Expanding the histological spectrum of salivary gland neoplasms with HMGA2::WIF1 fusion emphasising their malignant potential: A report of eight cases Journal Article


Authors: Katabi, N.; Sukhadia, P.; DiNapoli, S. E.; Weinreb, I.; Hahn, E.; Ghossein, R.; Xu, B.
Article Title: Expanding the histological spectrum of salivary gland neoplasms with HMGA2::WIF1 fusion emphasising their malignant potential: A report of eight cases
Abstract: Aims: Recently, HMGA2::WIF1 fusion has been reported in pleomorphic adenoma (PAs) originating from the parotid gland with a characteristic canalicular adenoma (CAA)-like pattern. However, it is unclear whether HMGA2::WIF1 fusion may occur in salivary gland carcinoma or tumours originating from the minor salivary glands. We herein conducted a detailed clinicopathological review of eight salivary gland tumours harbouring HMGA2::WIF1 fusions. Methods and results: The reviewed diagnoses of salivary gland neoplasms with HMGA2::WIF1 fusion were PA (n = four), myoepithelioma (n = one), myoepithelial carcinoma ex PA (n = two) and high-grade carcinoma with basaloid features (n = one). Two tumours originated from the minor salivary glands. Six tumours (80%) contained areas reminiscent of CAA characterised by interconnected trabeculae/canaliculi of monotonous oncocytic or cuboidal tumour cells associated with a hypocellular, hyalinised to myxoid stroma. Areas typical of PA were seen in four (50%) cases. All tumours showed diffuse S100 and CK7 immunopositivity. Adverse events were detected in two cases, including local recurrence in a patient with PA, and local and distant recurrences and disease-related death in a patient with a high-grade carcinoma of the minor salivary gland of the buccal space, showing tumour necrosis and perineural invasion. Conclusion: Salivary gland neoplasms with HMGA2::WIF1 fusion are predominantly characterised by CAA/striated duct adenoma-like histology and a S100+/CK7+ immunoprofile. These tumours are not always benign, as among all reported cases approximately 20% showed malignancy (six of 28) and adverse outcome (three of 15), including recurrence, distant metastasis and disease-specific mortality. © 2023 John Wiley & Sons Ltd.
Keywords: case report; pathology; carcinoma; adaptor proteins, signal transducing; salivary gland tumor; salivary gland neoplasms; signal transducing adaptor protein; parotid gland; myoepithelial carcinoma; pleomorphic adenoma; adenoma, pleomorphic; humans; human; salivary gland neoplasm; hmga2::wif1 fusion; wif1 protein, human
Journal Title: Histopathology
Volume: 84
Issue: 2
ISSN: 0309-0167
Publisher: Wiley Blackwell  
Date Published: 2024-01-01
Start Page: 387
End Page: 398
Language: English
DOI: 10.1111/his.15074
PUBMED: 37849332
PROVIDER: scopus
PMCID: PMC10841865
DOI/URL:
Notes: Article -- Source: Scopus
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  1. Ronald A Ghossein
    483 Ghossein
  2. Nora Katabi
    304 Katabi
  3. Bin   Xu
    227 Xu