ArticleFrontiers in immunology2026
Recurrent anti-TIF1γ-positive dermatomyositis coexisting with postoperative parotid lymphoepithelial carcinoma: a case report with pathogenesis analysis.
Article in Frontiers in immunology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Background: Anti-TIF1γ-positive dermatomyositis (DM) is a classic paraneoplastic syndrome in adults, but its coexisting with lymphoepithelial carcinoma (LEC) of the parotid gland is exceptionally rare. This rarity poses significant challenges for clinical management. Objective: To report a unique case of parotid LEC emerging three years after a diagnosis of anti-TIF1γ-positive DM, followed by a post-oncologic DM recurrence. We aimed to investigate the underlying immunopathogenesis through peripheral blood mononuclear cell (PBMC) analysis and genetic profiling. Case presentation: A 28-year-old male presented with anti-TIF1γ-positive DM. Three years later, he developed parotid LEC, with Epstein-Barr virus (EBV) detected in both tumor tissue and serology. He was treated with surgical resection and adjuvant therapy, achieving a near-complete oncologic response. However, DM recurred eight months after the cancer diagnosis. Initial cyclophosphamide treatment was effective, but its withdrawal led to relapse; subsequent therapies with methotrexate and tofacitinib provided minimal benefit. Results: PBMC analysis during the DM recurrence revealed a highly active B-cell population and a reduction in cytotoxic cells. This B-cell expansion subsequently decreased 10 months later, suggesting a delayed effect of the documented EBV activation. Germline genotyping identified a panel of deleterious germline mutations in immune regulation genes, including a variant in Conclusion: This case illustrates that refractory anti-TIF1γ-DM can persist even after the associated malignancy is well controlled and underscore the need for long-term vigilance and personalized management strategies in paraneoplastic DM.
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