Rayan Rammal, Bethany Batson, Matthew E Spector, Simion I Chiosea, Raja R Seethala
{"title":"模仿 \"肉瘤外型腺瘤 \"的带高级别鳞状腺体和软骨肉瘤样变的针状细胞癌：肉瘤性唾液腺肿瘤命名修订提案中的一个难题。","authors":"Rayan Rammal, Bethany Batson, Matthew E Spector, Simion I Chiosea, Raja R Seethala","doi":"10.1007/s12105-024-01650-5","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":null,"pages":null},"PeriodicalIF":3.2000,"publicationDate":"2024-05-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11111628/pdf/","citationCount":"0","resultStr":"{\"title\":\"Acinic cell Carcinoma with high-grade Squamoglandular and Chondrosarcomatous Transformation Mimicking 'Carcinosarcoma ex-pleomorphic Adenoma': A Wrinkle in the Proposed Nomenclature Revision for Sarcomatoid Salivary Gland Neoplasms.\",\"authors\":\"Rayan Rammal, Bethany Batson, Matthew E Spector, Simion I Chiosea, Raja R Seethala\",\"doi\":\"10.1007/s12105-024-01650-5\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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. 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引用次数: 0
摘要
虽然针状细胞癌（AciCC）可发生高级别转化（HGT），变为高级别腺癌或分化不良癌，但纺锤形细胞/肉瘤样癌等其他形态却很罕见，且特征不清。我们在此报告了一例新的AciCC病例，其鳞状腺体和软骨肉瘤样HGT模仿了所谓的 "癌肉瘤前多形腺瘤"。患者是一名81岁的男性，有两个月的颈部肿胀和耳痛病史，出现左侧咽旁间隙肿块并延伸至咽后间隙和翼状肌。切除后，肿瘤显示出相当大的形态多样性，既有高级别浆液性和粘液性尖锐湿疣成分，也有楔形至实性尖锐湿疣样成分，并伴有瘤坏死和鳞状分化，所有这些成分都被包埋在软骨瘤样背景中，软骨瘤样背景从白细胞和平淡无奇到高级别软骨肉瘤/褶皱肉瘤样外观不等。只有少量传统的AciCC成分。免疫印迹显示 AR 阴性，GCDFP-15 仅局部阳性，这与真正的腺分泌表型不符，而 PLAG1 和 HMGA2 阴性，这与先天性多形性腺瘤不符。另一方面，SOX-10、DOG-1 和舒张酶后的 PAS 突出了浆液性尖锐湿疣分化，而粘液胭脂红和 NKX3.1 则突出了粘液性尖锐湿疣分化。NR4A3免疫组化染色和NR4A3荧光原位杂交在癌变和肉瘤样成分中均呈阳性，而这两种成分的测序分析表明，TP53、PIK3CB、ARID1A和STK11发生了相同的改变。这一独特的病例表明，将所有具有异源成分的唾液肉瘤样癌归入 "癌前多形性腺瘤 "家族需要慎重。
Acinic cell Carcinoma with high-grade Squamoglandular and Chondrosarcomatous Transformation Mimicking 'Carcinosarcoma ex-pleomorphic Adenoma': A Wrinkle in the Proposed Nomenclature Revision for Sarcomatoid Salivary Gland Neoplasms.
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.
期刊介绍：
Head & Neck Pathology presents scholarly papers, reviews and symposia that cover the spectrum of human surgical pathology within the anatomic zones of the oral cavity, sinonasal tract, larynx, hypopharynx, salivary gland, ear and temporal bone, and neck.
The journal publishes rapid developments in new diagnostic criteria, intraoperative consultation, immunohistochemical studies, molecular techniques, genetic analyses, diagnostic aids, experimental pathology, cytology, radiographic imaging, and application of uniform terminology to allow practitioners to continue to maintain and expand their knowledge in the subspecialty of head and neck pathology. Coverage of practical application to daily clinical practice is supported with proceedings and symposia from international societies and academies devoted to this field.
Single-blind peer review
The journal follows a single-blind review procedure, where the reviewers are aware of the names and affiliations of the authors, but the reviewer reports provided to authors are anonymous. Single-blind peer review is the traditional model of peer review that many reviewers are comfortable with, and it facilitates a dispassionate critique of a manuscript.
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