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索拉非尼治疗伴有弥漫性结节性肺病需要机械通气的儿童甲状腺癌患者的反应。

Response to sorafenib in a pediatric patient with papillary thyroid carcinoma with diffuse nodular pulmonary disease requiring mechanical ventilation.

机构信息

1 Endocrinology & Diabetes, All Children's Specialty Physicians, All Children's Hospital/Johns Hopkins Medicine , St. Petersburg, Florida.

出版信息

Thyroid. 2014 Jan;24(1):169-74. doi: 10.1089/thy.2012.0468. Epub 2013 Jul 25.

Abstract

BACKGROUND

Papillary thyroid cancer (PTC) presents with local and distant metastases more frequently in children than in adults. However, hypoxemia secondary to pulmonary metastases has not been reported in the literature. Sorafenib is a small-molecule multikinase inhibitor used in radioactive iodine ((131)I)-refractive papillary thyroid carcinoma.

PATIENT FINDINGS

An eight-year-old boy presented with fever and hypoxemia and was found to have H1N1 viral pneumonia requiring mechanical ventilation and antiviral and glucocorticoid treatment. After initial improvement, he was readmitted one month later because of persistent hypoxemia. A high-resolution computed tomography of the chest and neck revealed multiple 1-4 mm nodules and necrotic lymph nodes on the right side of the neck. Left lung wedge resection and right deep cervical node biopsies were consistent with PTC. He underwent a total thyroidectomy with a central neck node dissection and postoperatively required prolonged mechanical ventilation. Due to the inability to be weaned from mechanical ventilation, treatment with sorafenib 200 mg daily (10 mg/kg/day, 250 mg/m(2)) was initiated. Eight days thereafter, ventilation support was discontinued and the child was extubated. However, he failed extubation secondary to supraglottic edema, and seven days later tracheostomy was performed. The dosage of sorafenib was increased to 200 mg twice daily (20 mg/kg/day, 500 mg/m(2)). After 52 days of therapy, a repeat computed tomography scan showed reduction in the lung nodule size to 1-2 mm. He underwent (131)I therapy 87 days after sorafenib was started. A post-treatment scan showed extensive uptake throughout the lungs and thyroid bed, supraclavicular nodes, and cervical nodes.

CONCLUSIONS

This is the first reported case of a pediatric patient with respiratory failure possibly secondary to diffuse micronodular PTC requiring mechanical ventilation and subsequent delay in definitive therapy. Sorafenib could be considered for gap therapy when (131)I therapy cannot be administered in a timely manner. Treatment with this multikinase inhibitor does not seem to adversely affect the uptake of (131)I in radiation-naïve patients.

摘要

背景

与成人相比,儿童甲状腺乳头状癌 (PTC) 更常出现局部和远处转移。然而,文献中并未报道过肺转移引起的低氧血症。索拉非尼是一种小分子多激酶抑制剂,用于治疗放射性碘 ((131)I) 难治性甲状腺乳头状癌。

病例发现

一名 8 岁男孩因发热和低氧血症就诊,被诊断为 H1N1 病毒性肺炎,需要机械通气和抗病毒及糖皮质激素治疗。初始治疗后病情改善,但一个月后因持续低氧血症再次入院。高分辨率胸部和颈部计算机断层扫描显示右侧颈部多个 1-4 毫米大小的结节和坏死性淋巴结。左肺楔形切除术和右侧深部颈淋巴结活检均符合 PTC。患者接受了甲状腺全切除术和中央颈部淋巴结清扫术,术后需要长时间机械通气。由于无法脱离机械通气,开始每天服用 200 毫克索拉非尼(10 毫克/公斤/天,250 毫克/平方米)。8 天后,停止机械通气,患儿拔管。但由于声门上水肿导致拔管失败,7 天后进行了气管切开术。索拉非尼的剂量增加到每天两次 200 毫克(20 毫克/公斤/天,500 毫克/平方米)。治疗 52 天后,重复计算机断层扫描显示肺部结节大小缩小至 1-2 毫米。在开始使用索拉非尼 87 天后进行 (131)I 治疗。治疗后扫描显示肺部和甲状腺床、锁骨上淋巴结和颈部淋巴结广泛摄取。

结论

这是首例报道的儿童呼吸衰竭病例,可能由弥漫性微结节性 PTC 引起,需要机械通气,随后延迟进行确定性治疗。当无法及时进行 (131)I 治疗时,可以考虑索拉非尼进行间隙治疗。在未接受过放疗的患者中,这种多激酶抑制剂的治疗似乎不会对 (131)I 的摄取产生不利影响。

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