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Purpose <p>This systematic review summarizes the current literature on Warthin-like mucoepidermoid carcinoma (WL-MEC), highlighting its clinicopathological, immunohistochemical and molecular characteristics.</p> Methods <p>This study followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines and was registered by PROSPERO (protocol CRD42024523598). Case reports and case series published in English on WL-MEC of major and minor salivary glands were included. The search was conducted in MEDLINE/PubMed, Scopus, Web of Science, and Embase databases in November 2024, along with a search in grey literature. Twenty-one articles met the eligibility criteria. The extracted information included clinicopathological data, immunohistochemical and molecular tests, treatment, recurrence, metastasis, follow-up, and status. The data analysis was performed descriptively, and presented as absolute and relative frequencies, and means or median for numerical variables.</p> Results <p>78 WL-MEC cases were analyzed, with a female predominance observed and a wide age range, peaking in the sixth decade of life. Most cases involved the parotid gland and presented as indolent nodular masses, often asymptomatic. Histologically, the majority were low-grade tumors, with multicystic growth patterns and a lymphocytic infiltrate. Immunohistochemical analysis revealed consistent expression of p63, p40, CK5/6, and exhibited low Ki67 proliferation index (&lt; 5%). <i>MAML2</i> rearrangement was identified in all molecularly analyzed cases (<i>n</i> = 72/ 100%). Surgical treatment was the most common approach, which typically resulted in low recurrence rates, no distant metastasis, and favorable overall outcomes.</p> Conclusion <p>WL-MEC cases typically present as indolent, asymptomatic nodules in the parotid gland, often with a female predominance and a wide age range, peaking in the sixth decade of life. Surgery is the primary treatment and is associated with a favorable prognosis, characterized by low rates of recurrence and metastasis. These tumors commonly exhibit low-grade histopathological features, a multicystic pattern, and lymphocytic inflammatory infiltrate. The detection of <i>MAML2</i> rearrangement, typically via FISH, is an essential diagnostic tool, emphasizing the molecular similarities between WL-MEC and conventional MEC.</p>

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Clinicopathological Analysis of Salivary Glands Warthin-like Mucoepidermoid Carcinoma: A Systematic Review

  • 期刊论文

  • 发表日期：

    2025年5月8日

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  • Ricardo Anderson de Oliveira Vasconcelos,
  • Isaac Santos Araújo,
  • Luiz Miguel Ferreira,
  • João Paulo Gonçalves Paiva,
  • Maíra Medeiros Pacheco de Andrade,
  • Igor Fernandes,
  • Lucas Faria Abrahao-Machado,
  • Claudia Haydee Sarai Caro-Sánchez,
  • Ana Cano Cano-Valdez,
  • Adalberto Mosqueda-Taylor,
  • Ciro Dantas Soares

摘要

Purpose

This systematic review summarizes the current literature on Warthin-like mucoepidermoid carcinoma (WL-MEC), highlighting its clinicopathological, immunohistochemical and molecular characteristics.

Methods

This study followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines and was registered by PROSPERO (protocol CRD42024523598). Case reports and case series published in English on WL-MEC of major and minor salivary glands were included. The search was conducted in MEDLINE/PubMed, Scopus, Web of Science, and Embase databases in November 2024, along with a search in grey literature. Twenty-one articles met the eligibility criteria. The extracted information included clinicopathological data, immunohistochemical and molecular tests, treatment, recurrence, metastasis, follow-up, and status. The data analysis was performed descriptively, and presented as absolute and relative frequencies, and means or median for numerical variables.

Results

78 WL-MEC cases were analyzed, with a female predominance observed and a wide age range, peaking in the sixth decade of life. Most cases involved the parotid gland and presented as indolent nodular masses, often asymptomatic. Histologically, the majority were low-grade tumors, with multicystic growth patterns and a lymphocytic infiltrate. Immunohistochemical analysis revealed consistent expression of p63, p40, CK5/6, and exhibited low Ki67 proliferation index (< 5%). MAML2 rearrangement was identified in all molecularly analyzed cases (n = 72/ 100%). Surgical treatment was the most common approach, which typically resulted in low recurrence rates, no distant metastasis, and favorable overall outcomes.

Conclusion

WL-MEC cases typically present as indolent, asymptomatic nodules in the parotid gland, often with a female predominance and a wide age range, peaking in the sixth decade of life. Surgery is the primary treatment and is associated with a favorable prognosis, characterized by low rates of recurrence and metastasis. These tumors commonly exhibit low-grade histopathological features, a multicystic pattern, and lymphocytic inflammatory infiltrate. The detection of MAML2 rearrangement, typically via FISH, is an essential diagnostic tool, emphasizing the molecular similarities between WL-MEC and conventional MEC.