Rammal Rayan, Batson Bethany, Spector Matthew E, Chiosea Simion I, Seethala Raja R
Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, PA, USA.
Department of Otolaryngology, University of Pittsburgh Medical Center, Pittsburgh, PA, USA.
Head Neck Pathol. 2024 May 22;18(1):44. doi: 10.1007/s12105-024-01650-5.
While acinic cell carcinoma (AciCC) can undergo high-grade transformation (HGT) to high-grade adenocarcinoma or poorly differentiated carcinoma, other morphologies such as spindle cell/sarcomatoid carcinoma are rare and not well-characterized. We herein report a novel case of AciCC with squamoglandular and chondrosarcomatous HGT mimicking a so-called 'carcinosarcoma ex-pleomorphic adenoma'. The patient is an 81-year-old male with a two-month history of neck swelling and referred otalgia who presented with a left parapharyngeal space mass extending into retropharyngeal space and pterygoid muscles. On resection, the tumor showed considerable morphologic diversity with high-grade serous and mucous acinar components as well as cribriform to solid apocrine-like components with comedonecrosis and squamous differentiation, all of which were embedded in a chondromyxoid background ranging from paucicellular and bland to a high-grade chondrosarcoma/pleomorphic sarcoma-like appearance. Only a minor conventional AciCC component was noted. Immunostains were negative for AR and only focally positive for GCDFP-15 arguing against a true apocrine phenotype, while PLAG1 and HMGA2 were negative arguing against an antecedent pleomorphic adenoma. On the other hand, SOX-10, DOG-1 and PAS after diastase highlighted serous acinar differentiation, and mucicarmine, and NKX3.1 highlighted mucous acinar differentiation. NR4A3 immunohistochemical staining and NR4A3 fluorescence in situ hybridization were positive in the carcinomatous and sarcomatoid components while sequencing analysis of both components revealed identical alterations involving TP53, PIK3CB, ARID1A, and STK11. This unique case warrants caution in designating all salivary sarcomatoid carcinomas with heterologous elements as part of the 'carcinoma ex-pleomorphic adenoma' family.
虽然腺泡细胞癌(AciCC)可发生高级别转化(HGT)为高级别腺癌或低分化癌,但其他形态,如梭形细胞/肉瘤样癌则较为罕见且特征不明。我们在此报告一例具有鳞状腺泡和软骨肉瘤样HGT的AciCC新病例,其类似所谓的“多形性腺瘤恶变肉瘤”。该患者为81岁男性,有两个月颈部肿胀和牵涉性耳痛病史,就诊时发现左咽旁间隙肿块延伸至咽后间隙和翼内肌。切除标本显示肿瘤具有显著的形态学多样性,有高级别浆液性和黏液性腺泡成分,以及筛状至实性大汗腺样成分,并伴有粉刺样坏死和鳞状分化,所有这些成分均包埋于从少细胞、平淡到高级别软骨肉瘤/多形性肉瘤样外观的软骨黏液样背景中。仅发现少量传统AciCC成分。免疫组化结果显示AR阴性,GCDFP - 15仅局灶阳性,不支持真正的大汗腺表型,而PLAG1和HMGA2阴性,不支持之前存在多形性腺瘤。另一方面,淀粉酶消化后的SOX - 10、DOG - 1和PAS突出了浆液性腺泡分化,黏液卡红和NKX3.1突出了黏液性腺泡分化。NR4A3免疫组化染色和NR4A3荧光原位杂交在癌性和肉瘤样成分中均为阳性,而对这两种成分的测序分析显示涉及TP53、PIK3CB、ARID1A和STK11的相同改变。这一独特病例警示在将所有具有异源性成分的唾液腺肉瘤样癌指定为“多形性腺瘤恶变”家族的一部分时应谨慎。