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Background <p>Parapharyngeal space (PPS) tumors are rare, comprising less than 1% of all head and neck neoplasms, with pleomorphic adenoma (PA) being the most common benign tumor in this region. Submandibular gland pleomorphic adenomas extending into the PPS are exceedingly rare. Carcinoma ex pleomorphic adenoma (CEPA) is an uncommon malignant transformation of PA, typically occurring after a long-standing or recurrent lesion.</p> Case Presentation <p>We report a rare case of a 36-year-old female presenting with a recurrent painless swelling in the left submandibular region, with extension into the PPS. She had a history of two prior excisions of PA in 2015 and 2021. Clinical examination and imaging revealed a well-defined lobulated mass extending from the submandibular gland into the PPS, displacing adjacent structures. FNAC was suggestive of myoepithelial cell-rich PA. Surgical excision was performed via a transcervical approach with preservation of vital neurovascular structures. Histopathological analysis confirmed the diagnosis of myoepithelial carcinoma ex pleomorphic adenoma.</p> Discussion <p>CEPA of the submandibular gland is rare, accounting for less than 15% of all CEPA cases. Malignant transformation risk increases with tumor duration and recurrence. Diagnosis based on FNAC is often challenging due to overlapping features with benign myoepithelial neoplasms. Complete surgical excision with clear margins remains the cornerstone of management, supplemented by radiotherapy in selected cases. Surgical approach must be tailored to tumor extent and location, with the transcervical route offering optimal access in this case.</p> Conclusion <p>This case underscores the rarity of malignant transformation of submandibular salivary gland tumour and its extension into parapharyngeal space. With need for early suspicion of malignant transformation in recurrent cases, and the need for a multidisciplinary approach in managing complex PPS tumors. Accurate diagnosis and timely surgical intervention are critical to improving outcomes in CEPA.</p>

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Carcinoma Ex Pleomorphic Adenoma of the Submandibular Gland with Parapharyngeal Extension: A Rare Case Report

  • 期刊论文

  • 发表日期：

    2025年8月8日

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  • Kiran Kumar,
  • Vijay Bidkar,
  • Nittika Garg,
  • K Khadeeja,
  • Sanskriti Jaiswal

摘要

Background

Parapharyngeal space (PPS) tumors are rare, comprising less than 1% of all head and neck neoplasms, with pleomorphic adenoma (PA) being the most common benign tumor in this region. Submandibular gland pleomorphic adenomas extending into the PPS are exceedingly rare. Carcinoma ex pleomorphic adenoma (CEPA) is an uncommon malignant transformation of PA, typically occurring after a long-standing or recurrent lesion.

Case Presentation

We report a rare case of a 36-year-old female presenting with a recurrent painless swelling in the left submandibular region, with extension into the PPS. She had a history of two prior excisions of PA in 2015 and 2021. Clinical examination and imaging revealed a well-defined lobulated mass extending from the submandibular gland into the PPS, displacing adjacent structures. FNAC was suggestive of myoepithelial cell-rich PA. Surgical excision was performed via a transcervical approach with preservation of vital neurovascular structures. Histopathological analysis confirmed the diagnosis of myoepithelial carcinoma ex pleomorphic adenoma.

Discussion

CEPA of the submandibular gland is rare, accounting for less than 15% of all CEPA cases. Malignant transformation risk increases with tumor duration and recurrence. Diagnosis based on FNAC is often challenging due to overlapping features with benign myoepithelial neoplasms. Complete surgical excision with clear margins remains the cornerstone of management, supplemented by radiotherapy in selected cases. Surgical approach must be tailored to tumor extent and location, with the transcervical route offering optimal access in this case.

Conclusion

This case underscores the rarity of malignant transformation of submandibular salivary gland tumour and its extension into parapharyngeal space. With need for early suspicion of malignant transformation in recurrent cases, and the need for a multidisciplinary approach in managing complex PPS tumors. Accurate diagnosis and timely surgical intervention are critical to improving outcomes in CEPA.