RADI-04. Stereotactic radiosurgery in alveolar soft part sarcoma brain metastasis

Abstract Background Alveolar soft part sarcoma (ASPS), although rare, has the highest incidence of brain metastasis amongst all sarcomas. Stereotactic radiosurgery (SRS) has been shown to be a well tolerated and effective treatment of intracranial sarcomatous metastasis. However, there is a paucity of published literature that guides radiation therapy in this condition. Methods This is a single centre retrospective review of all ASPS patients with intraparenchymal brain metastasis in our centre treated with stereotactic radiosurgery (SRS). SRS dosing is dichotomised into high and low dose (≥25 Gy and <25 Gy respectively) and outcomes such as local recurrence (LR) and radiation effects are noted. Successful treatment was defined as a lesion that regressed, is stable, or has less than 25% increase in tumour volume. Local recurrence (LR) was defined as increase in tumour volume by more than 25% during follow up. Results There were three patients with 11 ASPS metastatic brain lesions, one of which underwent retreatment. Each lesion was followed up for a mean duration of 12 months (range: 5 – 22 months). Five lesions treated with a high dose regime and six lesions were given low dose. Lesions treated with high dose SRS experienced significantly less LR (20% vs 83.3%, OR 20.0 [95%CI 0.93 – 430), p = 0.036) with no increase in undue symptomatic radiation effects. Retreatment of lesions with LR after initial SRS using a low dose regime was successful, albeit only in the single recurrent lesion. Conclusions We conclude that SRS can be used as a first line treatment for ASPS brain metastasis that are not surgically accessible and that using a high dose for treatment is effective and safe. Multicentre collaborative studies can be performed to validate this claim.

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Brain Metastasis of Alveolar Soft Part Sarcoma treated Successfully with Hypothermia: A Case Report

Japanese Journal of Neurosurgery ◽

10.7887/jcns.6.408 ◽

1997 ◽

Vol 6 (6) ◽

pp. 408-412

Author(s):

Satoshi Sawauchi ◽

Shigehiko Kabe ◽

Kouichi Takahashi ◽

Masahiko Akiyama ◽

Ikuo Shukuya ◽

...

Keyword(s):

Case Report ◽

Brain Metastasis ◽

Soft Part ◽

Alveolar Soft Part Sarcoma

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A case of multiple synchronously diagnosed brain metastases from alveolar soft part sarcoma without concurrent lung involvement

Surgical Neurology International ◽

10.25259/sni_554_2020 ◽

2021 ◽

Vol 12 ◽

pp. 111

Author(s):

Mark A. Damante ◽

Kristin M. Huntoon ◽

Joshua D. Palmer ◽

David A. Liebner ◽

James Bradley Elder

Keyword(s):

Soft Tissue ◽

Brain Metastasis ◽

Lung Metastasis ◽

Soft Tissue Mass ◽

Soft Part ◽

Primary Lesion ◽

Alveolar Soft Part Sarcoma ◽

Lung Involvement ◽

Tissue Mass ◽

Intracranial Involvement

Background: Alveolar soft part sarcoma (ASPS) is a rare soft-tissue sarcoma with a propensity for early hematogenous dissemination to the lungs and frequent brain metastasis. The development of lung metastasis almost invariably precedes intracranial involvement. There are no previously reported cases in which a patient was synchronously diagnosed with ASPS and multiple brain metastasis without lung involvement. Case Description: A 29-year-old gentleman was found to have three intracranial lesions following the onset of generalized seizures. Staging studies identified a soft-tissue mass in the left thigh and an adjacent femoral lesion. Biopsy of the soft-tissue mass was consistent with ASPS. The patient then underwent neoadjuvant stereotactic radiotherapy to all three brain lesions, followed by en bloc resection of the dominant lesion. The patient was then started on a programmed death-ligand 1 (PD-L1) inhibitor. Subsequent surgical resection of the primary lesion and femur metastasis demonstrates a histopathologic complete response of the bony metastasis and partial response of the primary lesion. At present, the patient has received 14 cycles of atezolizumab without recurrence of the primary or bony lesions and the irradiated intracranial disease has remained stable without recurrence of the resected dominant lesion. Conclusion: While intracranial involvement is relatively common in ASPS, a case with multiple, synchronously diagnosed brain metastasis without concurrent lung metastasis has not been described. The presented case discusses the safety and efficacy of aggressive management of intracranial disease in the setting of atezolizumab. Prospective evaluation of the efficacy of checkpoint inhibitors and the prognostic value of PD-L1 expression in ASPS with brain metastasis are necessary.

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Ablation therapy using a low dose of radioiodine may be sufficient in low- to intermediate-risk patients with follicular variant papillary thyroid carcinoma

Journal of International Medical Research ◽

10.1177/0300060520966491 ◽

2020 ◽

Vol 48 (11) ◽

pp. 030006052096649

Author(s):

Fuxin Li ◽

Wei Li ◽

Katherine D. Gray ◽

Rasa Zarnegar ◽

Dan Wang ◽

...

Keyword(s):

Lymph Node ◽

Papillary Thyroid Carcinoma ◽

Local Recurrence ◽

Thyroid Carcinoma ◽

Low Dose ◽

High Dose ◽

Intermediate Risk ◽

Papillary Thyroid ◽

Follicular Variant ◽

Remnant Ablation

Objectives Follicular variant papillary thyroid carcinoma (FVPTC) is treated similarly to classical variant papillary thyroid carcinoma (cPTC). However, FVPTC has unique tumour features and behaviours. We investigated whether a low dose of radioiodine was as effective as a high dose for remnant ablation in patients with FVPTC and evaluated the recurrence of low-intermediate risk FVPTC. Methods Data from cPTC and FVPTC patients treated with I-131 from 2004 to 2014 were reviewed. Demographics, tumour behaviour, lymph node metastasis, and local recurrence data were compared between FVPTC and cPTC patients. Then, low-intermediate risk FVPTC patients were divided into low, intermediate, and high I-131 dose groups, and postoperative I-131 activities were analysed to evaluate the effectiveness of I-131 therapy for thyroid remnant ablation. Results In total, 799 cases of FVPTC (n = 168) and cPTC (n = 631) treated with I-131 were identified. Patients with FVPTC had a larger primary nodule size than cPTC, but lymph node metastases and local recurrence were more prevalent in cPTC than in FVPTC. For the low-, intermediate-, and high-dose groups, success rates of ablation did not differ (82.0%, 80%, and 81.3%, respectively). Conclusion FVPTC differs from cPTC in behaviour. Low-dose ablation may be sufficient in FVPTC patients with low-intermediate disease risk.

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Advanced Alveolar Soft Part Sarcoma Treated with Pazopanib over Three Years

Case Reports in Oncological Medicine ◽

10.1155/2017/3738562 ◽

2017 ◽

Vol 2017 ◽

pp. 1-4 ◽

Cited By ~ 1

Author(s):

Yoji Shido ◽

Yukihiro Matsuyama

Keyword(s):

Brain Metastasis ◽

Brain Metastases ◽

Kinase Inhibitor ◽

Lung Tumor ◽

Lung Metastases ◽

Soft Part ◽

Objective Response ◽

Alveolar Soft Part Sarcoma ◽

Longitudinal Assessment ◽

Metastatic Lung Tumor

Alveolar soft part sarcoma (ASPS) is a rare malignant tumor that generally occurs in adolescents and young adults. It progresses slowly, but lung and brain metastases often occur in the early phase of the clinical course, and chemotherapy has been reported as not being effective for ASPS. Pazopanib is a multitargeted tyrosine kinase inhibitor that has been clinically available from November 2012 in Japan. This is a case report of a patient presented with multiple lung metastases and unresectable primary abdominal ASPS. We initially treated this patient by systemic chemotherapy with combination use of ifosfamide and doxorubicin. Stable disease was observed without any objective response. Then, we finally started to administrate pazopanib 800 mg/day. After 25 months of pazopanib administration, slight tumor reduction and a decrease of enhancement were observed. Objective responses were achieved for both the primary tumor and metastatic lung tumor; however, a newly developed brain metastasis was subsequently identified. Based on this case, pazopanib appears effective against ASPS, except for brain metastases. This case suggests that pazopanib may be useful as a first-line drug against unresectable ASPS and that longitudinal assessment of brain metastasis should be performed in similar cases.

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Pediatric Primary Alveolar Soft Part Sarcoma of the Bladder

Case Reports in Urology ◽

10.1155/2018/1284756 ◽

2018 ◽

Vol 2018 ◽

pp. 1-4 ◽

Cited By ~ 1

Author(s):

Kazuya Tanabe ◽

Shigeru Nakamura ◽

Taiju Hyuga ◽

Shina Kawai ◽

Masahiro Yamazaki ◽

...

Keyword(s):

Local Recurrence ◽

Bladder Tumor ◽

Clinical Course ◽

Soft Part ◽

Surgical Margin ◽

Residual Tumor ◽

Alveolar Soft Part Sarcoma ◽

Positive Surgical Margin ◽

Continent Urinary Reservoir ◽

Urinary Reservoir

A 9-year-old girl was diagnosed with primary alveolar soft part sarcoma of the bladder after imaging examinations and transurethral resection (TUR) of the bladder tumor. As a positive surgical margin of the TUR indicated residual tumor cells, we performed a cystourethrectomy to remove the tumor. A continent urinary reservoir for self-catheterization was constructed using the Mainz pouch technique, and an abdominal (umbilical) continent catheterizable stoma using the appendix was performed. For 2.5 years postoperatively, the patient remained free of local recurrence and distant metastasis. The patient’s clinical course has been favorable, with good management of clean intermittent self-catheterization.

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Stereotactic radiosurgery in alveolar soft part sarcoma brain metastases: Case series and literature review

Journal of Clinical Neuroscience ◽

10.1016/j.jocn.2021.09.002 ◽

2021 ◽

Vol 93 ◽

pp. 227-230

Author(s):

Jia Xu Lim ◽

Bengt Karlsson ◽

Angela Pang ◽

Balamurugan A. Vellayappan ◽

Vincent Nga

Keyword(s):

Literature Review ◽

Brain Metastases ◽

Stereotactic Radiosurgery ◽

Soft Part ◽

Case Series ◽

Alveolar Soft Part Sarcoma

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Successful treatment for solitary brain metastasis from alveolar soft part sarcoma

Journal of Neuro-Oncology ◽

10.1007/bf01057760 ◽

1995 ◽

Vol 25 (2) ◽

pp. 161-166 ◽

Cited By ~ 11

Author(s):

Cheng -Hsu Wang ◽

Ning Lee ◽

Liang -Shong Lee

Keyword(s):

Brain Metastasis ◽

Successful Treatment ◽

Soft Part ◽

Alveolar Soft Part Sarcoma ◽

Solitary Brain Metastasis

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Integrating salvage hypofractionated radiosurgery plus low-dose adjuvant bevacizumab for locally recurrent brain metastasis with high dose irradiation previously.

Journal of Clinical Oncology ◽

10.1200/jco.2019.37.15_suppl.e13579 ◽

2019 ◽

Vol 37 (15_suppl) ◽

pp. e13579-e13579 ◽

Cited By ~ 1

Author(s):

Yun Guan ◽

Huaguang Zhu ◽

Chaozhaung Wang ◽

Jing Li ◽

Wenqian Xu ◽

...

Keyword(s):

Overall Survival ◽

Brain Metastasis ◽

Adverse Events ◽

Low Dose ◽

Salvage Treatment ◽

Karnofsky Performance Scale ◽

High Dose ◽

Intracranial Metastasis ◽

Dose Irradiation ◽

Locally Recurrent

e13579 Background: Treatment for recurrent brain metastasis remains uncertain. The purpose of this research was to evaluate the efficacy and toxicity of FSRS with adjuvant bevacizumab as a new salvage treatment for locally recurrent brain metastases with high dose irradiation previously. Methods: Patients with locally recurrent intracranial metastasis with irradiation previously were enrolled in this prospective study. Salvage FSRS dose ranged from 9.5 to 29 Gy in two to five sessions with 62–75% isodose line. Adjuvant bevacizumab was prescribed four cycles (5 mg/kg, q3w). Magnetic resonance imaging (MRI), Karnofsky Performance Scale (KPS), adverse events and clinical outcomes were recorded. The primary objective of this study was to identify the overall survival after salvage treatment. Secondary objectives included clinical response (KPS), imaging response (MRI) and adverse events. Results: From December 2009 to October 2016, 24 patients were enrolled. Of these patients, 9 received WBRT followed by SRS, and 15 underwent SRS. The median 1-year overall survival (OS) after salvage SRS was 87.5%. No treatment-related grade 4 adverse events were recorded. Grade 1/2 fatigue was observed in 8 (33%) patients followed by headache, hypertension and nausea. Grade 3 fatigue and headache occurred in 1 patient. No grade 4 toxicity was observed. Conclusions: Salvage FSRS with early use of low dose adjuvant bevacizumab treatment showed favourable clinical and radiologic control with manageable toxicity for locally recurrent brain metastasis patients who underwent high dose irradiation previously. The diagnosis of RN and LR after salvage FSRS merit further study. [Table: see text][Table: see text][Table: see text]

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Subnecrotic stereotactic radiosurgery controls epilepsy produced by kainic acid injection in rats

Journal of Neurosurgery ◽

10.3171/jns.2000.93.6.1033 ◽

2000 ◽

Vol 93 (6) ◽

pp. 1033-1040 ◽

Cited By ~ 49

Author(s):

Satoshi Maesawa ◽

Douglas Kondziolka ◽

C. Edward Dixon ◽

Jeffrey Balzer ◽

Wendy Fellows ◽

...

Keyword(s):

Stereotactic Radiosurgery ◽

Kainic Acid ◽

Probe Trial ◽

Low Dose ◽

Morris Water Maze Test ◽

High Dose ◽

Behavioral Impairment ◽

Content Type ◽

Chronic Epilepsy ◽

Fine Print

Object. Any analysis of the potential role of stereotactic radiosurgery for epilepsy requires the experimental study of its potential antiepileptogenic, behavioral, and histological effects. The authors hypothesized that radiosurgery performed using subnecrotic tissue doses would reduce or abolish epilepsy without causing demonstrable behavioral side effects. The kainic acid model in rats was chosen to test this hypothesis.Methods. Chronic epilepsy was successfully created by stereotactic injection of kainic acid (8 µg) into the rat hippocampus. Epileptic rats were divided into three groups: high-dose radiosurgery (60 Gy, 16 animals), low-dose (30 Gy, 15 animals), and controls. After chronic epilepsy was confirmed by observation of the seizure pattern and by using electroencephalography (EEG), radiosurgery was performed on Day 10 postinjection. Serial seizure and behavior observation was supplemented by weekly EEG sessions performed for the next 11 weeks. To detect behavioral deficits, the Morris water maze test was performed during Week 12 to study spatial learning and memory. Tasks involved a hidden platform, a visible platform, and a probe trial.After radiosurgery, the incidence of observed and EEG-defined seizures was markedly reduced in rats from either radiosurgically treated group. A significant reduction was noted after high-dose (60 Gy) radiosurgery in Weeks 5 to 9 (p < 0.003). After low-dose (30 Gy) radiosurgery, a significant reduction was found after 7 to 9 weeks (p < 0.04). During the task involving the hidden platform, kainic acid—injected rats displayed significantly prolonged latencies compared with those of control animals (p < 0.05). Hippocampal radiosurgery did not worsen this performance. The probe trial showed that kainic acid—injected rats that did not undergo radiosurgery spent significantly less time than control rats in the target quadrant (p = 0.03). Rats that had undergone radiosurgery displayed no difference compared with control rats and demonstrated better performance than rats that received kainic acid alone (p = 0.04). Radiosurgery caused no adverse histological effects.Conclusions. In a rat model, radiosurgery performed with subnecrotic tissue doses controlled epilepsy without causing subsequent behavioral impairment.

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