Brain Metastases in Pediatric Extracranial Solid Tumors: A 20-Year Experience in Challenging a Rare Diagnosis.

Dilek Gul, Beste M Atasoy, Burcu Tufan Tas, Adnan Dagcinar, Zerrin Ozgen
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Abstract

Aim: To investigate the clinical features, treatments, and outcomes of pediatric patients with brain metastasis from extracranial solid tumors, which has been known as a rare condition.

Material and methods: Over a 20-year period, 520 children treated for extracranial solid tumors in our radiotherapy clinic were reviewed, and 12 cases (2.2%) of brain metastases were identified. The primary tumors included neuroblastoma (n=5), osteosarcoma (n=3), Ewing sarcoma (n=2), rhabdoid tumor of the kidney (n=1), and Wilms tumor (n=1). Retrospective analysis was performed to evaluate tumor and treatment features. Overall survival was measured from the initial diagnosis. Overall survival after brain metastasis, local control, and brain metastasis progression-free survival were calculated from the time of brain metastasis diagnosis to death.

Results: Median follow-up was 31 months (range, 13-72 months). Brain metastasis developed at a median of 13 months after primary diagnosis (range, 3-69 months). Most brain metastases were supratentorial and solitary, with nine of 12 patients (75%) having solitary lesions and eight of 12 (67%) having supratentorial lesions. Surgical excision was performed in 9 patients (75%). Radiotherapy was administered locally (20-30 Gy, n=7) or to the craniospinal axis (21.6 or 23.4 Gy, n=2). Ten patients died during follow-up, 70% of whom did not experience cranial progression. Local control rate for irradiated lesions was 81.5% (median duration: 22 months). The brain metastasis-free survival was 71.4% (95% confidence interval [CI]: 2.59%-55.41%) with a median of 10 (range, 1-41) months. The 2-year survival rate was 58.3% (95% CI: 6.15%-71.84%). The 2-year overall survival rate after brain metastasis was 16.7% (95% CI: 7.13%-21.53%).

Conclusion: Brain metastasis remains rare in pediatric solid tumors. Most patients died from extracranial disease progression rather than cranial relapse. Radiotherapy can effectively relieve symptoms and may delay the progression of brain metastasis. Guidelines may help optimize the treatment of patients with brain metastasis.

儿童颅外实体瘤的脑转移:挑战罕见诊断的20年经验。
目的:探讨小儿颅内外实体瘤脑转移的临床特点、治疗及预后。材料与方法:回顾20年来我院放射治疗门诊收治的520例颅外实体瘤患儿,发现脑转移12例(2.2%)。原发肿瘤包括神经母细胞瘤(n=5)、骨肉瘤(n=3)、尤文氏肉瘤(n=2)、肾横纹肌样瘤(n=1)和肾母细胞瘤(n=1)。回顾性分析评价肿瘤及治疗特点。总生存率从最初诊断开始测量。计算脑转移后的总生存期、局部控制期和脑转移无进展生存期(从脑转移诊断至死亡)。结果:中位随访31个月(范围13-72个月)。脑转移发生在初次诊断后的中位13个月(范围3-69个月)。大多数脑转移灶为幕上和孤立灶,12例患者中有9例(75%)为孤立灶,12例患者中有8例(67%)为幕上灶。手术切除9例(75%)。局部放疗(20-30 Gy, n=7)或颅脊髓轴放疗(21.6或23.4 Gy, n=2)。10例患者在随访期间死亡,其中70%没有出现颅骨进展。放射病灶局部控制率为81.5%(中位持续时间22个月)。无脑转移生存期为71.4%(95%可信区间[CI]: 2.59%-55.41%),中位数为10个月(范围1-41个月)。2年生存率为58.3% (95% CI: 6.15% ~ 71.84%)。脑转移后2年总生存率为16.7% (95% CI: 7.13% ~ 21.53%)。结论:儿童实体瘤的脑转移仍然罕见。大多数患者死于颅外疾病进展,而不是颅内复发。放疗可有效缓解症状,并可延缓脑转移的进展。指南可能有助于优化脑转移患者的治疗。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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