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<p>High-grade polymorphous adenocarcinoma (HG-PAC) is an exceptionally rare salivary gland malignancy, with very few cases reported in the sinonasal tract. A 74-year-old man presented with recurrence of a sinonasal tumor initially diagnosed as keratinizing squamous cell carcinoma. At recurrence, pathology showed a highly atypical, mitotically active neoplasm with solid growth and focal necrosis. Immunohistochemistry was positive for CK5, CK7, CK8/18, CD117, p16, p40, p63, and BCL2, with a Ki67 of 70%. Molecular testing excluded MYB rearrangement, common oncogenic variants (BRAF, EGFR, c-KIT, KRAS, PIK3CA), and high-risk HPV, supporting a diagnosis of HG-PAC. The patient underwent endoscopic-assisted resection followed by adjuvant radiotherapy and remains disease-free after 48 months. Literature confirms the rarity and diagnostic challenges of sinonasal HG-PAC and highlights the value of comprehensive pathological and molecular assessment. Despite aggressive features, long-term control is achievable with complete surgery and radiotherapy. This case adds to the limited evidence and supports multidisciplinary management.</p>

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High-Grade Polymorphous Adenocarcinoma, An Extremely Rare Entity: A Case Report and Literature Review

  • 期刊论文

  • 发表日期：

    2026年7月20日

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  • Gianluca Dalfino,
  • Letizia Neri,
  • Deborah Marchiori,
  • Maurizio Bignami

摘要

High-grade polymorphous adenocarcinoma (HG-PAC) is an exceptionally rare salivary gland malignancy, with very few cases reported in the sinonasal tract. A 74-year-old man presented with recurrence of a sinonasal tumor initially diagnosed as keratinizing squamous cell carcinoma. At recurrence, pathology showed a highly atypical, mitotically active neoplasm with solid growth and focal necrosis. Immunohistochemistry was positive for CK5, CK7, CK8/18, CD117, p16, p40, p63, and BCL2, with a Ki67 of 70%. Molecular testing excluded MYB rearrangement, common oncogenic variants (BRAF, EGFR, c-KIT, KRAS, PIK3CA), and high-risk HPV, supporting a diagnosis of HG-PAC. The patient underwent endoscopic-assisted resection followed by adjuvant radiotherapy and remains disease-free after 48 months. Literature confirms the rarity and diagnostic challenges of sinonasal HG-PAC and highlights the value of comprehensive pathological and molecular assessment. Despite aggressive features, long-term control is achievable with complete surgery and radiotherapy. This case adds to the limited evidence and supports multidisciplinary management.