- Department of Neurosurgery, The University of Tokyo Hospital, Tokyo, Japan,
- Department of Neurosurgery, Teikyo University Hospital, Tokyo, Japan,
- Department of Radiology, The University of Tokyo Hospital, Tokyo, Japan,
- Department of Pathology, The University of Tokyo Hospital, Tokyo, Japan,
- Department of Otorhinolaryngology, The University of Tokyo Hospital, Tokyo, Japan.
Motoyuki Umekawa, Department of Neurosurgery, The University of Tokyo Hospital, Tokyo, Japan.
DOI:10.25259/SNI_675_2022Copyright: © 2022 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Yuki Nakamura1, Motoyuki Umekawa1, Yuki Shinya1, Hirotaka Hasegawa1, Masahiro Shin2, Atsuto Katano3, Aya Shinozaki-Ushiku4, Kenji Kondo5, Nobuhito Saito1. Stereotactic radiosurgery for skull base adenoid cystic carcinoma: A report of two cases. 04-Nov-2022;13:509
How to cite this URL: Yuki Nakamura1, Motoyuki Umekawa1, Yuki Shinya1, Hirotaka Hasegawa1, Masahiro Shin2, Atsuto Katano3, Aya Shinozaki-Ushiku4, Kenji Kondo5, Nobuhito Saito1. Stereotactic radiosurgery for skull base adenoid cystic carcinoma: A report of two cases. 04-Nov-2022;13:509. Available from: https://surgicalneurologyint.com/surgicalint-articles/11976/
Background: Adenoid cystic carcinoma (ACC) is an uncommon salivary gland tumor with a relatively favorable prognosis. However, treating ACC is potentially challenging because radical resection is usually difficult once the skull base is involved due to the adjacent critical structures. Stereotactic radiosurgery (SRS) is a less invasive alternative for surgically recalcitrant lesions.
Case Description: We report two patients with three metastatic skull base ACCs who underwent SRS using the Gamma Knife with a marginal dose of 20 Gy to a 50% isodose line. All tumors were effectively controlled without any adverse events.
Conclusion: This case report and our review of the literature suggest that SRS can be considered for local control of ACC invading the skull base when surgical resection is unsuitable or a postoperative residual lesion is suspected. Further, investigations on the accumulated subjects are warranted to confirm the role of SRS for the treatment of ACCs.
Keywords: Adenoid cystic carcinoma, Case report, Recurrence, Skull base, Stereotactic radiosurgery
Adenoid cystic carcinoma (ACC) accounts for only ~1% of head and neck malignancies; it typically arises from salivary glands.[
Stereotactic radiosurgery (SRS) comprises focused high-dose irradiation at a single session. It yields 1-year local control of 60–90% of malignant metastatic brain tumors and is efficacious in the treatment of brain metastases and skull base tumors.[
A 50-year-old man with a nosebleed was referred to our hospital. He had a medical history of pituitary adenoma which was totally resected at the age of 17 without recurrence, myocardial infarction, hypertension, and dyslipidemia. Magnetic resonance imaging (MRI) revealed a heterogeneously enhanced tumor extending from the nasal cavity to the suprasellar region [
Adenoid cystic carcinoma (ACC) in a 50-year-old man (Case 1). Gadolinium-enhanced, T1-weighted magnetic resonance, imaging revealed an enhanced tumor extending from the nasal cavity to the suprasellar region (a). The tumor was pathologically confirmed as ACC, exhibiting cribriform growth with myoepithelial differentiation (b). After two resections, he experienced recurrence in the superior orbital fissure, at age 56, and stereotactic radiosurgery (SRS) was performed with a marginal dose of 20 Gy to a 50% isodose line (yellow line) in axial (c), coronal (d), and sagittal plane (e) Green lines indicated marginal dose line of 10 Gy, 18 Gy, and 24 Gy. A remote metastasis was detected in the parapharyngeal space, with marginal recurrence after its resection. We performed the second SRS for the parapharyngeal metastasis with the same parameters and the planning was shown in axial (f), coronal (g), and sagittal plane (h) with the previous planning in blue line. Yellow line indicated 20 Gy to a 50% isodose line, and green lines indicated marginal dose line of 10 Gy, 12 Gy, and 18 Gy. Both lesions were in remission at the last follow-up: 36 months after the first SRS for recurrence in the superior orbital fissure and 9 months after the second SRS for recurrence in the parapharyngeal space (i-k).
A 56-year-old woman with the right facial numbness and trigeminal neuralgia was referred to our hospital. She had a medical history of gastric cancer which was treated with surgery 7 years before her visit and showed no recurrence. MRI revealed a large tumor extending from the orbit to the cavernous sinus. We considered this skull base lesion as the primary site, because the initial whole body-CT scan revealed no evidence of any tumor. It was resected and pathologically confirmed as ACC arising from the lacrimal gland [
A 56-year-old woman underwent resection for a tumor growing next to the right orbit and the anterior skull base (Case 2). The tumor was pathologically diagnosed as adenoid cystic carcinoma arising from the lacrimal gland (a). Despite repeated resection, she experienced recurrence around the right cavernous sinus and orbit. We performed stereotactic radiosurgery for the anterior skull base metastasis, with a marginal dose of 20 Gy to a 50% isodose line (yellow line) in axial (b), coronal (c), and sagittal plane (d) Green lines indicated marginal dose line of 10 Gy, 18 Gy, and 24 Gy. The tumor shrank considerably in the next 5 months (e).
We reported on two cases of skull base metastatic ACCs that were successfully controlled with SRS without significant adverse radiation effects. Patients with ACCs have a relatively favorable prognosis even with metastatic tumors; the 5-, 10-, and 15-year survival rates for all stages of ACCs are 90%, 80%, and 69%, respectively.[
The minimally invasive nature and low rate of treatment-associated complications of SRS can be a major benefit for ACC, in which frequent local recurrence is a major issue. Although surgical removal is the standard treatment for the primary tumors, the optimal strategy for recurrent ACC remains to be determined. Given the possibility of various perioperative complications,[
We described two cases with three skull base metastases of ACC, for which we achieved successful local control with Gamma Knife-based SRS. SRS is a promising treatment option for skull base ACCs, both in terms of local control and functional preservation. Further, observations in more cases are required.
The authors certify that they have obtained all appropriate patient consent.
JSPS KAKENHI (grant number: 20K17919).
There are no conflicts of interest.
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
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