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Case Report Open Access

Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report

  • Authors:
    • Bin He
    • Jie Shi
    • Qing-Yan Yan
    • Ling Zhang
  • View Affiliations / Copyright

    Affiliations: Department of Oncology, The General Hospital of Western Theater Command of the Chinese People's Liberation Army, Chengdu, Sichuan 610083, P.R. China
    Copyright: © He et al. This is an open access article distributed under the terms of Creative Commons Attribution License.
  • Article Number: 566
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    Published online on: October 1, 2025
       https://doi.org/10.3892/ol.2025.15312
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Abstract

Maxillary adenoid cystic carcinoma (ACC) is rare, with pleural and brain metastases representing an unprecedented presentation. The present case report illustrates the challenges and potential for prolonged survival in such an advanced stage of malignant disease. A 52‑year‑old male patient presented with a left maxillary mass, initially misdiagnosed as ameloblastoma; subsequently, it was confirmed as maxillary ACC (T4aN0M0, stage IVA). Following primary tumor resection and radiotherapy, the patient developed pleural (February 2021) and brain metastases (March 2022). A multimodal treatment strategy was used, involving aggressive surgical resection of the primary tumor, comprehensive radiotherapy, systemic chemotherapy regimens and adjuvant targeted molecular therapy. By using this sequential multimodal therapy, the patient achieved long‑term survival exceeding 5 years. The present case report highlights the potential for favorable, prolonged survival even in the presence of multifocal metastatic maxillary ACC when managed with a persistent, sequential and tailored combination of modern therapeutic modalities.
View Figures

Figure 1

Initial imaging findings of the left
maxillary mass. (A) Oral panoramic radiograph indicating an oval
osteolytic lesion ~3 cm in size. (B) Maxillofacial CT indicates a
localized osteolytic lesion in the left maxilla involving adjacent
alveolar bone and left maxillary sinus floor, with a centered
4.7×4.2 cm soft tissue mass.

Figure 2

Histopathological and
immunohistochemical features of primary adenoid cystic carcinoma.
(A) Tumor cells are arranged in nests of varying sizes, exhibiting
a cribriform pattern with cystic spaces containing basophilic
mucoid material, reminiscent of a lotus root cross-section. A layer
of mucin-secreting myoepithelial cells surrounds the cystic spaces
(magnification, ×100). (B) Positive for actin (magnification,
×100). (C) Positive for CD117 (magnification, ×100). (D) Positive
for cytokeratin 8/18 (magnification, ×100). (E) Positive for
epithelial membrane antigen. (F) Ki-67 proliferation index is 20%
(magnification, ×100). (G) Positive for P63 (magnification, ×100).
(H) Positive for S-100 (magnification, ×100).

Figure 3

Treatment strategy and clinical
course. (A) Outline of a CARE-guided workflow for comprehensive
case reporting, encompassing initial assessment, therapeutic
interventions and longitudinal follow-up of this rare malignancy.
(B) Timeline illustrating the sequential adaptation of multimodal
therapy for pleural and intracranial metastases, achieving
sustained disease control and 68-month overall survival. ACC,
adenoid cystic carcinoma; IHC, immunohistochemistry; CK,
cytokeratin; MRI, magnetic resonance imaging; PR, partial response;
PD, progressive disease; SD, stable disease.

Figure 4

Histopathological confirmation of
pleural metastasis. (A) Poorly differentiated tumor cell components
demonstrating significant atypia against a background of necrosis
(magnification, ×100). (B) Positive for cytokeratin 8/18
(magnification, ×100). (C) Weakly positive for smooth muscle actin
(magnification, ×100). (D) Positive for vimentin (magnification,
×100).

Figure 5

Serial chest CT demonstrating
treatment response of pleural metastases. March 2021: (Aa-Ca)
Multiple irregular, enhancing pleural masses in the right chest
wall (arrows); (Da) a soft tissue nodule in the left lower lobe
posterior basal segment invading adjacent bronchus, with distal
obstructive inflammation. June 2021: (Ab-Db) Notable reduction in
right pleural and left lung lesions. (Ac-Dc) March 2025: No new
lesions in right pleura or lungs.

Figure 6

Follow-up brain MRI of the left
occipital lobe metastasis. Initial MRI. (Aa) T2-FLAIR: 2.1×1.9 cm
left occipital lobe mass with extensive perilesional edema (arrow).
(Ba) T1WI: Mass isointense to slightly hypointense (arrow). (Ca)
Contrast T1WI: Irregular rim enhancement (arrow). (Ab-Cb) August
2022: Lesion size reduced, enhancing rim thinned, and perilesional
edema nearly resolved. (Ac-Cc) March 2025: Stable residual cavity,
no significant change, no new nodules/edema. T1WI, T1-weighted
imaging.

Figure 7

Follow-up brain MRI of the cerebellar
metastasis. (Aa) T2WI/FLAIR: Patchy left cerebellar hemisphere
hyperintensity (edema) with indistinct margins and mass effect
(arrow). (Ba) T1WI: Slightly hypointense lesion(arrow). (Ca)
Contrast T1WI: Irregular patchy lesion with peripheral-predominant
(rim) heterogeneous enhancement (arrow). (Ab-Cb) October 2024:
Marked lesion regression, decreased enhancement, near-complete
edema resolution. (Ac-Cc) March 2025: Punctate/small patchy
residual enhancement only, no new nodular enhancement or edema.
T1WI, T1-weighted imaging.
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Copy and paste a formatted citation
Spandidos Publications style
He B, Shi J, Yan Q and Zhang L: Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report. Oncol Lett 30: 566, 2025.
APA
He, B., Shi, J., Yan, Q., & Zhang, L. (2025). Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report. Oncology Letters, 30, 566. https://doi.org/10.3892/ol.2025.15312
MLA
He, B., Shi, J., Yan, Q., Zhang, L."Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report". Oncology Letters 30.6 (2025): 566.
Chicago
He, B., Shi, J., Yan, Q., Zhang, L."Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report". Oncology Letters 30, no. 6 (2025): 566. https://doi.org/10.3892/ol.2025.15312
Copy and paste a formatted citation
x
Spandidos Publications style
He B, Shi J, Yan Q and Zhang L: Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report. Oncol Lett 30: 566, 2025.
APA
He, B., Shi, J., Yan, Q., & Zhang, L. (2025). Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report. Oncology Letters, 30, 566. https://doi.org/10.3892/ol.2025.15312
MLA
He, B., Shi, J., Yan, Q., Zhang, L."Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report". Oncology Letters 30.6 (2025): 566.
Chicago
He, B., Shi, J., Yan, Q., Zhang, L."Multimodal treatment for multifocal metastatic maxillary adenoid cystic carcinoma: A case report". Oncology Letters 30, no. 6 (2025): 566. https://doi.org/10.3892/ol.2025.15312
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