Yoshihiro Tomoyasu, Tsuchihashi Kenji, Nio Kenta, Arita Shuji, Nakano Takafumi, Yasumatsu Ryuji, Jiroumaru Rina, Ariyama Hiroshi, Kusaba Hitoshi, Oda Yoshinao, Akashi Koichi, Baba Eishi
Department of Hematology, Oncology and Cardiovascular medicine Department of Comprehensive Clinical Oncology, Faculty of Medical Sciences Department of Otorhinolaryngology Department of Anatomical Pathology, Pathological Sciences, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.
Medicine (Baltimore). 2017 Nov;96(44):e8470. doi: 10.1097/MD.0000000000008470.
The multi-targeted tyrosine kinase inhibitors such as cediranib, sunitinib and pazopanib have been reported to be effective for alveolar soft part sarcoma (ASPS). The efficacy of pazopanib for the patient with lingual ASPS has yet to be reported.
A 23-year old man presented with articulation disorder and swelling of the tongue. Diagnosis of lingual ASPS was made after incisional biopsy and complete excision of the mass was performed. Three months later, he presented with a protruding mental region.
Computed tomography revealed mental region mass and lung metastasis.
After the failure of combination therapy of doxorubicin and ifosfamide, pazopanib was administered.
Shrinkage of both the mental region and lung mass continued for more than two months, but regrowth was confirmed at the fourth month.
Lingual ASPS is an exceedingly rare subset of ASPS with distinct molecular and histological characteristics and appropriate therapy remains to be established. Our findings suggest a possible therapeutic strategy for lingual ASPS.
据报道，多靶点酪氨酸激酶抑制剂如西地尼布、舒尼替尼和帕唑帕尼对肺泡软组织肉瘤（ASPS）有效。帕唑帕尼对舌部ASPS患者的疗效尚未见报道。
一名23岁男性出现言语不清和舌部肿胀。经切开活检后诊断为舌部ASPS，并对肿块进行了完整切除。三个月后，他出现颏部肿物。
计算机断层扫描显示颏部肿物和肺转移。
在阿霉素和异环磷酰胺联合治疗失败后，给予帕唑帕尼治疗。
颏部和肺部肿块缩小持续两个多月，但在第四个月证实有复发。
舌部ASPS是ASPS中极为罕见的一个亚型，具有独特的分子和组织学特征，仍有待确定合适的治疗方法。我们的研究结果提示了一种针对舌部ASPS的可能治疗策略。
