Sugita Yoshihiko, Hasegawa Hiromasa, Roy Rita R, Shimada Katsumitsu, Ochiai Takanaga, Nagayama Motohiko, Maeda Hatsuhiko
Department of Oral Pathology/Forensic Odontology, School of Dentistry, Aichi Gakuin University, Nagoya, JPN.
Hard Tissue Pathology Unit, Graduate School of Oral Medicine, Matsumoto Dental University, Shiojiri, JPN.
Cureus. 2025 Oct 11;17(10):e94365. doi: 10.7759/cureus.94365. eCollection 2025 Oct.
Lipoblastoma-like tumors (LLTs) are benign adipocytic tumors characterized by an admixture of lipoblasts within the myxoid stroma, primarily affecting the vulvovaginal area in young patients. Rare cases have been reported in other locations, including the orofacial region. Here, we present a unique case of LLT of the tongue in a 62-year-old woman with a slowly developing painless nodule. The tumor showed no signs of recurrence five years after surgery. Histopathological examination revealed a myxo-fatty lobulated lesion with numerous uni- and multi-vacuolated lipoblasts and prominent chicken-wire capillaries. Immunohistochemistry showed positivity for S100, whereas the tumor was negative for cluster of differentiation 34 (CD34), pleomorphic adenoma gene 1 (PLAG1), B-cell lymphoma 2(BCL-2), mouse double minute 2 homolog (MDM2), cyclin-dependent kinase 4 (CDK4), and DNA damage-inducible transcript 3 (DDIT3). The retinoblastoma protein (pRB) was partially negative and exhibited mosaic patterns. Fluorescence in situ hybridization (FISH) analysis indicated a hemizygous deletion of the locus in 36% of tumor cells, with no amplification detected. This case highlights the diagnostic challenges of differentiating LLTs from spindle cell lipomas (SCLs), particularly in the context of pRB deficiency and deletion. This case exemplifies the molecular heterogeneity of neoplasms with lipoblastoma-like features.
脂肪母细胞瘤样肿瘤（LLTs）是一种良性脂肪细胞肿瘤，其特征是在黏液样基质中存在脂肪母细胞混合，主要影响年轻患者的外阴阴道区域。其他部位包括口面部区域也有罕见病例报道。在此，我们报告一例62岁女性舌部LLT的独特病例，该患者有一个缓慢生长的无痛性结节。术后五年肿瘤无复发迹象。组织病理学检查显示为黏液样脂肪小叶状病变，有大量单泡和多泡脂肪母细胞以及明显的鸡笼状毛细血管。免疫组化显示S100阳性，而肿瘤细胞分化簇34（CD34）、多形性腺瘤基因1（PLAG1）、B细胞淋巴瘤2（BCL-2）、小鼠双微体2同源物（MDM2）、细胞周期蛋白依赖性激酶4（CDK4）和DNA损伤诱导转录本3（DDIT3）均为阴性。视网膜母细胞瘤蛋白（pRB）部分阴性并呈现镶嵌模式。荧光原位杂交（FISH）分析表明36%的肿瘤细胞中存在该位点的半合子缺失，未检测到扩增。该病例突出了鉴别LLTs与梭形细胞脂肪瘤（SCLs）的诊断挑战，特别是在pRB缺乏和缺失的情况下。该病例体现了具有脂肪母细胞瘤样特征的肿瘤的分子异质性。
