.2025.15342
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