Thigh leiomyosarcoma-derived brain metastasis with intracerebral hematoma: A case report and literature review
- Department of Neurosurgery, Yokohama City University, Yokohama, Japan.
- Department of Orthopedics, Yokohama City University, Yokohama, Japan.
- Department of Pathology, Yokohama City University, Yokohama, Japan.
Yohei Miyake, Department of Neurosurgery, Yokohama City University, Yokohama, Japan.
DOI:10.25259/SNI_113_2023Copyright: © 2023 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: Chihiro Oka1, Yohei Miyake1, Kensuke Tateishi1, Yusuke Kawabata2, Hiromichi Iwashita3, Tetsuya Yamamoto1. Thigh leiomyosarcoma-derived brain metastasis with intracerebral hematoma: A case report and literature review. 03-Mar-2023;14:80
How to cite this URL: Chihiro Oka1, Yohei Miyake1, Kensuke Tateishi1, Yusuke Kawabata2, Hiromichi Iwashita3, Tetsuya Yamamoto1. Thigh leiomyosarcoma-derived brain metastasis with intracerebral hematoma: A case report and literature review. 03-Mar-2023;14:80. Available from: https://surgicalneurologyint.com/surgicalint-articles/12178/
Background: Brain metastases with hematoma are clinically important as they indicate the potential for rapid neurological deterioration. Non-uterine leiomyosarcoma-derived brain metastases are particularly rare, and their clinical features, including the bleeding rate, are unclear. Herein, we present a rare case of thigh leiomyosarcoma-derived brain metastasis with intratumoral hematoma and review previous case reports.
Case Description: A 68-year-old man with a right thigh leiomyosarcoma presented with multiple brain metastases. The patient received stereotactic radiotherapy; however, he reported sudden right-sided hemiparesis. We found a right frontal irradiated lesion with intratumoral hemorrhage and performed gross total tumor resection. Histopathological examination showed highly atypical cells with prominent necrosis and hemorrhage. Abnormal thin-walled vessels were prominent within the brain tumor, and vascular endothelial growth factor was diffusely expressed immunohistopathologically. To date, 11 cases of brain metastasis from non-uterine leiomyosarcoma, including the present case, have been reported. Of note, six patients had hemorrhage. Three out of six patients presented with hemorrhage before therapeutic intervention, three cases were from residual sites after surgery or radiation.
Conclusion: More than half the patients with non-uterine leiomyosarcoma-derived brain metastases presented with intracerebral hemorrhage. Furthermore, these patients are at risk of developing rapid neurological deterioration due to intracerebral hemorrhage.
Keywords: Brain metastasis, Hemorrhage, Non-uterine leiomyosarcoma
Brain metastases occasionally present with intratumoral bleeding, especially in renal cell carcinoma and malignant melanoma.[
A 68-year-old man presented with swelling on the right thigh. Magnetic resonance imaging (MRI) revealed a contrast-enhanced tumor with a hemorrhagic and unclear border [
Clinicopathological features of the thigh leiomyosarcoma. Gadolinium-enhanced T1-weighted magnetic resonance imaging demonstrating a heterogeneously enhanced tumor in the right thigh (a), hematoxylin and eosin staining indicate dense tumor cell proliferation with necrosis and hemorrhage (b), and tumors showing spindle cells with highly pleomorphic nuclei (c).
Radiographic features of the metastatic brain tumor derived from leiomyosarcoma of the thigh. Gadolinium-enhanced T1-weighted magnetic resonance imaging (MRI) demonstrating multiple brain tumors (a and b, arrows). Computed tomography and MRI showing the right frontal tumor with a surrounding hematoma (c-e). MRI indicating total resection of the tumor (f).
Pathological characteristics of the metastatic brain tumor derived from leiomyosarcoma of the thigh. Macroscopic view showing the tumor (arrowhead) and the surrounding hematoma (arrows) (a). Hematoxylin and eosin staining indicates diffuse growth of atypical cells with necrosis (arrowhead) and hemorrhage (arrows) (b). Hemorrhage comprising tumor cells (c). Tumor cells showing spindle and epithelioid features with highly pleomorphic nuclei (d). Immunostaining for desmin (e), caldesmon (f), Ki-67 (g), vascular endothelial growth factor (h), and CD31 (i). Thin-walled vessels are prominent within the tumor (i, arrows).
Leiomyosarcoma is a soft-tissue sarcoma that occurs in the uterus, gastrointestinal tract, blood vessels, and extremities.[
Notably, six (54.5%) patients demonstrated co-occurrence of brain metastases with hemorrhagic components.[
Although the underlying mechanisms of hemorrhage within brain tumors are unclear, abnormal tumor vessels, such as thin-walled, poorly formed, or dilated vessels, may contribute to intratumoral hemorrhage.[
In our patient, the small lesions disappeared after stereotactic radiotherapy, except for a 2 cm lesion in the right frontal lobe. Similarly, whole-brain irradiation controlled the small lesions, but the most significant lesion, measuring 2 cm, grew and required resection.[
We reported a rare case of thigh leiomyosarcoma-derived brain metastasis. The majority of non-uterine leiomyosarcoma-derived brain metastases present with intracerebral hemorrhage even after treatment. These findings suggest that non-uterine leiomyosarcoma-derived brain metastases may pose a risk of rapid neurological deterioration due to intracerebral hemorrhage and require early therapeutic intervention.
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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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